International Journal of Medical Reviews and Case Reports (Int J Med Rev Case Rep) : Year: 2017, Volume: 1, Issue: 1-3, PP: 1-55 ISSN: 2534-9821 - Editor-in-Chief: Dr. Ivan Inkov

Table of Cont ent s - Int J Med Rev Case Rep.
Year: 2 0 1 7 , Volume: 1 , Issue: 1 -3 , pp: 1 -5 5
ISSN: 2 5 3 4 -9 8 2 1 – Edit or-in-Chief: Dr. Ivan Inkov
Review Art icle
1 . Los hombres y los accident es de t ránsit o: un vist azo al riesgo, conduct a de riesgo,
not ificación de accident es, educación y formación profesional
Cint ia Rodrigues, Thiago Vendramini, Carlos Górios, Cleo Chinaia, Rodrigo Armond, Jane
Armond, Pat ricia Colombo-Souza, Elias Jirjoss Ilias
Int J Med Rev Case Rep. 2 0 1 7 ; 1 ( 1 ) : 1 -5
» Abstract » PDF» doi: 10.5455/IJMRCR.los-accidentes-de-transito
2 . Kikuchi-Fujimot o disease: case report and review
Irami Araujo-Filho, Art ur Dant as Freire, Irami Araújo-Net o, Carolina Chianca Dourado
Lemos, Let ícia Araújo Cost a Uchôa, Amália Cínt hia Meneses Rêgo, Marco Ant ônio Bot elho
Soares, José Francisco Correia-Net o, Carlos André Nunes Jat obá, Ana Maria de Oliveira
Ramos
Int J Med Rev Case Rep. 2 0 1 7 ; 1 ( 1 ) : 6 -1 0
» Abstract » PDF» doi: 10.5455/IJMRCR.kikuchi-fujimoto-disease
Case Report
3 . Pancreas Divisum Causing Recurrent Pancreat it is: A Case Report

Nadeem Ahmed Siddiqui, Rizwan Sult an, Fareed Ahmed Shaikh
Int J Med Rev Case Rep. 2 0 1 7 ; 1 ( 1 ) : 1 1 -1 3
» Abstract » PDF» doi: 10.5455/IJMRCR.pancreas-divisum-recurrent-pancreatitis
4 . Vulvar non-filarial elephantiasis associat ed wit h scabies
Sarah Sabur, Saoussane Bounajma, Mounia Nasr, Samir Mazouz
» Abstract » PDF» doi: 10.5455/IJMRCR.vulvar-non-filarial-elephantiasis-scabies
5 . Pit uit ary microadenoma wit h prolact in, cort icot ropic and t hyreot ropic deficiency: from
infert ilit y t o pregnancy : About a case.
Diallo Moussa, Koulimaya Cyr Esperence Gombet , Diallo Ast ou Coly Niassy, Diouf Abdoul
Aziz, Gassama Omar, Gueye Mame Diarra Ndiaye, Leye Yakham, Diouf Alassane
» Abstract » PDF» doi: 10.5455/IJMRCR.pituitary-microadenoma
6 . Surgical Management Of Traumat ic Manubrio-St ernal Dislocat ion Wit h Locking Compression
Plat e; A Case Report And Review Of Lit erat ure.
Fareed Ahmed Shaikh, Syed Shahabuddin, Haroon Rashid, Noman Shahzad
» Abstract » PDF» doi: 10.5455/IJMRCR.surgical-management-traumatic-manubrio-sternal-dislocation
Review Art icle
7 . Test icular Descend, How and Why: A Review Art icle

Sujan Narayan Agrawal
» Abstract » PDF» doi: 10.5455/IJMRCR.testicular-descend
Case Report
8 . Efficacy of Int ravenous immunoglobulin and phot ot herapy in t he management of ext reme-
hyperbilirubinaemia: A Case Report
Oluf unke Bosede Bolaji, Sandeep Dhamaraj, Colin Lumsden, Olusegun Joseph Adebami
» Abstract » PDF» doi: 10.5455/IJMRCR.immunoglobulin-and-phototherapy-in-the-management-of-
extreme-hyperbilirubinaemia
9 . Surgical management of severe t rauma involving group of muscles around t he brisket of

Dongola breed of horse:A case report
Abayomi Kayode Olaif a, Cecelia Omowunmi Ogunt oye, Adenike Olat unji Akioye
» Abstract » PDF» doi: 10.5455/IJMRCR.dongola-breed-of-horse-severe-trauma-surgery
1 0 . A case report of delayed diagnosis of ut erine rupt ure following vaginal delivery
Vanessa Falé Rosado, Sara Rocha, Cat arina Vasconcelos, Maria Luisa Mart ins, Maria José
Alves
» Abstract » PDF» doi: 10.5455/IJMRCR.uterine-rupture-delayed-diagnosis
1 1 . Misdiagnosis of an Acardiac Twin in t he First Trimest er, A Case Report .

Cat arina Vasconcelos, Vanessa Falé Rosado, Álvaro Cohen, Ana Teresa Mart ins, Rit a Torres
» Abstract » PDF» doi: 10.5455/IJMRCR.acardiac-twin-first-trimester
1 2 . Spider procedure for sacral pressure sore reconst ruct ion:A case report .
Sarah Sabur, Imane El Aissaoui, Mohammed Raboune, Lamiaa Bensaida, Samir Mazouz,
Noureddine Gharib, Abdellah Abassi
» Abstract » PDF» doi: 10.5455/IJMRCR.Spider-procedure-for-sacral-pressure-sore-reconstruction
Review Art icle
1 3 . Met a Analysis Of Ant iepilept ic Drugs Induced Choreoat het osis In Paediat ric Pat ient s
Sriram Shanmugam, Lidhu Daniel, Jaleel Ahamed
» Abstract » PDF» doi: 10.5455/IJMRCR.ANTIEPILEPTIC-DRUGS-INDUCED-CHOREOATHETOSIS
Case Report
1 4 . Case report of Dengue encephalopat hy in pregnancy mimicking at ypical eclampsia – An

insight of neurological manifest at ions of dengue in pregnancy
Sasirekha Rengaraj, Sut hrasika Thiyagalingam
» Abstract » PDF» doi: 10.5455/IJMRCR.dengue-encephalopathy-in-pregnancy-mimicking-atypical-
eclampsia
REVIEW ARTICLE
LOS HOMBRES Y LOS ACCIDENTES DE TRÁNSITO:

UN VISTAZO AL RIESGO, CONDUCTA DE RIESGO,
NOTIFICACIÓN DE ACCIDENTES, EDUCACIÓN Y
FORMACIÓN PROFESIONAL
Cintia Leci Rodrigues∗,1 , Thiago Leão Vendramini∗∗ , Carlos Górios∗ ∗ ∗ , Cleo Chinaia♦ , Rodrigo de Eston Armond♦♦ , Jane de
Eston Armond♦♦♦ , Patricia Colombo-Souza4 and Elias Jirjoss Ilias44
∗ Maestríaen Salud Pública FSP/USP. Curso de Medicina de la Faculdade das Américas., ∗∗ Especialista en alcohol y drogas FMUSP/USP., ∗ ∗ ∗ Mastría en
Ortopedia y Traumatología USP. Curso de Medicina del Centro Universitario São Camilo., ♦ Especialista en Gestión de Servicios de Enfermería UNIFESP.
Curso de Medicina y Enfermería de la Universidade de Santo Amaro., ♦♦ Cursando maestría en Ciencias de la Salud de la Universidade de Santo Amaro.,
♦♦♦ Doctora en Salud Pública FSP/USP. Stricto sensu Ciencias de la Salud de la Universidade de Santo Amaro., 4 Doctora en Nutricion UNIFESP. Stricto
sensu Ciencias de la Salud de la Universidade de Santo Amaro., 44 Doctorado en Medicina (Cirugía) de la Facultad de Ciencias Médicas de la Santa Casa de
São Paulo. Departamento de Cirugía, Facultad de Ciencias Médicas de la Santa Casa de São Paulo.
ABSTRACT Los accidentes de tránsito son un grave y complejo problema de salud pública. La literatura señala que los
accidentes de tránsito están asociados confactores de comportamiento, seguridad de los vehículos y la precariedad del
espacio urbano.Reducir el número de accidentes de tránsito es un reto para los gestores deárea. La educación en el tránsito
debe considerar la vigilancia como una estrategia eficaz para cambiar el comportamiento del conductor,principalmente
en relación con el exceso de velocidad y el consumo de alcohol asociado con la conducción.La ingeniería tiene un papel
importante para promover un entorno seguro,en el que la convivencia de peatones, ciclistas y conductores sea posible. El
escenario en que se configura los accidentes de tránsito muestra la necesidad de los profesionales de la salud reconsiderar
sus prácticas con el fin de replantear la imagen de la víctima de accidentes, las formas de trabajo de prevención de
accidentes, la educación en el tránsito de peatones y conductores, con el fin de promover la salud y la cultura de paz.
Existe la necesidad de políticas de salud públicay estrategias que hacen posible acceso a las acciones tanto en el campo
preventivo como en el campo de la rehabilitación.
KEYWORDS accidentes de tránsito, prevención de accidentes, educación, notificación de accidentes.
Introducción:
Los accidentes de tránsito son un grave y complejo problema de

salud pública. Han aumentado con el desarrollo económico y
tecnológico de las sociedades modernas y pueden tener impli-
Copyright © 2017 by the Bulgarian Association of Young Surgeons
caciones sociales y económicas, ya que predominan en la gente
DOI: 10.5455/IJMRCR.los-accidentes-de-transito joven y económicamente activa[1,2].
First Received: April 24, 2017 De conformidad, en São Paulo, durante el año 2014, los ac-
Accepted: May 11, 2017
Manuscript Associate Editor: George Baytchev (BG)
cidentes de tránsito fueron responsables del 2% de las muertes
Editor-in Chief: Cvetanka Hristova (BG) totales[3].
Reviewers: Ivan Inkov (BG) La bibliografía señala que los accidentes de tránsito están
1
Cintia Leci Rodrigues.Dirección: Calle Professor Cândido Nogueira da Mota Nº 409
Barrio Interlagos, São Paulo – SP, Brasil C.P. 04786-035. Correo electrónico:
relacionados con factores de comportamiento, seguridad de los
[email protected] vehículos y la precariedad del espacio urbano. Se configuran-
Cintia Leci Rodrigues et al./ International Journal of Medical Reviews and Case Reports (2017) 1(1): 1-5
como causas importantes de mortalidad y la morbilidad debido realicen programas de prevención e intervención, como no con-
al creciente número de vehículos, los cambios en el estilo de vida ducir en estado de ebriedad constantemente, las investigaciones
y el comportamiento de riesgo en la población general[2, 4,5]. muestran que los accidentes de tránsito provocados por conduc-
Los factores asociados con accidentes de tránsito, implícita tores en estado de ebriedad siguen presentando tasas altas en
o explícitamente, son:el hombre; vehículo; vía y entorno; y en todo el mundo[9, 10]. Otros factores juntos y posibles tales como
relación con la legislación y su cumplimiento. Según Almeida somnolencia, fatiga, estrés/ ansiedad, el uso de medicamentos
y Cols[5] el desglose de estos factoresy el estudio de sus aso- (inadecuados y asociados con las bebidas alcohólicas)y la sensi-
ciaciones son necesarios para comprendery mejor intervenir en bilidad individual (susceptibilidad neurofuncional) combinados
fenómeno accidente de tránsito. Es que su combinación puede aumentan el riesgo de accidentes de tránsito[11].
aumentar la probabilidad de ocurrencia de los accidentesde
El uso de alcohol sugiere la baja percepción de riesgo de los
manera diferente en ciertos lugares[5].
conductores. Los motivos de salud públicaque pueden subrayar
En este contexto, el objetivo de esta revisión fue preparar
las medidas preventivasson mayor vigilancia y leyes estrictas.La
una reflexión sobre la importancia de los accidentes de tránsi-
posición tolerante de algunas sociedades en relación con el con-
toen el ámbito de la salud pública y, más en concreto, sobre la
sumo de alcohol- que es intensificado por fuertes campañas
subjetividad en la evaluación de riesgo de los conductoresy su
publicitarias[12].
posible influencia en la aplicación de medidas de prevención de
accidentes de tránsito. Estudio de revisión sistemática realizada por Aguilera y
cols[13] en relación con la legislación, leyes que establecen
Los accidentes de tránsito: la percepción de riesgo de los límites de concentración de alcohol en la sangre no fueron efi-
conductores caces, sobre todo cuando no estén acompañados de otras me-
Reducir el número de accidentes de tránsito es un reto para didas.Estas leyes tuvieron un impacto únicamente cuando se
los gestores de área[6].La gestión del tránsito es un asunto asocia con la suspensión de la licencia del infractor o prohibi-
complejo.El sistema de transporte prioriza el uso de vía ter- ciones más coercitivas. La vigilancia tiene un efecto significativo
restrey en consecuencia favorece el movimiento y la expansión en la reducción de violaciones por conducir bajo los efectos del al-
del número de los automóviles. Este hecho,combinado con la cohol,por ejemplo, con el aumento de las operaciones policiales.
falta de infraestructura (aceras, vías, señalización), y otros prob- Los países con larga tradición en la vigilancia del conductor en
lemas en el transporte (costo, la capacidad de los medios de estado de ebriedad, con leyes que establecen un límite bajo de
transporte, la contaminación, la congestión)forman un escenario concentración de alcohol en la sangre, y donde los conductores
favorable para el aumento de las tasas de mortalidad y morbili- están en alto riesgo de ser sorprendido en el sistema de vigi-
dad.También tenemos que considerar que el tránsito se compone lancia y detención, cuyo trabajo está apoyado por los medios
de personasy como tal hay que tener en cuentasu individuali- de comunicación, tienden a tener un bajo número de casos que
dad y el desarrollo constante,sus procesos que se componen de involucren alcohol y la conducción[13].
las funciones y actividades diarias,su contexto y el momento Como se ha visto los accidentes de tránsito se han convertido
histórico en que está insertada[7]. como un problema de salud pública, en vista de altos costos fi-
El riesgo podría definirse como la probabilidad de que ocurra nancieros (hospitalización, rehabilitación de las víctimas)y social
algo indeseable.Y los factores de riesgo son elementos con alta que resultan de ellos y, en cuanto a la discusión sobre conducir
probabilidad de desencadenaro estar asociado con el desencade- bajo los efectos del alcohol se ha formateado como un problema
namiento de un acontecimiento no deseado, no siendo necesari- que requiere políticas públicas. El aumento de los accidentes pro-
amente elemento causal. Por el contrario, los factores de protec- porciona una proyección esencialpara que este asunto se hiciera
ción son los recursos personales o socialesque tendría el papel público en los medios de comunicación. Estos se nutren de la in-
de reducir o neutralizar el impacto del riesgo.La percepción de vestigación, los datos estadísticos de organismos de tránsito, por
riesgo tendría la perspectiva del control preventivo de riesgos, y estudiosos sobre el tema, campañas sociales, y otros que trabajan
a través de la educación, influiría en el comportamiento perjudi- con el problema del alcohol y los accidentes de tránsito.
cial para la salud de la persona y el entorno en que viven [7,8].
El comportamiento del conductor se identificó como uno de los Según Almeida[14] hay una tensión en algunos campos de
factores responsables de los accidentes de tránsito con respecto a la actividad donde los intereses en conflicto están presentes en
la observación de señales de tránsito, velocidad, y las decisiones el tema de conduciren estado etílico. Por un lado, el problema
en el momento de adelantar a otro vehículoo cruzando una vía. de salud pública, por otra parte, la defensa del mercado libre y
Se necesitan estudios sobre culturas y las condiciones de vida lo- autorregulado.También debe analizar el papel de los medios de
calespara la comprensión de las actitudes de los conductores,con comunicación en este proceso, la regulación insuficiente de la
el fin de desarrollar programas de capacitación, rehabilitación y publicidad de alcohol, principalmente entre los países en desar-
educación[8]. rollo y el cambio de posición en relación con el uso y la venta de
bebidas alcohólicascomo un asunto de libre mercado[14].
El uso de alcohol y otras drogas entre los conductores: La eficacia de las leyes de tránsito requiere una vigilancia
Los conductores que conducen bajo la influencia del alcohol son eficaz junto con enfoques informativos/ educativos. Por lo tanto,
una de las principales causas de accidentes de tránsito, de tal su efectividad depende,por una parte, la percepción inmediata
modo que la ingesta de pocas dosis alcohólicas es suficientepara de la pena para los conductores. Aguilera y cols13mencionan
aumentar la predisposición del conductor para emitir alguna que la prueba de alcohol en la sangre obligatoriay el programa
conducta de riesgo[9]. de denuncia de conductor ebriomostraron una reducción de
El tema relacionado con los accidentes de tránsito provoca- las colisiones, sin embargo revelan que el conductor cambia el
dos por conductores en estado de ebriedadempieza a tener una comportamiento sólo cuando el siente que está siendo vigilado
dimensión más amplia en la bibliografía.Por un lado, aunque se por la autoridad pública u observado por la sociedad[13].
El comportamiento de riesgo: velocidad, violaciones de leyes, sentido, contribuye a la consolidación de la red de atención
el tiempo de licencia: completa a las víctimas de accidentes[18].
La educación siempre ha sido considerada una de las principales
estrategias de prevención.Sin embargo, el papel de la educación Educación vial:
en la prevención del tránsito ha sido reevaluado, con la vigilan- La educación vial debe considerar la vigilancia como una es-
cia como una estrategia más probable de impactoy la ingeniería trategia eficaz para cambiar el comportamiento del conductor,
la más eficaz para asegurar velocidades más lentas, por los cam- especialmente en relación con el exceso de velocidad y consumo
bios físicos en las vías (badenes, pasos de peatones elevados, de alcohol asociado con la conducción.La ingeniería tiene un pa-
ciclovías, entre otros)[13]. pel importante para promover un entorno seguro en el que sea
Iniciativas de normativa muestran cambios de conducta como posible la convivencia de peatones, ciclistas y conductores.Por
por ejemplo,límite de velocidad y uso restringido del teléfono último, la educación tenía carácter informativo y de apoyo para
celular disminuyeronal mismo tiempo que la reducción de las otras estrategias utilizadas[13].
tasas de colisiones y lesiones[13]. Se entiende que los accidentes de tránsito se consideran un
Varios autores, afirman que las intervenciones dirigidas a problema de salud pública responsable de generar altos costos
reducir la velocidad del tránsito se consideran esenciales para para el sector de la salud y las consecuencias físicas y/ o psi-
la prevención de los accidentes de tránsito.Entre esas, podemos cológicas para las personas que sufren de este tipo de accidentes,
mencionar la implementación de radares de control de veloci- además de las pérdidas sustanciales de las personas jóvenes que
dad.El uso de radar contribuye a la reducción de la velocidad forman parte de la población activa, debido a la muerte o la
media, disminución del porcentaje de vehículos que transitan invalidez como consecuencia de estos accidentes[19].
en el exceso de velocidady la reducción de la velocidad desar- Para mitigar esta realidad, se observa la necesidad de una
rollada por los conductores.Uno de los problemas asociados aplicación más estricta de las leyes de tránsito asociado a la
con la aplicación de detectores de velocidad es la tendencia de inversión en la educación de los peatones y conductores de
algunos conductores para frenar al pasar por el radar y, luego, vehículos[19].
superar el límite de velocidad cuando se está fuera del alcance
del mismo[13, 14,15]. Formación de profesionales para hacer frente a los accidentes
de tránsito:
La notificación de los accidentes de tránsito: Uno de los principales retos en el área de los accidentes de trán-
Al considerar las unidades de salud, incluidas las unidades de sito en Brasil es precisamente mejorar la práctica profesional en
emergencia como los "serviços sentinelas", los profesionales de la salud para incluir los profesionales, para sostener científica-
estas unidades deben ser capaces de identificar y llevar a cabo mente la aplicación, el seguimiento, la evaluación o elaboración
los procedimientos y encaminamientos asociados con casos de de políticas públicas de tránsito con el fin de intervenir en el
violencia, sobre todo con respecto a la notificación en el "Sistema comportamiento de los usuarios del tránsito y del transporte, ya
de Vigilância a Violências e Acidentes (VIVA)", del Ministerio sean peatones o conductores[20].
de Salud, en Brasil.Este sistema tiene por objeto determinar el Se resalta la importancia de la formación permanente de los
alcance y el perfil de las causas externas asistida, lo que permite profesionales de la salud que trabajan en el área de emergencia,
la aproximación de la situación real, especialmente en casos de puesto que cuanto más rápida y cualificada sea la primera asis-
lesiones menores que no determinaron la muerte u hospital- tencia, mayor será la probabilidad de un buen pronóstico[19].
ización, teniendo en cuenta el fuerte impacto de estos eventos El escenario que se configura los accidentes de tránsito mues-
en la salud, así como la gran demanda de estos casos en las tra la necesidad de profesionales de la salud de replantear sus
unidades de emergencia[16]. prácticas con el fin de redefinir la imagen de la víctima de acci-
En la ciudad de São Paulo, que se encuentra en la región dente, las formas de prevención de accidentes, la educación vial
sudeste de Brasil, durante el año 2015, se registraron 10.107 ac- de peatones y conductores con objetivo de promover la salud y
cidentes de tránsito, que se producen principalmente entre los la cultura de paz.La formación de los profesionales en el tema;
automóviles (30,1%) y motocicletas (49,2%)[17].Las víctimas de debe dar prioridad a la programación de trabajo; educación
accidentes son mayoritariamente hombres (73,3%) y jóvenes.En permanente para la creación de redes de apoyo y protección
el mismo periodo, en 2,3% de los casos fueron reportados el (incluyendo desde la prevención hasta el seguimiento de los
consumo de alcohol y drogas entre las víctimas de accidentes casos en la emergencia, rehabilitación, asistencia psicológica a
trânsito[17]. Frente a casos de accidentes y desde la perspec- las víctimas y sus familias) sigue desafiando a los gestores y
tiva de los "serviços sentinela", la unidad de emergencia es una profesionales de la salud para exigir acciones concretas[21].
de las "puertas de entrada" del Sistema de Salud que funciona En Brasil, como los accidentes y la violencia son objeto de no-
como una oportunidad para el desarrollo del caso, lo que hace tificación obligatoria, hay una necesidad de acciones de carácter
este sector estratégico para la investigación de indicadores de educativo e informativo, haciendo hincapié en la distinción entre
accidentes, sus diversas manifestaciones y consequências[16]. notificación y denuncia y proporcionando un foro de discusión
La notificación de accidentes ha sido una herramienta para los para los profesionales de diversos campos reflexionar sobre el
vínculos entre los sistemas de salud y garantía de los derechos, miedo que impregna las notificaciones, su responsabilidad, el
con la integración de las acciones de promoción de la salud, pre- compromiso y la conducta ética delante de la persona que ex-
vención y control de accidentes. Contribuyendo así a aumentar perimenta accidentes en su área de alcance, así como evaluar
el conocimiento de los accidentes de tránsito, en la perspectiva estrategias de apoyo[22].
del Sector de la Salud, para ayudar en la investigación de indi- El tema de los accidentes de tránsito, haciendo hincapié en
cadores, así como subvencionando la expansión de las políticas, la notificación obligatoria, debe incluirse en los contenidos de
programas y prácticas destinadas a la prevención, para hacer los planes de estudios de los espacios de formación, así como en
frente a accidentes de tránsito y la reducción de daños. En este entornos profesionales[22].
Destaca la inclusión del tema en los contenidos de los planes 9. Leopoldo K, Leyton V, Oliveira LG. Uso exclusivo de álcool
de estudios de los cursos, por lo que los accidentes y la violencia e em associação a outras drogas entre motoristas de cam-
se incorporan como objeto de la salud y así investigada durante inhão que trafegam por rodovias do Estado de São Paulo,
el contacto con el cliente[22]. Brasil: um estudo transversal. Cad. Saúde Pública. 2015; 31
(9): 1916-1928.
Consideraciones finales:
10. Almeida ND, Roazzi A. Álcool e direção em universitários,
La prevención de los accidentes de tránsito y sus consecuencias comunicação persuasiva e prevenção.Psicol. cienc. prof.
están directamente relacionados con la atención prehospitalaria 2014; 34 (3): 715-732.
y hospitalaria de las víctimas, la atención de rehabilitación y
para, además, la vigilancia de los accidentes y la violencia, la 11. Abreu AMM, Lima JMB, Matos LN, Pillon SC. Uso de álcool
adopción de medidas educativas y legislativas de seguridad em vítimas de acidentes de trânsito: estudo do nível de
vial que contribuya a la reducción de la morbilidad y la mortal- alcoolemia. Rev. Latino-Am. Enfermagem. 2010; 18 [Spec]:
idad por estas enfermedades.Por lo tanto, se recomienda una 513-520.
mayor inversión en la prevención de accidentes de transporte
y rehabilitación de las víctimas con secuelas, apoyo psicológico 12. Boni R, Benzano D, Leukefeld C, Pechansky F. Uso de be-
y así reduciendo el impacto social de estos agravios.Existe la bidas alcoólicas em postos de gasolina de Porto Alegre:
necesidad de políticas de salud pública y estrategias que dan estudo piloto. Rev. psiquiatr. Rio Gd. Sul. 2008; 30 (1):
acceso a las acciones tanto en el campo preventivo como en el 65-68.
campo de la rehabilitación[23].
13. Aguilera SVU, Moysés ST, Moysés SJ. Intervenções de segu-
rança viária e seus efeitos nas lesões causadas pelo trânsito:
Conflicto de intereses uma revisão sistemática. Rev Panam Salud Publica. 2014;
Los autores declaran que no existe conflicto de interés en este 36 (4): 257-265.
manuscrito.
14. Almeida NA. Os acidentes e mortes no trânsito causados
pelo consumo de álcool: um problema de saúde pública. R.
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CASE REPORT
KIKUCHI-FUJIMOTO DISEASE
Irami Araújo-Filho∗, 1 , Artur Dantas Freire∗∗ , Irami Araújo-Neto∗∗ , Carolina Chianca Dourado Lemos∗∗ , Letícia Araújo Costa
Uchôa∗∗ , Amália Cínthia Meneses Rêgo∗∗ , Marco Antônio Botelho Soares∗∗ , José Francisco Correia-Neto♦ , Carlos André Nunes
Jatobá♦ and Ana Maria de Oliveira Ramos♦
∗ Department of Surgery, Potiguar University – Laureate International Universities – Natal, Rio Grande do Norte, Brazil., ∗∗ Potiguar University – Laureate
International Universities – Natal, Rio Grande do Norte, Brazil., ♦ Department of Pathology, Potiguar University – Laureate International Universities, Natal, Rio
Grande do Norte, Brazil.
ABSTRACT Objective:The development of associated febrile to lymphadenomegaly leads to several diagnostic hy-
potheses, among them the Kikushi-Fujimoto disease.Method:This review was set up by searching PubMed/Medline,
Web of Science and Scopus database using the following key words: “Kikushi disease”, “Kikuchi-Fujimoto disease”,
“histiocytic necrotizing”, “lymphadenitis”,“lymphadenopathy”. Results:We report a case in a young patient with a fever
associated with the emergence of adenomegaly and weight loss in two months, associated with hepatosplenomegaly.
In laboratory tests showed anaemia with erythrocyte sedimentation rate (ESH) and lactate dehydrogenase (LDH) test
elevated, widened mediastinum, with bilateral pleural effusion. Conclusion:Excisional biopsy of supraclavicular lymph
node showed Kikuchi-Fujimoto disease on microscopic examination. After histopathological confirmation, were other
causes of febrile adenomegaly apart, starting dose corticosteroid immunosuppressive therapy.
KEYWORDS Kikushi disease; Kikuchi-Fujimoto disease; histiocytic necrotizing lymphadenitis; lymphadenopathy.
Herpes virus type 6 and 8[3], Human Immunodeficiency virus

(HIV), Parvovirus B19[4], paramyxovirus, parainfluenza viruses,
Introduction Yersinia enterocolitis, and Toxoplasma[3].
Kikuchi’s disease, also called Kikuchi-Fujimoto disease or His- Apoptotic cell death mediated by CD8 + cytotoxic T lympho-
tiocytic Necrotizing Lymphadenitis, was initially described in cytes positive is the primary mechanism of cell destruction[5-7].
young women. Is a rare pathology, of unknown cause, benign, Although initially described in young women, the Kikuchi
generally characterised by cervical lymphadenopathy and fever. disease clearly also occurs in men. The proportion of men and
The histopathological examination of the lymph nodes involved women affected by three reviews was 1:4, 1:1,6 and 1:1,26, always
differentiates Kikuchi disease of other more serious conditions. with a predominance of women affected[8-10]. Most patients
Although the pathogenesis of Kikuchi disease is unknown, are under 40 years of age[11-14].
the clinical presentation is clear, and histological changes sug-
The most common clinical presentation of the disease is fever
gest an immune response of T cells and histiocytes to an infec-
and cervical lymphadenopathy in a previously healthy young
tious agent. Numerous agents urging the sickness have been
patient. Fever, usually low and persisted for about a week is a
proposed, including the Epstein-Barr Virus (EBV)[1,2] Human
primary symptom in 30 to 50% of patients [8].
DOI: 10.5455/IJMRCR.kikuchi-fujimoto-disease
First Received: April 04, 2017
Accepted: May 11, 2017
Manuscript Associate Editor: George Baytchev (BG) Methods:
Editor-in Chief: Cvetanka Hristova (BG)
Reviewers: Ivan Inkov (BG) This review was set up by searching PubMed, Web of Science
1
Prof. Dr. Irami Araújo Filho, Full Professor of the Department of Surgery, PhD in and Scopus database using the following key words: “Kikushi
Health Sciences. Full Professor of the Post-Graduate Program in Biotechnology at
Potiguar University – Laureate International Universities, Natal, Rio Grande do Norte,
disease”, “Kikuchi-Fujimoto disease”, “histiocytic necrotizing”,
Brazil. E-mail: [email protected] “lymphadenitis”, “lymphadenopathy”.
Irami Araújo-Filho et al./ International Journal of Medical Reviews and Case Reports (2017) 1(1): 6-10
Case report splenomegaly and hepatomegaly, which may be associated with
abdominal lymphadenopathy, as was evidenced in the case re-
The woman, 20 years old, he began to rub the frame with sym-
ported[8].
metrical fists, knees and ankles and daily 39oC fever. Two
The involvement of lymph nodes is usually cervical and
months after the onset of symptoms, cervical tumour arose
located in Kikuchi disease[18]. The nodules are moderately
and painful. Associated to the feverish, she referred 7% of
increased in diameter (1-2cm) but occasionally are higher (≥
body weight in two months. Was admitted to the hospital
7cm)[18]. They are typically firm, fibroelastic, discreet and furni-
with the symptoms described, showing Traube’ space busy with
ture. The increased lymph node is often associated with a mild
splenomegaly (4cm of the costal edge left), hepatomegaly (6cm
pain[18].
from the costal edge right) and generalized lymphadenopathy
(axillary, supraclavicular chains left and cervical inguinal), being The increase in the size of the mediastinal lymph nodes is
the largest lymph node found in the cervical region left mea- minimal and only. However retroperitoneal nodules may be
suring 06cm in your larger diameter, fibroblastic, painful, not involved[19,20]. The diagnosis is unlikely to be confirmed un-
acceded to plans. The laboratory tests revealed hemoglobin til the completion of lymph node biopsy in such patients that
9.2 g/dL; hematocrit 26.3%; leukocytes 3900/mm³ with 84% present with fever of unknown origin.
segmented and 8% of lymphocytes; 119000 platelets/mm³; ery-
throcyte sedimentation rate (ESR) 110 mm; 1.7 mg creatinine/dL; Laboratory tests
total protein 5 g/dL; albumin 2,4g/dL; AST 220 U/L; ALT: 79
Most patients with Kikuchi disease have a normal blood
U/L, GGT: 247 U/L, LDH:2046 IU/Ml; alkaline phosphatase 551
count[8], even though leukopenia is observed in 20-32%[9,21].
U/L, 0,6% reticulocytes and direct Coombs test negative. The
Atypical lymphocytes are reported in up to 25% of patients[18].
serology for hepatitis B, C and HIV negative. Had an antinuclear
Other less common findings include thrombocytopenia, pancy-
factor (ANF) profile with core: no reagent; metaphase chromo-
topenia, and, in those with severe disease, chronic anemia[1,22].
somal plate reagent and antibody ribonucleoprotein no reagent.
The erythrocyte sedimentation rate can be normal but was
The chest x-ray showed a widening of the mediastinum, pleural
elevated to more than 60 mm/h in 70% of patients in a series[19].
effusion and bilateral pulmonary consolidation right. Antibiotic
Other non-specific findings may include slightly abnormal liver
therapy was initiated with Cefepime for eight days. During
function tests and high levels of lactate dehydrogenase[20].
the diagnostic investigation was prompted computed tomog-
raphy (CT) of total abdomen whose result revealed moderate Antinuclear antibodies (ANA), rheumatoid factor, lupus ery-
ascites and lymphadenopathy in the various chains of retroperi- thematosus and preparations are negative. Some patients ini-
toneal and bilaterally iliac lymph nodes. The chest CT evicted tially diagnosed with Kikuchi disease presented later systemic
small bilateral pleural effusion, with atelectasis, mediastinal lym- lupus erythematosus (SLE)[8,23]. The FAN should be performed
phadenopathy, bilaterally axillary and left the supraclavicular in patients with suspicion of Kikuchi disease that have features
region. Held left supraclavicular lymph node biopsy, showing suggestive of LES to delete such a diagnosis. A study describes
histolytic necrotizing lymphadenitis compatible with Kikuchi- transient elevation anti-DNA antibody levels of protein anti-
Fujimoto disease, represented in figures 1-4. Corticotherapy ribonuclear[9].
began in dose immunosuppressive therapy for 21 days. Evolved
with dramatic improvement of the clinical picture, improvement Diagnosis
of joint pain and significant reduction of adenomegaly.
Lymph node biopsy makes diagnosis of the disease of Kikuchi-
Fujimoto. The biopsy should be performed, despite the self-
Discussion: limiting nature of this syndrome to rule out more serious con-
We report a case of Kikuchi-Fujimoto disease. The actual in- ditions that require aggressive therapy such as lymphoma.
cidence of this disease is estimated at between 0.5%, and 5% Kikuchi disease patients were diagnosed as having lymphoma
of all analysed lymph histologically; Not found any infectious treated with cytotoxic agents when doctors and pathologists still
microorganism (virus or bacteria) to the case reported, but it is did not know in detail this entity[8]. Other pathologies that were
possible that the antigenic stimulus has been triggered by one of confused with Kikuchi disease tuberculous adenitis, venereum
these[15]. lymphogranuloma and Kawasaki disease[24-26].
Though excisional biopsy is often recommended because
it often breaks up a framework of lymphoma, the fine needle
Clinical condition
aspiration is increasingly useful in the hands of the experienced
After reviewing the literature on the topic, it was found that the pathologist, using colours and cell block preparations, allowing
most common symptoms were fever (35%), fatigue (7%) and diagnosis[27,28].
joint pain (7%) [16]. The most common clinical and laboratory
findings were lymphadenopathy (100%), rash (10%), arthritis
Pathology
(7%), hepatosplenomegaly (3%), leukopenia (43%), high sedi-
mentation rate (40%) and anaemia (23%)[16]. The histology of the lymph node in Kikuchi-Fujimoto disease can
Systemic symptoms may accompany fever and lym- easily be differentiated from more well-known infectious condi-
phadenopathy and seem to be more prominent in patients tions in the differential diagnosis of fever and Lymphadenopathy
with extranodal involvement[17]. Systemic symptoms include [8-11],[29]. Yellow necrotic foci can hardly be noticed in the cut-
night sweats, nausea, vomiting, weight loss (by about 10%) and ting surface of the nodule. Microscopic examination usually
diarrhea[15-17]. shows outbreaks paracortical with necrosis and cellular infiltrate
A variety of other symptoms and physical signs occur spo- histiocytic. These outbreaks can be single or multiple. The cap-
radically in patients with Kikuchi disease. These include sule can be infiltrated, and perinodal inflammation is common.
chills, myalgia, arthralgia, pain in the chest and abdomen, The necrotizing process is often confined to circumscribed areas
of fibrinoid Eosinophilic material with an irregular distribution
of fragments of rubble nuclear[11].
The histological appearance changes as the disease progress.
Early biopsies on "proliferative phase" show follicular hyper-
plasia and paracortical expansion by T cells, B cells and mono-
cytes and miscellaneous plasmacytoid histiocytes with numer-
ous deep down apoptosis[30].
In "the proliferative phase," the presence of several blastic
cells raises the differential diagnosis of lymphoma, infection
with Epstein-Barr virus (EBV), and herpes simplex infection.
The preservation of nodal architecture, the mole, and polyclonal
immunohistochemistry negative viral conditions exclude[30].
Subsequent biopsies on "Necrotizing phase" have shown
without a neutrophilic infiltrate necrosis associated with the
domain of histiocytes. The histiocytes often have nuclei in grow-
ing and contain dendrites phagocytosed. Immunohistochemical
staining shows positive monocytes plasmacytoid and CD68 histi-
ocytes with predominantly CD8 positive T lymphocytes[30]. The Figure 2: Axillary lymph node blade, representing necrotizing
absence of neutrophils in "Necrotizing phase" is useful to distin- lymphadenitis with nuclear fragmentation - Image with a mag-
guish this condition from LES and drug-induced lymphadenopa- nification of 400x.
thy.
Figure 3:Axillary lymph node blade, representing necrotizing

Figure 1:Axillary lymph node blade, representing necrotizing lymphadenitis with nuclear fragmentation and reactive hyper-
lymphadenitis with nuclear fragmentation and reactive hyperplasia - Image with a magnification of 50x.
plasia - Image with a magnification of 100x.
Radiology
Computed tomography (CT) of the affected lymph nodes typ-
Differential diagnosis ically demonstrates perinodal infiltration (81%) and homoge-
neous enhancement (83%)[32]. On ultrasound, the lymph nodes
The differential diagnosis of the histological point of view in- can present radiological features of malignancy[33].
cludes LES, herpes simplex and lymphoma (non-Hodgkin’s lym-
phoma and Hodgkin’s Lymphoma). In the LES, bodies of hema-
Treatment
toxylin and plasma cells are also seen. In herpes simplex, there
are fewer surrounding mononuclear cells and neutrophils are No effective treatment for the disease has been established of
commonly present. In contrast to the Kikuchi disease, necrosis Kikuchi-Fujimoto to the present. The signs and symptoms usu-
associated with Hodgkin’s lymphoma usually includes neu- ally disappear within one to four months. Patients with severe or
trophils and large atypical cells (cell variants of Reed-Sternberg persistent symptoms are treated with high-dose glucocorticoids
cells), positive for CD30, CD15, CD45. It is suggested that associated with intravenous immunoglobulin, or not showing
the plasmacytoid dendritic cells infiltrate in lymph nodes with results apparently promising[34,35]. There have been reports of
higher frequency in Kikuchi-Fujimoto disease when compared success in the treatment of recurrent disease of Kikuchi-Fujimoto
to any other reactive lymphadenitis or B or T cell lymphoma, with hydroxychloroquine[36]. Affected patients should be fol-
regardless of the size of the lesion. Thus, the predominance lowed for a few years because they may be affected of LES and
of plasmacytoid dendritic cells can be a useful indicator in the relapses of the disease of Kikuchi-Fujimoto are applicants for
diagnosis of hepatological disease of Kikuchi[31]. several years, after the first episode of disease[22-26].
Conclusion 13. Payne JH, Evans M, Gerrard MP. Kikuchi-Fujimoto disease:
a rare but important cause of lymphadenopathy. Acta Pae-
Excisional biopsy of supraclavicular lymph node showed
diatr 2003; 92:261.
Kikuchi-Fujimoto disease on microscopic examination. After
histopathological confirmation, were other causes of febrile ade- 14. Ray A, Muse VV, Boyer DF. Case records of the Mas-
nomegalias apart, starting dose corticosteroid immunosuppres- sachusetts General Hospital. Case 38-2013. A 30-year-old
sive therapy. man with fever and lymphadenopathy. N Engl J Med 2013;
369:2333.
Competing Interests
15. Infante MJ, Lovillo C, Santaella IO, Checa RM, González
Written informed consent obtained from the patient for publica- MR. Enfermedad de Kikuchi-Fujimoto como causa de lin-
tion of this case report and any accompanying images. fadenopatías. An Pediatr (Barc). 2007;67(1):83-5.
16. Kucukardali Y, Solmazgul E, Kunter E, et al. Kikuchi-

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CASE REPORT
PANCREAS DIVISUM CAUSING RECURRENT

PANCREATITIS: A CASE REPORT
Nadeem Siddiqui∗ , Rizwan Sultan∗ and Fareed Shaikh∗,1
∗ Department of Surgery Aga Khan University Hospital, Karachi, Pakistan.
ABSTRACT Background: Pancreas divisum is one of the rare causes of recurrent pancreatitis. Diagnosis is challenging
and diagnostic failure leads to recurrent episodes of pancreatitis and ultimately pancreatic failure. Case: Here we are
presenting a case of a young female with a history of recurrent attacks of upper abdominal pain which on work up was
diagnosed as pancreas divisum. This patient was successfully treated with ERCP with minor papillotomy followed by
stenting in major papilla. Written informed consent was taken from the patient before writing this case report.Conclusion:
This report emphasises the need for early suspicion of disease, in patients with recurrent idiopathic pancreatitis, by
clinician and efficacy of minimally invasive procedures (ERCP) as a definitive treatment option.
KEYWORDS Pancreas Divisum, Recurrent pancreatitis, Endoscopic Papillotomy
endoscopic sphincterotomy.
Here we are presenting a case of 24 years young female with
Introduction a history of recurrent attacks of pancreatitis, which on workup is
found to be secondary to pancreas divisum. She was successfully
Pancreas divisum is one of the most commonly encountered treated with endoscopic sphincterotomy with the pain-free post-
congenital anomalies in the hepatobiliary system. The incidence procedure course. Report points out the need for early disease
is variable, but it ranges from 5% to 14% of the general pop- suspicion and efficacy of endoscopic sphincterotomy for the
ulation [1]. The disease occurs due to failure of fusion of the treatment of this disease.
two embryonic parts of the pancreas i.e. non-fusion of dorsal
and ventral ducts of pancreas resulting in openings at abnormal
Case Presentation:
positions, This abnormal opening of primary pancreatic duct at
minor papilla is not enough to completely drain the duct secre- Written informed consent was taken before reporting this case
tions resulting in stasis of enzymes and premature activation of report. Our patient was a 24 years old lady, presented in the
these enzymes results in recurrent attacks of pancreatitis [2, 3]. emergency department with a complaint of sudden onset severe
Most of the patients are asymptomatic, and diagnosis is in- continuous epigastric pain for last four days. This pain was
cidental. Abdominal ultrasound is not a very useful modality not radiating, aggravated by food intake and relieved only with
for diagnosis. Computed tomography (CT) of the abdomen may opioid analgesics. The pain was also associated with non-bilious
help in diagnosis, but imaging of choice is cholangiography vomiting. She had an episode of similar pain about one year
(ERCP or MRCP). MRCP being better as it is noninvasive but back for which she was admitted and managed as acute pan-
ERCP has an advantage in its diagnostic as well as therapeu- creatitis. She had a history of the acid peptic disease, anorexia,
tic capacity. Treatment is usually nonsurgical in the form of and malaise and weight loss of 6 Kg in last three years. On
examination, she was dehydrated and jaundiced. Her pulse was
96/minute; BP was 117/68, afebrile. Abdominal examination
DOI: 10.5455/IJMRCR.pancreas-divisum-recurrent-pancreatitis
First Received: April 12, 2017 revealed tenderness in an epigastric area without any mass or
Accepted: May 09, 2017 palpable viscera. Rest of systemic examination was unremark-
Manuscript Associate Editor: George Baytchev (BG) able.
Editor-in Chief: Cvetanka Hristova (BG) Her initial laboratory workup showed TLC of 10600/mm3,
Reviewers: Ivan Inkov (BG)
1
[Department of Surgery Aga Khan University Hospital, Karachi, Pakistan. Email:
total bilirubin 1.2 mg% (0.2-1.2) with direct bilirubin 0.9 mg%
[email protected] (0.0 to 0.4), alanine aminotransferase of 71 IU/l (normal 0-31
Fareed Shaikh et al./ International Journal of Medical Reviews and Case Reports (2017) 1(1): 11-13
IU/l), aspartate aminotransferase of 251 IU/l (normal 0-34 IU/l), which confirmed the presence of minor papilla about 5 mm
alkaline phosphatase of 85 IU/l (12-38 IU/l), serum amylase proximal to the major papilla, major pancreatic duct was opening
was 99 IU/l (25-125 IU/l) and serum lipase was 167 IU/l (3-60 at minor papilla while minor pancreatic duct was opening at
IU/l). major papilla (figure 2), Minor papillotomy was done and plastic
Ultrasonography (US) of abdomen surprisingly revealed a stent placed in major pancreatic duct. The patient tolerated the
hypoechoic solid mass in the head of the pancreas resulting in procedure well, and no immediate or delayed complication was
atrophy of pancreatic body and tail, dilatation of pancreatic duct identified.
and prominent common bile duct suggestive of the neoplastic
process.
To further delineate this situation, CT scan of the abdomen
was done which showed a hypodense mass involving the unci-
nate process of the pancreas causing atrophy of body and tail
of pancreas without any infiltration into surrounding structures.
Pancreatic duct and common bile duct were prominent with-
out dilatation of intrahepatic biliary channels appeared dilated.
(Figure 1) This CT scan was discussed in length with radiol-
Figure 2: ERCP showing presence of minor papilla about 5

mm proximal to the major papilla, major pancreatic duct was
opening at minor papilla (upper figure) while minor pancreatic
duct was opening at major papilla (lower figure).
Post procedure, the patient had a smooth recovery with sig-

nificant improvement in abdominal pain and appetite. She was
discharged on 3rd post procedure day. On clinic follow-up of
6 months, she has remained pain-free and leading a normal
healthy life.
Discusion:
Pancreatic Divisum is a congenital anatomical anomaly charac-
Figure 1: CT scan showing hypodense mass involving the un- terised by the lack of fusion of the ventral and dorsal parts of
cinate process of pancreas and dilated common bile duct and the pancreas during the eighth week of fetal development. This
pancreatic duct. condition is found in 5% to 14% of the general population [1].
In a large retrospective study from India, pancreas divisum was
ogists as it is hard to differentiate between Pancreas Divisum more frequent in patients with pancreatitis than in those with
and pancreatic malignancy. Finally, a provisional diagnosis of biliary diseases or obscure abdominal pain (9 versus 2 percent).
Pancreas Divisum was made as there was a convincing evidence The major pancreatic duct (Wirsung’s duct), drains the secre-
of opening of dilated primary pancreatic duct at minor papilla. tions from the head, body and tail of the exocrine pancreas, and
ERCP was done as diagnostic as well as therapeutic measure, ends at the major duodenal papilla (hepatopancreatic ampulla);
the accessory pancreatic duct (Santorini’s duct) extends through References
the head of the pancreas, crosses Wirsung’s duct and ends at the
1. Liao Z, Gao R, Wang W, et al. A systematic review on
minor duodenal papilla; both pancreatic outlets are located on
endoscopic detection rate, endotherapy, and surgery for
the medial wall of the second part of the duodenum at a distance
pancreas divisum. Endoscopy 2009;41:439-44.
of approximately 10 to 15 mm from each other; the minor papilla
are above, the major duodenal papilla below. 2. Vasile D, Grigoriu M, Turcu F, et al. [Pancreas divisumea
In Pancreatic Divisum, the dorsal pancreatic section drains rare cause of chronic pancreatitis]. Chirurgia (Bucur) 2007;
into the minor duodenal papilla through the major pancreatic 102:83-7.
duct; the ventral pancreatic duct, the smaller part of the pan-
creas, merges with the common bile duct at the hepatopancreatic 3. Gonoi W, Akai H, Hagiwara K, et al. Pancreas divisum
ampulla. There are two types of Pancreatic Divisum: complete as a predisposing factor for chronic and recurrent idio-
(most common) and incomplete (much less common), in which pathic pancreatitis: initial in vivo survey. Gut (2011);
the ventral and dorsal systems remain connected through small- doi:10.1136/gut.2010.230011.
caliber branch ducts. Approximately 15 percent of cases of pan- 4. Kamisawa T, Tu Y, Egawa N, et al. Clinical implications of
creas divisum are of the incomplete type. However, the clinical incomplete pancreas divisum. JOP 2006; 7:625-30.
implications of incomplete pancreas divisum are the same as for
classic (or complete) pancreas divisum, 5. Ng WK, Tarabain O. Pancreas divisum: a cause of idiopathic
In PD, the increased incidence of acute and chronic pancreati- acute pancreatitis. CMAJ 2009; 180:949-51.
tis is caused by inadequate drainage of secretions produced by
6. Elena G, Dorin A, Roxana B, Gheorghe B. Pancreas divi-
the body, tail and part of the pancreatic head through an orifice
sum pancreatitis: a case report. Abdom Imaging (2011)
which is too small [4, 5]. There is a group of patients with pan-
36:215–217.
creas divisum who are subject to recurrent bouts of seemingly
idiopathic pancreatitis [6]. In these patients, the minor papilla 7. Kamisawa T, T. Y. (2007). MRCP of congenital pancreatico-
orifice is so small that excessively high intrapancreatic dorsal biliary malformation. Abdom Imaging, 129-133.
ductal pressure occurs during active secretion, which may result
in inadequate drainage, ductal distension, pain, and, in some 8. GA, L. (2003). Acute recurrent pancreatitis. Can J Gastroen-
cases, pancreatitis. Although many times, patients with pancreas terol, 381-383.
divisum remain clinically asymptomatic, other common forms
of clinical presentations range from recurrent attacks of vary- 9. Kamisawa T (2004) Clinical significance of the minor duo-
ing degree of pancreatitis, bowel obstruction, ascites, jaundice, denal papilla and accessory pancreatic duct. J Gastroenterol
shock and in its most severe form, can lead to shock. Alcohol 39:605–615.
seems to be the triggering factor for the attack of pancreatitis [6].
Our patient presented with upper abdominal pain secondary to
pancreatitis.
Diagnostic workup ranges for laboratory tests to imaging
modalities. Laboratory workup may show raised amylase or
lipase levels indicating an episode of pancreatitis, but they are
not accurate for diagnosing Pancreas Divisum. Imaging modali-
ties which may help in diagnosis include ultrasound, CT scan
abdomen but definitive diagnosis is made with some form of
cholangiography either ERCP or MRCP. PD is most of the times
diagnosed from MRCP but the important point to note here is
that currently used 64 slicer CT scan is a good modality to diag-
nose PD like in our case, especially when the diagnosis is not
being suspected [7].
The usual therapeutic solution for symptomatic Pancreatic
Divisum is a sphincterotomy of the minor duodenal papilla,
which decongests Wirsung’s duct [8, 9]. Clinical improvement
with such treatment has been observed in up to 75 percent of
patients. Rarely, in selected cases only, is surgical treatment
indicated: surgical sphincterotomy, draining or even partial
pancreatectomies and their results are comparable with those
achieved by endoscopic procedure [6].
Conclusion
This report emphasizes the need of early suspicion of disease, in

patients with recurrent idiopathic pancreatitis, by clinician and
efficacy of minimally invasive procedures (ERCP) as definitive
treatment option.
CASE REPORT
VULVAR NON-FILARIAL ELEPHANTIASIS

ASSOCIATED WITH SCABIES
Sarah Sabur∗,1 , Saoussane Bounajma∗ , Mounia Nasr∗ and Samir Mazouz∗
∗ Department of Plastic Surgery, Ibn Sina teaching hospital, Mohamed V University, Rabat, Morocco.
ABSTRACT Vulvar elephantiasis is a very rare condition often caused by filariasis infection. Other causes like general
inflammatory disease, cancer treatment, traumatic causes or other infections can also explain the occurrence of this
condition. In some cases, it is due to unknown causes. In this study, we present the clinical case of a Moroccan youth
female patient with no significant history, who presented vulvar elephantiasis associated with scabies with no evidence
of filariasis infection, general inflammatory disease, malignancy, or other infection. This clinical case is unusual because
of the large volume of lymphedema, its atypical localisation, its association with generalised scabies, and the absence of a
well-defined aetiology, which may explain its occurrence.
KEYWORDS Elephantiasis, vulvar, scabies, lymphedema.
Introduction
Elephantiasis describes a massive enlargement of a limb or the
external genitalia because of chronic lymphedema [1]. The ele-
phantiasis of the vulva is a rare condition often caused by fi-
lariasis in developing nations [3]. In this study, we present the
clinical case of a 27-year-old patient with no significant medical
history, who presented elephantiasis of the vulva evolved one
year ago, associated with scabies. The patient did not show any
evidence of filariasis infection or any other known cause which
can explain the occurrence of this condition.
Case report:
A female patient of 27-years-old, with no previous medical his-
tory, was admitted to our department for vulvar elephantiasis
of one-year evolution associated with generalised itchy skin le-
sions. The general clinical examination found: BMI=26, normal
body temperature, generalised, smooth and slightly elevated

DOI: 10.5455/IJMRCR.vulvar-non-filarial-elephantiasis-scabies
First Received: March 22, 2017
Accepted: April 15, 2017
Editor-in Chief: Ivan Inkov (BG)
Reviewers: Khadija Bellahammou (MA)
Figure 1: Anterior view of the vulva.
1
Sarah Sabur, Plastic Surgery Departement - Ibn Sina Teaching Hospital Mohamed V
University Rabat, Morocco. [email protected]
Sarah Sabur et al./ International Journal of Medical Reviews and Case Reports (2017) 1(1): 14-16
Figure 4: the two masses after excision.
Figure 2: Posterior view of the vulva.
Figure 5: Vulvoplasty.
tures were realised to close the residual substance loss (figure 5).
The pieces were sent to the anatomopathological study which re-
turned for vulvar lymphedema, without any sign of malignancy.
The surgical follow-up was simple, but the patient presented
again after two months with a new recurrence of lymphedema
in the left labia majora (figure 6), without any associated general
skin lesions.
Discussion:
Figure 3: view of the left and right labia majora with the two Lymphedema can be defined as swelling of soft tissues which are
masses. the result of the accumulation of protein-rich interstitial fluids
caused by a low output failure of lymph [3].
The Impairment of lymphatic drainage can be due to con-
erythematous papules, and scratch lesions. The vulvar exam- genital abnormal vessel development (primary lymphedema) or
ination found a voluminous pendulous swelling mass in the damaged lymphatic vessels from infection, trauma, surgery, ra-
left labia majora with minimal skin inflammation, and moder-
ate lymphedema in the right labia majora, (figure 1, 2). The
clinical examination also found bilateral inguinal adenopathy,
and lower limb varices. However, the clinical and radiological
examinations did not reveal the presence of an abdominal or
gynaecological mass. The biological testing showed an increas-
ing number of circulating eosinophilic granulocytes. C-reactive
protein (CRP) was also elevated (84 mg/L), and the routine bio-
chemistry, glucose, serological testing for sexually transmitted
infection (hepatitis B, HIV, syphilis) didn’t show any particular-
ity. We also performed a Midnight peripheral blood smear for
filarial worms, but the result was negative. The patient received
an antiscabies treatment for her skin lesions, and she underwent
into a complete excision of her vulvar masses, which carried out
a mass of 2 kg from the left labia majora and a mass of 500 g from
the right labia majora (Figures 3 and 4). Simple cutaneous su- Figure 6: Recurrence of lymphedema after one month.
diation, persistent inflammation, obstruction due to metastases Conclusion
or parasite infestation (secondary lymphedema) [1].
This clinical case is unusual because of the large volume of
Lymphedema is frequently encountered in the limbs, less lymphedema, its atypical localisation, its association with gen-
commonly in the face and the genital organs [3]. In its earli- eralised scabies, and the absence of a well-defined aetiology,
est stages, lymphedema is soft and pitting; however, with per- which may explain its occurrence.
sistence, lymphedema has a brawny, non-pitting appearance
likened to peau d’orange because of accentuation of skin folds Acknowledgements
and follicular ostia. Most of the swelling occurs in the subcu-
taneous tissue, but the skin exhibits the most changes, which, Sabur Sarah wrote the case report. All authors have read and
when severe, is termed elephantiasis [1]. Elephantiasis described agreed to the final version of this manuscript and have equally
a gross enlargement of the part of the body that has been in- contributed to its content and the management of the case.
volved. Review of the world’s literature reveals that chronic
vulvar elephantiasis is a very rare condition [3]. Disclosure Statement
The majority of cases of vulvar elephantiasis found in the lit- There were no financial supports or relationships between the
erature are due to filariasis [3] [4]. Other causes include bacterial authors and any organisation or professional bodies that could
sexually transmitted infections (STI’s), especially lymphogranu- pose any conflict of interests.
lomavenereum (LGV) and donovanosis; tuberculosis, haemato-
logical malignancies, and dermatological diseases and in some Competing Interests
cases from unknown cause [4, 1, 2].
Written informed consent obtained from the patient for publica-
Lu et al. [1] presented a clinical series of 24 cases of localised tion of this case report and any accompanying images.
lymphedema; he made a comparison between those patients
and the patients with diffuse lymphedema. Lu found that the
most frequent localisation in these patients is anogenital, and
References
overall, women were more frequently affected by lymphedema, 1. Lu S, Tran TA, Jones DM, Meyer DR, Ross JS, Fisher HA,
particularly in the anogenital region and trunk, than men. Carlson JA: Localized lymphedema (elephantiasis): a case
The patients in Lu et al. series did not have any form of cancer series and review of the literature. J Cutan Pathol 2009.
treatment, they were significantly more likely to have history of 2. Antonio A, Caleo A, Boscaino A, Mossetti G, Lan-
trauma to the site (vaginal delivery), and less likely to have a nantuoni N, Vulvar lymphoedematous pseudotumours
coexisting, chronic inflammatory process (rosacea and Crohn’s mistaken for aggressive angiomyxoma: Report of two
disease), No etiology or associated disease was found in about cases.Gynecol.Obstet Invest 2010 ;69 :212-216.
one-third of all cases. Lu and al. Also, show in his study that
85% of patients were obese or overweight which can promote 3. Chaudhary R, Rathi S, Maheshwari A, Nigam S.Vulvar ele-
lymphedema by obstruction of the lymphatic canals by fat cells. phantiasis of filarial origin: A case report.Indian journal of
Antonio et al. [2] presented two clinical cases of unilabial clinical practice 2013;24 (2).
vulvar lymphedema similar to the Lu and Al series, but unlike 4. Ipyana HM, Bonaventura CTM, Januarices H.Vulvar filarial
the Lu et al. series, they did not find any factors that may explain elephantiasis in a Tanzanian woman, a rare presentation
the onset of Lymphedema except obesity in one patient. of lymphatic filariasis. A case report and review of litera-
In our study the patient did not have any previous medical ture.Sudan Journal of Medical Sciences 2014; 9(4):256-270.
history or an apparent cause that can explain the vulvar ele-
phantiasis, the research of filariasis was negative and also no 5. Kos M, Ljubojevic N, Ilic-Forko J, Babic D, Jukic S. [Elephan-
sign of sexually transmitted infection, neither a sign of malig- tiasis of the vulva of an unclear aetiology: a case report].
nancy or inflammatory disease. Histological examination of the Lijeniki Vjesn 1996;118(7-8):158–60.
excised mass showed Acanthosis papillomatous epidermis sur-
mounted by hyperkeratosis and orthokeratosis without cytonu-
clear atypia; in the dermis, we found many dilated lymphatic
vessels of variable size. Important interstitial oedema without
any sign of malignancy. The only abnormality she presented
beside the vulvar elephantiasis was the skin lesions which were
resolved after antiscabies treatment and, the slightly elevated
level of circulating granulocyte and the CRP which may correlate
with the previous scabies infestation.
Our patient presented a recurrence of lymphedema after ex-
cision. In Lu and al series the majority of lymphedematous
tumours were cured with excision, and those that recurred or
progressed were associated with factors known to aggravate
lymphedema: obesity, cellulitis, signs of persistent inflamma-
tion. In our patient, we believe that persistent of inflammation
can explain recurrence. However we planned to realise more
investigation to explain the recurrence, but the patient refuse to
go under supplementary examination.
CASE REPORT
PITUITARY PROLACTIN MICROADENOMA WITH,

CORTICOTROPIC AND THYREOTROPIC DEFICIENCY:
FROM INFERTILITY TO PREGNANCY: ABOUT A CASE
Diallo Moussa∗,1 , Koulimaya Cyr Esperence Gombet∗ , Diallo Astou Coly Niassy∗∗ , Diouf Abdoul Aziz∗ , Gassama Omar∗∗ , Gueye
Mame Diarra Ndiaye∗∗ , Leye Yakham∗ ∗ ∗ and Diouf Alassane∗
∗ Department of obstétric and gynecology of Pikine National Hospital., ∗∗ Department of obstétric and gynecology of Aristide Le Dantec Hospital., ∗∗ Department
of internal medicine of Pikine National Hospital.
ABSTRACT
Pituitary adenomas are benign tumours developed at the expense of different cellular populations of the pituitary gland.
Within the pituitary gland, several cell populations may be involved, but the lactotrophic cells remain the most frequently
affected by this hyperplasia. For both sexes, the overall frequency of adenomas is 100 per million, of which 40% are
prolactinomas. The stimulating effect of oestrogens (combined oral contraceptives and pregnancy) on lactotrophic
cells has long been demonstrated, and in general, only large tumours (macroadenomas) have an evolving risk to be
feared during pregnancy. The diagnosis rests on the one hand on the evidence of a hormonal hypersecretion of the cell
population concerned as well as a hormonal deficiency of the other cell groups which can be compressed by the tumour.
On the other hand, this diagnosis uses hypophyseal magnetic resonance imaging (MRI) to distinguish, according to
their size, microadenomas (diameter less than 10 mm) from macroadenomas (diameter greater than 10 mm) pituitary.
The risk of increasing the volume of the adenoma during pregnancy depends on the initial size of the tumour. This
risk is evaluated at 2% for microadenoma and 15-35% for macroadenomas. However, the most severe complication
during pregnancy remains acute paroxysmal growth or pituitary apoplexy by necrotic-haemorrhagic phenomena. The
management is mainly based on prolactinoma on bromocriptine or cabergoline and sometimes surgery, urgently in the
presence of a pituitary apoplexy or the presence of an evolutionary macroadenoma.
KEYWORDS: Pituitary microadenoma, infertility, pregnancy, prolactin
Introduction
Pituitary adenomas are benign tumours developed at the ex-

pense of different cellular populations of the pituitary gland.
Within the pituitary gland, several cell populations may be in-
volved, but the lactotrophic cells remain the most frequently af-
Copyright © 2017 by the Bulgarian Association of Young Surgeons fected by this hyperplasia. For both sexes, the overall frequency
DOI:10.5455/IJMRCR.pituitary-microadenoma
First Received: March 26, 2017
of adenomas is 100 per million [1], of which 40% are prolactino-
Accepted: March 30, 2017 mas. The stimulating effect of oestrogens (combined oral con-
Manuscript Associate Editor: George Baitchev (BG) traceptives and pregnancy) on lactotrophic cells has long been
Editor-in Chief: Cvetanka Hristova (BG) demonstrated, and in general, only large tumours (macroade-
Reviewers: Derun Taner Ertugrul (TR) nomas) have an evolving risk to be feared during pregnancy. A
1
Diallo Moussa, Department of Obstetrics and Gynecology, Centre Hospitalier National
de Pikine Sis Camp de Thiaroye, Dakar, Senegal.;
significant aspect is represented by fertility disorders induced in
E-mail: [email protected] both sexes and which sometimes constitutes the circumstance of
Diallo Moussa et al./ International Journal of Medical Reviews and Case Reports (2017) 1(1): 17-19
discovery. The diagnosis rests on the one hand on the evidence rest of the gland (Figure 2). In summary, the diagnosis of pitu-
of a hormonal hypersecretion of the cell population concerned itary prolactin’s microadenoma was retained with corticotropic
as well as a hormonal deficiency of the other cell groups which and thyrotropic insufficiency complicated by primary infertil-
can be compressed by the tumour. On the other hand, this di- ity. She had subsequently benefited from a cabergoline-based
agnosis uses hypophyseal magnetic resonance imaging (MRI) treatment at a dosage of 0.25 mg per week, levothyroxine due to
to distinguish, according to their size, microadenomas (diam- 50 mg daily in two doses and 40 mg hydrocortisone. Nineteen
eter less than 10 mm) from macroadenomas (diameter greater months later, the patient, with very irregular follow-up, was
than 10 mm) pituitary. The risk of increasing the volume of the admitted to maternity for the management of a pregnancy at
adenoma during pregnancy depends on the initial size of the tu- 38 weeks of amenorrhea complicated by severe preeclampsia
mour. This risk is evaluated at 2% for microadenoma and 15-35% (blood pressure of 240/130 mmHg). A caesarian was performed
for macroadenomas [2]. However, the most severe complication in emergency and had resulted in the birth of a female newborn
during pregnancy remains acute paroxysmal growth or pitu- weighing 3100 g. The return home was allowed after 12 days
itary apoplexy by necrotic-haemorrhagic phenomena. The man- without complications. Three months after the patient was stable
agement is mainly based on prolactinoma on bromocriptine or with a normal diaper return.
cabergoline and sometimes surgery, urgently in the presence of a
pituitary apoplexy or the presence of an evolutionary macroade-
noma. We reported here a case of microadenoma found during
the infertility assessment and followed during pregnancy and
childbirth.
Observation
Ms K B, 36, nulliparous, was referred the management of a
galactorrhoea without amenorrhea, and primary infertility of
the unexplored 10-year-old couple. The interrogation found an
age of onset of menars at 12 years, a regular menstrual cycle.
We showed a delay in ideation, pubic and axillary depilation
and bilateral galactorrhea. Examination of the thyroid gland
found a homogeneous goitre without clinical sign of hypothy-
roidism. There was no evidence of intracranial hypertension.
The patient had a body mass index of 27.04. The thyroxine
(tetraiodothyronine) level was decreased to 9.96 pmol / L, a level
of thyrotropin releasing hormone (usTSH) normal to 1.689 uUI
/ ml, a prolactin level of 45.42 ng/ml and a cortisol level of 78
ng/ml. Thyroid ultrasound noted a moderate left lobe heteron-
odular goitre. Pituitary magnetic resonance (MRI) imaging with
gadolinium injection resulted in a 5.5 mm left micro-adenoma
(Figure 1) with delayed contrast enhancement compared to the
Figure 2:Coronal section of the brain passing the pituitary in
T1 sequence with a dynamic injection, showing the delayed
enhancement of the adenoma
Discussion
Also known as pituitary hyperplasia, it is defined as an absolute
increase in a cell population within the pituitary gland, mani-
fested radiologically by an increase in its size [3]. The overall
frequency of adenomas is about 100 patients per million [1].
However, prolactin adenomas (or prolactinomas) remain the
most frequent of pituitary tumours. In both sexes, this con-
dition is responsible for impairment of reproductive function.
Indeed, hyperprolactinemia is responsible for suppression of the
pulsatile secretion of gonadotropin-releasing hormone (GnRH),
the positive feedback of oestradiol on gonadotropin secretion
and inhibition of progesterone production by The granulosa of
the ovary. Moreover, a mere duplication of its primary level
(prolactin) is enough to produce these effects [4, 5]. The asso-
ciation with an insufficiency of other hormonal axes is rare [3].
Our patient simultaneously affected three cell populations (hy-
perprolactinemia, corticotropic insufficiency and thyrotropin)
Figure 1: Coronal section through the pituitary gland (T2), show- causing galactorrhea without amenorrhea with infertility of the
ing a tumour of 5.5 mm diameter. couple.
Diagnosis as an amenorrhea-galactorrhea syndrome or infertility of the
The diagnosis of adenoma is most often made before pregnancy couple. Its association with pregnancy, on the one hand of the
[6]. The circumstances of the discovery are, in the woman in good evolution of the pathology, also raises the problem of ex-
reproductive period, an amenorrhea-galactorrhea syndrome as- cessive tumour growth but also exposes these patients to major
sociated with infertility, a syndrome of Cushing and more rarely complications such as pituitary apoplexy.
a tumour syndrome which makes suspicion a macroadenoma.
The pituitary hormone assay, especially prolactin, regains very References
high levels greater than 30 or 35 mg / L, which are also strongly
1. Fatfouta I, Delotte J, Mialon O, Isnard V, Bongain A. Ade-
correlated with the size of the adenoma. Pituitary MRI reveals
nome a prolactine : du desir de grossesse a l’ accouchement.
an increase in the volume of the pituitary, specifies its size and
journal de gynecologie obstetrique et biologie de la repro-
its relationship with adjacent structures (optic chiasm and cav-
duction 2013;42:316—24.
ernous sinus) and delayed contrast with the rest of the gland
as it was For our patient (Fig. 2). It is classical to distinguish 2. Molitch ME. Pituitary Diseases in Pregnancy. Seminars in
the pituitary microadenomas, whose diameter is less than 10 perinatology. 1998;22(6):457-70.
mm, from macroadenomas more than 10 mm in diameter [7].
Exceptionally, the adenoma is discovered during pregnancy by 3. Sunita MC, Sousa D, Peter E, Ann IM. Pituitary hyperplasia:
a tumoral syndrome related to pituitary hyperplasia on a pre- case series and literature review of an under-recognised
existing adenoma. The latter form provides complications such and heterogeneous condition. Endocrinology, Diabete
as apoplexy. The microprolactinoma is usually expressed by a and Metabolism cases report May 2015 D: 15-0017 DOI:
hypointense image T1 and, in 4 cases out of 5, a T2 hyperintense 10.1530/EDM-15-0017.
signal. The hypersignal T2 may correspond to only a part of the
adenoma [7]. 4. Melmed S, Casanueva F, Hoffman A. Diagnosis and
treatment of hyperprolactinemia: an Endocrine Society
Management clinical practice guideline. J Clin Endocrinol Metab ;:.
The management of prolactinomas relies on medical treatment 2011;96:273—88.
and neurosurgical excision of the tumour. The latter, although 5. Casanueva F, Molitch M, Schlechte J. Guidelines of the Pi-
assuring the definitive cure, is only a second intention proposed. tuitary Society for the diagnosis and management of pro-
However, surgery becomes indispensable in the presence of a lactinomas. Clin Endocrinol. 2006;65:265—73.
pituitary apoplexy or acute necrotic-haemorrhagic form and af-
ter the failure of the drug treatment. Moreover, the occurrence 6. Soto-Ares G, Cortet-Rudelli C, C D, JP P. Adenomes hy-
of pregnancy in these patients is only possible after a medical pophysaires et grossesse: considerations morphologiques
treatment that allowed standardisation of prolactin. It is based en IRM. Journal de radiologie. 2002;83:329-35.
on the dopaminergic antagonist’s bromocriptine, cabergoline
and quinagolide. These molecules, by stimulating the D2 re- 7. Bonneville J-F, Cattin F, Bonneville F. Imagerie desade-
ceptors, are responsible for a cascade inhibiting the lactotrophic nomes hypophysaires. Encyclopedie medico-chirurgicale
cell. Their effectiveness varies between 70 and 90%. With a Elsevier Masson SAS; 2009. p. 84-91.
rate of tumour growth rate of 1.4% of microadenomas during
8. Delemer B. Adenomes a prolactine : diagnostic et prise en
pregnancy, it is recommended to stop antidopaminergic drugs
charge. Presse Medicale. 2009;38:117-25.
to discover a pregnancy and to propose a simple monitoring
[8]. The pregnancy occurred in our patient after one year of 9. Molitch M. Prolactin-secreting tumors: what’s new? . Ex-
treatment, and the treatment could not be interrupted because pert Rev Anticancer Ther. 2006;6:S29—35.
she had been lost sight of and no malformation was detected at
birth.
Evolution and prognosis

In fact, during pregnancy, we see an increase in the lactotrophic
cell population in the pituitary, which can reach half of its total
cell population. This phenomenon is mostly observed during the
second and third trimesters of pregnancy where oestrogen levels
are highest. This suggests, according to its natural history, an
aggravation of the symptomatology (intracranial hypertension)
of the adenoma during the pregnancy. The evolutionary risk
seems more important in the case of macroadenoma [9]. In
our case, medical treatment had made it possible to correct the
various disorders. It was followed by a pregnancy during which
the patient presented no symptoms related to a complication of
the adenoma (a headache, vomiting, visual disturbances).
Conclusion
Pituitary adenomas are rare pathologies and have the particu-
larity of involving several entities or cell groups of the pituitary
gland. In women of childbearing age, it most often manifests
CASE REPORT
SURGICAL MANAGEMENT OF TRAUMATIC

MANUBRIO-STERNAL DISLOCATION WITH LOCKING
COMPRESSION PLATE: A CASE REPORT AND
REVIEW OF LITERATURE
Fareed Shaikh∗,1 , Syed Shahabuddin∗ , Haroon Rashid∗ and Noman Shahzad∗
∗ Department Of Surgery, Aga Khan University Hospital, Karachi, Pakistan.
ABSTRACT Background: Manubriosternal joint dislocation as a result of trauma is rare with only few case reports
published in the literature. Materials that have been used for fixation of displaced manubriosternal joint are steel wires,
polydioxanone ropes, and plates with screws. Case: We present a case of manubriosternal dislocation in which fixation
was done with locking compression plate. It is a case of 32 years old lady with a history of road traffic accident; car ran
over her chest. She had bilateral lung contusions with multiple rib fractures on right side and manubriosternal joint
dislocation of type-I. The patient was initially stabilised in high dependency unit, and once her contusions got better, she
underwent fixation of her manubriosternal dislocation with locking compression plate. Post-operatively she remained
pain-free and was discharged home. Conclusion: This case is an important addition to literature regarding options that
can be used for fixation of manubriosternal joint dislocation.
KEYWORDS Manubriosternal joint, Plate fixation, Joint dislocation, Chest trauma
letter from the Institutional Ethical Review Committee was ac-

quired (3368-Sur-ERC-14)
Introduction
Case Description:
Manubrio-sternal dislocation as a result of trauma is rare with
only few case reports published in the literature till now [1]. We had a 32 years old woman who presented to our emergency
The materials used for fixation of displaced manubrio-sternal department after sustaining blunt chest trauma as a result of
joint are steel wires [1], Polydioxanone ropes [2], and Plates with road traffic accident. She was a motorcycle rear seat rider and
screws [1, 3, 4]. had a head-on collision with a car. There was no history of loss
of consciousness. She was found to be awake, alert and vitally
We describe a case of the young lady with traumatic
stable. Airway, breathing and circulation were intact. On the
manubrio-sternal dislocation. She underwent fixation with lock-
subsequent survey, she was found to have a bruise over sternum
ing compression plate. Informed consent was taken from the
with a palpable deformity at the manubrio-sternal joint that was
patient for reporting the case with images and an exemption
tender to touch. Rest of the examination was unremarkable.
Her X-Ray chest and CT scan chest revealed multiple bilateral
DOI: 10.5455/IJMRCR.surgical-management-traumatic-manubrio-sternal-dislocation
rib fractures, bilateral lung contusions and manubrio-sternal
First Received: March 26, 2017 dislocation as shown in Figure 1. After initial management at
Accepted: May 09, 2017 the emergency department, she was kept in intensive care unit
Manuscript Associate Editor: George Baytchev (BG) (ICU) and later in high dependency unit for the management of
Editor-in Chief: Cvetanka Hristova (BG) lung contusions. Over next four days of conservative manage-
Reviewers: Atsushi Sano (JP)
1
[Department of Surgery Aga Khan University Hospital, Karachi, Pakistan. Email:
ment (Pain control, negative balance of fluids, O2 support and
[email protected] chest physiotherapy) her lung contusions improved. However,
gical treatment for sternal fractures, but due to the rarity of cases
and literature, no consensus has been reached at. In general
undisplaced fractures are considered for non-operative treat-
ment whereas displaced fractures, persistent pain affecting res-
piration, chronic non-union and sternal instability are the pri-
mary indications for surgical fixation as reported by Harston A
et al.[6].
In our patient, the manubrium was anteriorly displaced cat-
egorising it to be type I and the mechanism were a direct trau-
matic impact [6, 7].
Although people have used different tools for fixing this type
of fractures as mentioned above but consensus has not been
established yet. The reason for preferring locking compression
plate over the steel wires in our case was the former is much
safer if performed under fluoroscopic guidance, taking care not
to push screws much beyond posterior cortex; approximates
bone closely and it requires minimal dissection of tissues, hence
less interruptions in the blood supply of sternum as compared
to latter [8-10]. Moreover, the previous experience with sternal
wires, as reported by Salloum W et al.[1], was not good, as they
had to remove the wires and apply a metallic plate for fixation
two months after initial fixation by wires. In this regard CT scan
helped us in measuring the distance between the outer and inner
table of the sternum, hence the size of the screws to be used was
decided preoperatively. The timing of fixation has long been
debated, but Harston A et al.[6] reported that most of the sternal
fractures had been surgically fixed during the same hospital stay
as done in our case.
CONCLUSION:
her sternal pain persisted restricting movement of upper limbs.
Manubrio-sternal dislocation being the only factor responsible, Manubrio-sternal dislocations are extremely rare. Due to the
it was decided to fix the sternal dislocation. paucity of literature, a consensus has not been established yet on
Under general anaesthesia, in the supine position, the frac- the ideal method of fixation. However, this case report will be a
ture site was approached using a vertical incision in the midline valuable addition to literature to achieve desirable outcome after
over the palpable deformity. The hematoma was evacuated; surgical management of sternal fractures with titanium plate
the fracture was reduced and fixed using locking compression during the same hospitalisation.
plate with the help of screws. The length of the screws was
predetermined with the support of CT scan as the distance be- AUTHORS’ STATEMENTS
tween anterior and posterior cortices of the sternum. Fixation,
alignment and position of screws were confirmed using intraop- Competing Interests
erative fluoroscopy and with lateral radiograph postoperatively
Figure 2. She had unremarkable post-operative course; her pain
References
improved with a better range of motion in upper limbs. She was
discharged home on the third postoperative day. She is pain-free 1. Salloum W, Nikolaidis N, Weeden D. Manubriosternal Joint
with full range of motion in upper limbs at follow-up in the Dislocation–A Treatment Dilemma. The Internet Journal of
clinic after six months of surgery. Thoracic and Cardiovascular Surgery. 2009;15(1).
Discusion: 2. Lemaitre J, Koriche C, Massard G, Wihlm J. Manubrioster-

nal Disjunction: a New Approach for Surgical Repair. Acta
Traumatic dislocation of manubrio-sternal joint is rare and sur- chirurgica Belgica. 2004:593-5.
gical correction requires an individualised decision. There is
limited literature regarding large series regarding surgical fixa- 3. Nijs S, Broos P. Sterno-manubrial dislocation in a 9-year old
tion. Both wires and plates are being reported to be used with gymnast. Acta chirurgica Belgica. 2005;105(4):422.
an acceptable outcome. Thirupathi and Husted [5] classified
manubrio-sternal dislocations into two; Type-I, the rare variety, 4. Gaines RJ, Wilson A, Antevil J, Demaio M. Parallel
is one in which the body of the sternum is displaced posterior to plating for a sternomanubrial dislocation. Orthopedics.
manubrium, whereas, in type-II, the commoner variety, the body 2012;35(8):e1276-8.
is displaced anteriorly. Direct trauma to the chest is the common
mechanism involved in type-I, whereas deceleration injuries 5. Thirupathi R, Husted C. Traumatic disruption of the
resulting in hyperflexion of the torso, rheumatoid disease and manubriosternal joint. A case report. Bulletin of the Hospi-
kyphosis are the predisposing factors for type-II. tal for Joint Diseases Orthopaedic Institute. 1981;42(2):242-
There has been a lengthy discussion on conservative vs. sur- 7.
6. Harston A, Roberts C. Fixation of sternal fractures: a sys-
tematic review. Journal of Trauma and Acute Care Surgery.
2011;71(6):1875-9.
7. Chou SS, Sena MJ, Wong MS. Use of SternaLock plating

system in acute treatment of unstable traumatic sternal
fractures. The Annals of thoracic surgery. 2011;91(2):597-9.
8. Abdul-Rahman MR, Seong NK, Hee TG, Aljada ME, Reda

TA, SuMin JO, et al., editors. Comminuted sternal frac-
ture—a sternotomy wire fixation: report of 2 cases. The
heart surgery forum; 2009: Carden Jennings.
9. Al-Qudah A. Operative treatment of sternal fractures. Asian

Cardiovascular and Thoracic Annals. 2006;14(5):399-401.
10. Severson EP, Thompson CA, Resig SG, Swiontkowski MF.

Transverse sternal nonunion, repair and revision: a case
report and review of the literature. Journal of Trauma-Injury,
Infection, and Critical Care. 2009;66(5):1485-8.
CASE REPORT
TESTICULAR DESCEND, HOW AND WHY: A REVIEW

ARTICLE
SUJAN NARAYAN AGRAWAL∗, 1
∗ Associate professor, Department of Surgery. Late SBRKM Government Medical College, Jagdalpur (Bastar) C.G. PIN 494001, India.
ABSTRACT Background:The testis develops in the dorsal abdominal wall, and then descends to the scrotum. The
development begins as early as the 6th week of intrauterine life and is completed by the fifth month of intrauterine life.
The testis may get arrested during its descent from dorsal abdominal wall to the scrotum. The anomalies of descent
include cryptorchism (and its variant like anarchism, monarchism or partially descended testis), ectopic testis, persistent
processus vaginalis and encysted hydrocele of the spermatic cord, and others. Cryptorchism is usually diagnosed
during the newly born examination. The recognition of this condition, identification of associated syndromes, proper
diagnostic evaluation and timely treatment by a surgical urologist is important to prevent adverse consequences like
sterility, congenital hernia & hydrocoele, testicular carcinoma, and others. Objectives: The objective of this review is to
study the role of gubernaculum in the testicular migration process. Material & Method: We performed a descriptive
review of the literature about the role of the gubernaculum in testicular migration during the human fetal life. This
article provides an overview of the role of gubernaculum and other factors responsible for gonadal migration. Results:
In the first phase of testicular movement the gubernaculum enlarges to hold the testis near the groin and in the second
phase the gubernaculum migrates across the pubic region to reach the scrotum. The proximal end of gubernaculum
is attached to the testis and epididymis. The lower end reaches to the bottom of the scrotum. A failure in the proper
functioning of gubernaculum causes cryptorchism. Rarely male gonads may deviate from primary pathway due to the
presence of many tails of distal gubernaculum, and it may give rise to the ectopic testis. The processus vaginalis usually
closes by birth. If it remains patent, it leads to a congenital hernia, hydrocele and encysted hydrocele. Conclusion: the
gubernaculum presents a significant structure during testicular migration, and its failed mechanism gives rise to different
pathological conditions.
KEYWORDS testicular descend, gonads, gubernaculum, processus vaginalis, undescended testis, congenital hernia/hydrocele
Introduction
The urinary system consists of Kidney, Ureter, Urinary blad-

der and Urethra. The genital system consists of external and
internal genitalia. Male internal genitalia is testis, vas deference
Copyright © 2019 by the Bulgarian Association of Young Surgeons or ejaculatory ducts and seminal vesicles, whereas the external
DOI: 10.5455/IJMRCR.testicular-descend genitalia is scrotum and penis. The female genital system con-
First Received: May 20, 2017 sists of ovary, uterus and vagina. The testis is male, and ovaries
Accepted: June 18, 2017
are female gonads. The main difference lies in the fact that the
Editor-in Chief: Cvetanka Hristova (BG) male gonads have a well differentiated ductal system. The duct
Reviewers: Ivan Inkov (BG) system is suppressed in the female, and germ cells develop on
1
SUJAN NARAYAN AGRAWAL, Associate professor, Department of Surgery. Late the surface of female gonads.
SBRKM Government Medical College, Jagdalpur(Bastar) C.G. PIN 494001. INDIA,
E-mail: [email protected] The urogenital system develops from urogenital mesoderm.
SUJAN NARAYAN AGRAWAL et al./ International Journal of Medical Reviews and Case Reports (2017) 1(2):23-27
Soon after the formation of three germ layers, the mesoderm In the 8t h week of gestation, the testis and mesonephros
layer is divided into several parts i.e. axial, paraxial, interme- are linked to the posterior abdominal wall by a peritoneal
diate and lateral plates. This intermediate mesoderm gives rise fold. As the mesonephros degenerates, a portion of this fold,
to urogenital system. [1] The testis develops before birth in the cranial to the testis, called the diaphragmatic ligament, also
dorsal abdominal wall. During the human fetal period, the testis degenerates; it then turns in to the cranial position of the
migrates from the abdomen to scrotum traversing the abdom- gonadal mesentery. This structure is known as the caudal
inal wall and the inguinal canal between 15t h th to 28t h -week gonadal ligament, which gives rise to gubernaculum testis.
post conceptions. The development of gubernaculum is the sin- Cranially the gubernaculum approaches the mesonephric
gle most important structure in the testicular migration process. duct, while distally it approaches the inguinal region. At
The gubernaculum is a cylindrical structure, covered by the peri- this moment, the future inguinal canal is still only space in
toneum from all the sides except posteriorly, where the testicular the musculature of the anterior abdominal wall, where only
vessels and vas difference passes to it. The proximal portion of mesenchymal tissue exists.
the gubernaculum is adherent to the lower pole of the testis and The gubernaculum is a cylindrical structure, covered by the
the epididymis. During testicular migration, these structures peritoneum from all the sides except posteriorly where the
move through the inguinal canal as a single unit. During the testicular vessels and vas deference passes. Macroscopically
8t h month it reaches the superficial inguinal ring, and during it looks like Wharton’s jelly of the umbilical cord. Histologi-
the 9t h month, it comes to rest at the bottom of the scrotum. In cally it is composed of undifferentiated cells with elongated
the scrotum it bulges into the lower part of the processus vagi- shapes, surrounded by large quantities of extracellular ma-
nalis, causing the invagination of the posterior wall of processus terial. It is impossible to identify smooth or striated muscle
vaginalis. cells in it, except in its distal end and the peripheral por-
The factors which contribute to the descent of testis includes tion. [9] The genital branch of the genitor-femoral nerve,
Gubernaculum testis, the differential growth of abdominal wall, provides, the nerve supply to gubernaculum.
intra abdominal pressure and temperature, Calcitonin gene re-
lated peptide(CGRP), male sex hormones, insulin like hormone Around the 8t h week of gestation, a portion of epithelium
3(INSL3) and maternal gonadotrophins. The descent of testis starts a small invagination from the coelomic cavity, across
may become erratic and gives rise to undescended testis, ec- from the gubernaculum, slowly penetrating its mesenchy-
topic testis, congenital hernia and hydrocoele etc. This paper mal structures. This invagination occurs bilaterally and is
discusses the process of migration, the role of Gubernaculum considered as a start of the vaginal process. The growth of
and consequence of the failure of this mechanism. the vaginal process divides the gubernaculum into three
parts;-
1. Testicular migration 1). The main gubernaculum, which corresponds to the
The testis develops before birth in the dorsal abdominal portion covered by the visceral layer of the peritoneum of
wall. The development begins as early as 6t h weeks of the vaginal process.
intrauterine life and is completed by the 5t h month of in- 2). The vaginal gubernaculum- which corresponds to the
trauterine life. [2] Testicular migration is a complex process portion that externally surrounds the parietal portion of the
wherein the testis migrates from its abdominal position to vaginal process.
bottom of the scrotum. [3] During the human foetal period,
the testis migrates from the abdomen to scrotum traversing 3). The infra vaginal gubernaculum that corresponds to the
the abdominal wall and the inguinal canal between 15t h caudal portion of the gubernaculum which has not been
and 28t h -week post conception (WPC). [4-5] invaded by the vaginal process. [10-11].Both the guber-
naculum and vaginal process change in harmony during
1.1 Intrauterine events testicular migration. The maintenance of this undifferen-
In the early months of the intrauterine life, the scrotum is tiated mesenchyme along the inguinal canal and scrotum
rudimentary. It has no cavity and testis is high-up on the is essential for the downward extension of the vaginal pro-
posterior abdominal wall. Testicular migration happens cess to occur, during which it follows the pathway, created
in two distinct phases. The first phase corresponds to the by the dilatation of the gubernaculum, forming the canal,
testicular migration from the abdomen to the internal in- through which the testis will reach to the scrotum.
guinal ring. Moreover, the second phase corresponds to the 1.3 Proximal gubernaculum
transposition of testes through the inguinal canal until their
The proximal portion of the gubernaculum is adherent to
definite arrival at the scrotum. [6]
the lower pole of the testis and the epididymis, during testic-
The exact timing of the testicular migration is controversial. ular migration these structures move through the inguinal
Various authors have quoted this from as early as a 17t h canal as a single unit. According to Johansson and bloom,
week up to a 24rth week, but it is more agreed upon that [12] in this situation the proximal gubernaculum always ad-
the passage of testis through inguinal canal occurs very heres to the end of the vaginal process. Many studies have
quickly. shown that the changes in the proximal insertion of the gu-
1.2 The development of gubernaculum The development bernaculum are associated with epididymal anomalies and
of gubernaculum is the single most important anatomical can contribute to the occurrence of cryptorchism. [13] The
structure in the testicular migration process. Using contrac- proximal portion is necessary by uniting the scrotal region
tion and shortening, it imposes traction forces on the testis. and serving as a guide for testicular migration. Not only
The gubernaculum starts to develop in the human foetus that, but it also limits the mobility of testis and prevents
during the 6t h week of gestation, the same period when the testicular torsion.
germinative cells are arriving at the genital ridge. [7-8] 1.4 Distal gubernaculum
The insertion site of the distal gubernaculum is one of the invaginates the processus vaginalis, from the posterior as-
factors involved in testicular ectopia. [14] Many studies pect, the posterior border is not covered by tunica.
have shown that the distal gubernaculum has six tails or The orifice of communication between the cavity of the pro-
extensions. i.e. abdominal, pubo-penile, femoral, perineal, cessus vaginalis and the peritoneal cavity is usually closed
contra lateral scrotal and scrotal.[15-16] It is speculated that before birth. The cavity of the part of the processus that
these tails of the gubernaculum exists during the beginning extends from the deep ring to the scrotum is usually obliter-
of the foetal development and disappears during testicular ated during the first month after birth, and its wall becomes
migration. If any of these extensions of the distal portion a fibrous thread ( the vestige of the processus vaginalis).
exists, the individual may develop testicular ectopia. [16] The part of the processus that reaches the scrotum persists
The most accepted theory to explain testicular ectopia are;- as the tunica vaginalis testis. The cavity of tunica vaginalis
1). Failure of gubernaculum to dilate the inguinal canal, is now entirely separated from the peritoneal cavity, but
enabling the testis to migrate through other pathways and its wall is still connected with the peritoneum for a longer
not to the scrotum. or shorter time, by the vestige of the processus. However,
the vestige undergoes atrophy from below upwards and
2). Invasion of the gubernaculum by abdominal wall fascia frequently, as already mentioned, it entirely disappears.
near the inguinal canal, blocking the passage of testis to the
scrotum and diverting it to ectopic sites. 2. The factors responsible for, testicular descent.
3). The existence of multiple distal insertions of the guber- The exact cause of descent of testis and processus vaginalis
naculum testis, guiding the testis to ectopic sites. So the is still a subject of dispute. However, the following factors
most accepted theory to explain testicular ectopia is the contribute to the descent of testis;-
existence of multiple distal insertions of the gubernaculum. 2.1. Gubernaculum testis; - This is considered to be the
1.5 The process of testicular migration most important musculo-fibrous structure and factor, which
brings and guide testis to the scrotum. Failure of its proper
The different parts of the gubernaculum undergo various mechanism gives rise to cryptorchism and ectopic testis.
changes during the testicular migration. The vaginal and [20]
infra vaginal portion become proportionately longer as the
testis starts to descend into the scrotum. Their diameter 2.2. Intra abdominal pressure; - tends to displace the testis
also increases which helps to dilate the inguinal canal and downwards. In undescended testis, continuous intra ab-
to allow testis to pass through it. The growth of the guber- dominal pressure causes atrophy of testis, and they are
naculum can be divided into two phases. In the first phase, more prone to developing malignancies.
its volume increases and the second phase, it decreases in 2.3. The differential growth of abdominal wall.
size, coinciding with the complete descent of the testis. [17] 2.4. Intra abdominal temperature; - the temperature in the
The cremaster muscle presents structural alterations during abdomen is two degrees more than the scrotum. If the testis
this period as well. The rhythmic contraction of this muscle remains inside the abdomen (as in undescended testis), it
guides the testis into the scrotum. hurts spermatogenesis.
The first phase is characterised by the pronounced cell multi- 2.5. Calcitonin gene related peptide (CGRP);- It is a neuro-
plication and accumulation of glycosaminoglycans, mainly transmitter secreted by the genitofemoral nerve, supplying
hyaluronic acid. These substances act as hydrophilic agents the muscular fibres of gubernaculum testis.
and raise the quantity of water. There is also increase of
2.6. Male sex hormones-In the male Y chromosome has a
extracellular material. The presence of myoblast intensifies,
gene for testis determining factor (TDF). Under the influ-
and there are changes in the number and arrangement of
ence of TDF, the development of gonads proceeds in the
the collagen fibre and alteration in the elastic system.
male direction and testis develops, while in a female in
In the second phase, the gubernaculum shrinks, particularly the absence of TDF the development of gonads proceed in
in length, frequently accompanied by the descent of the female guidance and ovaries develop.
testis. This phenomenon appears to be androgen dependent 2.7. Sexual development begins at the approximately
and brings substantial degradation of the glycosaminogly- 8t h week of gestation with the production of Mullerian-
cans previously accumulated in the extracellular material, inhibiting substance/anti-Mullerian hormone (MIS/AMH).
with resulting dehydration of this space and condensation It triggers Mullerian duct regression along with testos-
of the gubernaculum. Understanding the relationship be- terone, stimulating the Wolffian duct to persist and to form,
tween regression of the gubernaculum and descent of testis the epididymis, vas deference and seminal vesicles. [21-22]
is vital to the comprehension of, how androgens control Mullerian inhibiting substance (MIS/AMH) is a glycopro-
testicular migration. Studies have demonstrated the asso- tein dimer (mw 140,000) produced by the Sertoli cells. It is
ciation of deficiency of androgens to failed regression of also secreted in the Wolffian duct and then diffuses laterally
gubernaculum and thus leading to cryptorchism. [18-19] into the adjacent Mullerian duct to trigger its regression
During the 8t h month it reaches the superficial inguinal in the male. It may also have some secondary role in the
ring, and during the ninth month, it comes to rest at the development of gubernaculum and has post natal functions
bottom of the scrotum, where it bulges forward into the in the ovarian cycle. [23]
lower part of the processus vaginalis and invaginates the 2.8. Insulin like hormone 3 (INSL 3): - It is a protein with
posterior wall of the processus. Two layers of it now cover homology to the insulin that is produced by the Leydig
the testis. The coverings are called tunica vaginalis having cells. It stimulates the growth of the gubernaculum, which
outer parietal layer and inner visceral layer. Since the testis is important for the first phase of the testicular descent. [24]
2.9. Calcitonin gene related peptide (CGRP); - it is a neuro- e. Scrotal- located high up in scrotum.
transmitter secreted by the genitofemoral nerve, supplying
4.1.2. Ectopic testis
the muscular fibres of gubernaculum testis. It provides a
chemotactic gradient for the gubernaculum to follow. [25] In this condition, the testis frequently descends, from the
2.10. Maternal gonadotrophins; - However the gubernac- posterior abdominal wall, negotiate through the deep in-
ulum testis is considered an important factor for the for- guinal ring to the inguinal canal and comes out through
mation of processus vaginalis and in guiding testis to the superficial inguinal ring. However, it fails to reach to the
scrotum. The descent is also required for the propagation of scrotum.It deviates from its path of normal descend. It is
the species since intra abdominal pressure and temperature thought to be due to many tails of gubernaculum. There is
has an inhibitory effect on spermatogenesis. a failure of primary gubernaculum functioning and activa-
tion of one of its “tail”, which pulls the testis to an unusual
Thus we see that the testicular descent is a complex and position. Thus ectopic testis may be found at abdominal,
multifactorial event, and cryptorchism should be viewed as pubo-penile, femoral, perineal, or contra lateral scrotal po-
a disease with multiple aetiologies. sitions.
3. Why the ovaries do not descent to labia? (In Females) 4.2. Congenital hydrocele/hernia
The initial phase of descent of ovaries is similar to that
The processus vaginalis is a diverticulum of the abdominal
of the testis. There is the formation of gubernaculum of
peritoneum. It obliterates once the descent of testis is over.
ovaries; there is also the formation of the inguinal canal
If it is not obliterated as happens, then it may give rise to
and processus vaginalis. In females, the gubernaculum is
following pathological conditions:-
attached to developing uterus close to fallopian tubes so it
may be seen as two parts of gubernaculum. a. Congenital hydrocele: - It is a persistent congenital sac
1). The first part extends from the lower pole of the ovary of the peritoneum, with a tiny communication between
to the uterus. This part forms the ligament of the ovary. processus vaginalis and peritoneal cavity. It may contain
fluid and thus presents as congenital hydrocele.
2). The other part extends from uterus to labia majora.
This part passes through inguinal canal and forms round b. Hydrocoel of cord: - The processus is obliterated at above
ligament of the uterus. and below having a cystic space shut off from peritoneal
cavity above and cavity of tunica below. This is called the
The first part of descent of ovary up to the pelvic brim takes
encysted hydrocele of cord.
place because of the growth of posterior abdominal wall
and pelvis. c. A congenital hernia: -The processus vaginalis fails to
Ovaries are prevented from entering the inguinal canal obliterate, and the communication to peritoneal cavity is
because of the attachment of gubernaculum to the uterus sufficiently significant to permit passage of abdominal con-
when uterus descends into the pelvic cavity the ovaries are tent into the sac. This condition is called congenital inguinal
also carried along with it. Since the ovaries do not traverse hernia.
the inguinal canal, the processus vaginalis and inguinal
canal remain small and rudimentary. 5. What this paper contributes/Highlights
4. The pathology related to descent of testis • It highlights the mechanism of testicular descent to the
The descent of testis may become erratic and gives rise to scrotum, which is essential for proper spermatogenesis
following pathological conditions:- and propagation of species.
• It explains why the blood supply of testis and epi-
4.1. Related to gubernaculum testis:-
didymis comes all the way from the abdomen.
1). Undescended testis. • It describes the mechanism and probable causation of
2). Ectopic testis. ectopic and undescended testis.
• It also highlights the factors responsible for a congeni-
4.2 Related to processus vaginalis:-
tal hernia and hydrocele.
1). Congenital hernia.
2). Congenital hydrocoel.
Authors’ Statements
4.1.1. Undescended testis
Competing Interests
The descent of testis may be arrested at any point in its The authors declare no conflict of interest.
journey, from lumber region to scrotum, depending upon
the arrest of descending and its abnormal location. [26-28]
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the mesonephros and of the gonads: a contribution to the torchidism: the state of the art in 2004. J Pediatr Surg, 2005;
development of the human gubernaculums (Hunteri). Anat 40:297–302.
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26. Susan Standring. Male Reproductive system. In: Chapter
11. Frey HL, Rajfer J. Role of the gubernaculum and Intra- 97, Neil R. Borley (ed). Gray’s Anatomy. 39t h ed. Edin-
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1988; 22:107-8. terns of undescended testis among primary school pupils
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WS.proximal insertion of gubernaculum testis in normal
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CASE REPORT
EFFICACY OF INTRAVENOUS IMMUNOGLOBULIN AND

PHOTOTHERAPY IN THE MANAGEMENT OF
EXTREME-HYPERBILIRUBINAEMIA: A CASE REPORT
Olufunke Bosede Bolaji∗ , Sandeep Dhamaraj∗∗ , Colin Lumsden∗∗ and Olusegun Joseph Adebami∗ ∗ ∗, 1
∗ Department of Paediatrics and Child Health, 1Federal Teaching Hospital, Ido-Ekiti, Nigeria., ∗∗ Royal Preston Hospital, Lancashire Teaching Hospitals NHS
Foundation Trust, United Kingdom., ∗ ∗ ∗ Department of Paediatrics and Child Health, Ladoke, Akintola University of Technology Teaching Hospital, Osogbo,
Nigeria.
ABSTRACT Background: Neonatal jaundice is one of the leading causes of neonatal morbidity and hospitalisations
worldwide, and in the developing countries particularly, it still represents one of the main causes of neonatal mortality.
While phototherapy is widely accepted as an effective treatment, adjunct modes of management such as the use of
Intravenous Immunoglobulin (IVIG) are not so widely utilised in developing countries like Nigeria. This present
case report is to show the usefulness of IVIG in the management of neonatal jaundice. Case summary: A case report
of a 42-hour-old Caucasian term male neonate with extreme-hyperbilirubinaemia secondary to ABO incompatibility
which was managed with a combination of guided phototherapy and Intravenous immunoglobulin (IVIG) is presented.
The patient responded well to treatment with no apparent immediate adverse effects. Conclusion: The implications
for clinical practice regarding reduction in the frequencies of Exchange Blood Transfusion (EBT) and the duration of
phototherapy are at this moment presented.
KEYWORDS Extreme-hyperbilirubinaemia, Intravenous immunoglobulin (IVIG), Guided phototherapy
phototherapy (PT) have traditionally been used to treat jaun-

dice so as to avoid the associated neurological complications of
Introduction severe hyperbilirubinaemia.[1] EBT is not without risk. Hence
Intravenous Immunoglobulin (IVIG) has been suggested as an
Neonatal jaundice is one of the most common causes of mor- alternative therapy for isoimmunehaemolytic jaundice to reduce
bidity in newborns globally, and severe neonatal jaundice is a the need for exchange transfusion.[3] Some of the complica-
frequent cause of hospitalisation or readmission for special care tions of exchange transfusion include haemodynamic instability,
in the first week of life. [1] The approaches to management apnea, pulmonary haemorrhage, thrombocytopaenia, coagu-
appear to differ considerably between developed and devel- lopathies, hypoglycaemia, hypocalcaemia, electrolyte imbalance,
oping countries. [1,2] Exchange Blood Transfusion (EBT) and vasospasm, vascular thromboses, hypertension, arrhythmias,
sepsis, necrotizing enterocolitis and bowel perforation with mor-
DOI: 10.5455/IJMRCR.10.5455/IJMRCR.immunoglobulin-and-phototherapy-in-the- bidity rates varying from 2.8-5.2% per procedure.[3-5]
management-of-extreme-hyperbilirubinaemia While international guidelines for the management of neona-
First Received: June 03, 2017 tal jaundice are widely available and accessible globally; uniform
Accepted: July 15, 2017
practice guidelines are rare at all levels of healthcare delivery in
Editor-in Chief: Cvetanka Hristova (BG) Nigeria.[4] The threshold for EBT is often based on a TSB level
Reviewers: Ivan Inkov (BG) of ≥ 20mg/dL and two-third of that level for commencing PT
1
Dr.Olusegun JosephAdebami, Department Paediatrics and Child Health, College of for term babies.[5]
Health Sciences, Ladoke Akintola University of Technology, Osogbo, Nigeria and Intravenous Immunoglobulin (IVIG) is an alternative ther-
Neonatology Unit, Department Paediatrics and Child Health, Ladoke Akintola
University of Technology Teaching Hospital, Osogbo, Nigeria,
apy which may be effective in treating isoimmunehaemolytic
E-mail: [email protected] jaundice. In isoimmunehaemolysis, red blood cells are proba-
Olusegun Joseph Adebami et al./ International Journal of Medical Reviews and Case Reports (2017) 1(2):28-31
bly destroyed by an antibody-dependent cytotoxic mechanism
mediated by Fc receptor bearing cells of the neonatal reticuloen- Table 1 Timeline of care
dothelial system. The putative mechanism of IVIG action is a Level of jaundice
nonspecific blockade of Fc receptors resulting in a decline in Age Treatment offered
carboxyhaemoglobin levels. Carboxyhaemoglobin levels are a (TSB mmol/L)
sensitive index of haemolysis and hence immunoglobulin could Referral to hospital by
decrease haemolysis.[2] 42 hour 421
The use of IVIG as an adjunct for the management of ex- midwife
treme and severe hyperbilirubinaemia in developing countries Quadruple phototherapy
is uncommon due to non-availability of the drug and sometimes
the limited knowledge of its use. EBT is, therefore, a common 46 hour 433 IVIG 0.5mg/kg,
modality of treatment for severe neonatal jaundice in developing IVF
countries like Nigeria.[5,6] Literature and data are sparse from
these regions in the use of standardised guidelines and IVIG in 52 hour 404
the management of NNJ. Therefore, the present case is presented Phototherapy lamps reduced
to illustrate the use and response of IVIG in the management of 58 hour 354
extreme hyperbilirubinaemia. to 3
64 hour 222
Case report:
76 hour 211 Phototherapy
Complaint: A 42-hour-old Caucasian male neonate (gesta- Day 4 169 Phototherapy discontinued
tional age 40 weeks, birth weight 3009g) presented at the neona- Day 5 150 Patient discharged
tal unit of Royal Preston Hospital, Lancashire Teaching Hospitals
NHS Foundation Trust, the UK with jaundice noticed on the 3r d Follow up at clinic – Child
3 months
day of life by the community midwife at 36 hours of life during was neurologically normal
the routine home visit.
Activity was good, and no other systemic symptoms were no-
ticed. The patient was feeding well on exclusive breast milk. The obtained, and intravenous fluid was started at 120 ml/kg/day.
patient was born by normal vaginal delivery to a 33-year-old The patient was placed on nil per by mouth. IVIG was given
mother. The mother’s and baby’s blood group were O posi- at 0.5g/kg at admission. By 6 hours into admission, which was
tive and A positive respectively. He had been passing urine after the administration of IVIG, TSB had reduced to 404 mi-
and opening bowels since birth. There were no risk factors for cromol/L, (conjugated fraction 43micromol/L). When plotted
sepsis and no history of jaundice in the elder sibling. He had on the NICE guideline jaundice treatment graphs, this value
earlier been discharged from the postnatal ward at 24 hours of was four boxes below the exchange transfusion line. 12 hours
life. The total serum bilirubin (TSB) recorded by the midwife at into admission showed a further reduction in the TSB to 354
home during the home visit with transcutaneous bilirubinome- micromol/L (conjugated fraction 38) which was eight boxes be-
ter was 421 micromol/L. By the published NICE guidelines for low exchange transfusion line.Phototherapy lamps were thus
treatment threshold in neonatal jaundice, TSB value was on the reduced to 3. TSB 6 hours later was 250 micromol/L which was
exchange blood transfusion line. Hence, the baby was referred on the phototherapy treatment line. On the second day of admis-
to the neonatal unit for possible exchange blood transfusion. At sion, haematocrit was 14.9 g/dL, and reticulocyte was 13.99%.
presentation, he was severely jaundiced, not pale, afebrile, not Enteral feeds were restarted by the 2nd day of admission when
dehydrated, weight was 2800g, representing a 6% weight loss the TSB had dropped significantly to 222 micromol/L, and this
from birth weight. Systemic examination was normal, and there value was by now four boxes below phototherapy treatment line.
were no clinical signs of bilirubin encephalopathy. The next TSB value by the end of the 2nd day of admission was
211 mmol/L, which was seven boxes below the phototherapy
Diagnosis: Severe neonatal jaundice (with TSB at exchange treatment line. Phototherapy was then reduced to 2 lamps. TSB
transfusion line) secondary to possible ABO incompatibility was was 169 mmol/L on the 4t h day of life after which phototherapy
made. Investigation results showed baby’s blood group as A was discontinued.
Rhesus positive. Direct Coombs Test was strongly positive. To- Outcome:The patient made a full recovery, as judged by the
tal serum bilirubin (TSB) taken immediately on admission was clinical appearance and the neurologic examination and the
433 mmol/L; 4 hours after the referral value. Haemoglobin TSB values. He was subsequently discharged from the hospital
values were 15.5 g/dL and 14.9 g/dL on the 1st and 2nd day on the 5t h day of life. Neurodevelopmental follow-up in the
of admission respectively. Reticulocyte counts were initially outpatient clinic at three months of age was normal.
16.09%. Blood film on admission also showed numerous sphe-
rocytes with polychromasia. Blood glucose and gases were
Discusion:
largely normal and infection screens were not suggestive of in-
fection. Serum electrolytes and urea were also normal. Other Extreme hyperbilirubinaemia is defined as a TSB greater than
liver function tests were normal with albumin 36 g/L, Alkaline 428micromol/L (25mg/dL). [7] The patient’s total serum biliru-
Phosphatase 159 U/L and Alanine transferase (ALT) 12 U/L. bin (TSB) taken immediately on admission was 433 micromol/L,
Interventions:The patient was admitted and started on and this was due to ABO blood group incompatibility as the
quadruple phototherapy with three overhead phototherapy mother’s and baby’s blood group were O positive and A positive
lamps and a Bilibed. Umbilical arterial and venous accesses were respectively.
ABO hemolytic disease remains the most common cause of age and birth weight.In sharp contrast to the practice in most
severe and early jaundice in the newborns. ABO blood group high-income nations, national or local guidelines for the effective
incompatibility is present in 15-25% of pregnancies.[8] ABO management of severe hyperbilirubinaemia are rare in the de-
feto-maternal red blood cell incompatibility induces an immune veloping countries even though the disease burden is high. [16]
hemolysis after fetal transfer of haemolysing maternal anti-A or The absence of harmonised protocols either for classification or
anti-B. High levels of consequent unconjugated hyperbilirubine- management in most low and middle-income countries renders
mia may induce both acute and chronic neurological complica- it difficult if not impossible for comparisons between locations.
tions.[8,9] As regards the long/life term effect of our choice of manage-
Although phototherapy (PT) and exchange transfusion (ET) ment, the patient had no sign of bilirubin encephalopathy at
are widely used in the management of severe jaundice, there is admission nor at follow-up at three months. We, therefore, had
no standard or evidence basis for the thresholds for each inter- no reason to fear that the prognosis will not remain excellent.
vention. It is also unclear what the safe upper limit of bilirubin
level is or whether bilirubin level alone is a sufficient predic- Implications for practice
tor of the need for exchange transfusion or as a determinant of
neurodevelopmental outcome.[10] Many practitioners in devel- Administration of IVIG to newborns with significant hyperbiliru-
oping countries use double set up or intensive phototherapy binemia due to ABO haemolytic disease with positive direct
and defer EBT until the serum bilirubin has risen above high Coomb’s test combined with the use of PT reduces the need for
thresholds. Some paediatricians still use early rate of rising of EBT and the duration of PT. The efficacy and good tolerance
bilirubin as an indicator for ET.[11] prompt consideration of IVIG as a therapeutic adjuvant to PT
Intravenous human polyclonal immunoglobulin (IVIG) has in severe hemolytic hyperbilirubinemia due to ABO incompat-
been proposed for concomitant use with PT to reduce the need ibility. Since it appears safe, it may also have a role in special
for ET in ABO haemolytic disease with positive direct Coomb’s circumstances such as parental refusal for exchange transfusion,
test.[1] failed cannulation for EBT or where appropriate blood compo-
The exact mechanism of action of IVIG in HDN is still un- nents for exchange transfusion are unavailable.
known. IVIG is thought to decrease haemolysis by blocking This case report also encourages the employment of standard
Fc receptor sites of reticuloendothelial cells preventing lysis of protocols which are widely available in determining the need for
neonatal erythrocytes. IVIG competes with sensitised neonatal phototherapy or exchange blood transfusion as well as the use of
erythrocytes for the Fc receptor sites of the reticuloendothelial alternative treatments like IVIG in cases of extreme hyperbiliru-
system to prevent further haemolysis (competitive inhibition) binaemia with isoimmunization. This maximises the chances
thus suggesting that early administration of IVIG is necessary of a favourable outcome for patients with severe neonatal jaun-
for efficacy in immune hemolytic diseases of the newborn. [12] dice regarding reducing the need for EBT and the duration of
accompanying phototherapy.
Recognising the potential benefits of IVIG over exchange
transfusion is easy. The administration is less complicated and
less labour intensive. As well as being a less invasive therapy, Authors’ Statements
IVIG may also allow treatment of some infants in Level IIcentres
Competing Interests
or avoid delaying treatment while transferring infants to tertiary
The authors declare no conflict of interest.
centres for exchange transfusion. Administration of IVIG has
also been shown to reduce the duration of phototherapy. [13]
This may particularly be helpful in developing countries where References
health care cost is related to duration of stay in the hospital and
1. Burke BL, Robbins JM, Bird TM, Hobbs CA, Nesmith C,
this cost is largely out of pocket expenditure.
Tilford JM. Trends in hospitalisations in neonatal jaundice
Case reports and case series have reported the success of IVIG
and kernicterus in the United States, 1988-2005. Pediatrics
in the treatment of jaundice due to both Rhesus and ABO incom-
2009; 123:524-32
patibility.A systematic review of the use of IVIG in hemolytic
disease of the newborn showed that significantly fewer infants 2. National Institute for Health and Clinical Excellence.
required EBT when IVIG was administered in combination with Neonatal Jaundice. (Clinical Guideline 98); 2010. Avail-
PT compared to those who had PT alone. Also, hospital stay and able from http://www.nice.org.uk/CG98. Last accessed
duration of phototherapy were reduced. [14] However, there 8th August 2016
are still debates on the routine use of IVIG in ABO hemolytic
disease of the newborn as some studies have failed to show the 3. Alcock GS, Liley. Immunoglobulin infusion for isoimmune-
efficacy of IVIG in reducing the need for EBT. The reasons for haemolytic jaundice in neonates.Cochrane Database Syst
this discrepancy have not been explained but it should be noted Rev. 2002; 3: CD003313
that in some of the studies that failed to show significant effects,
IVIG was used more or less prophylactically for all apparently 4. BO Olusanya, FB Osibanjo, CA Mabogunje, TM Slusher,
immunized infants, whereas in the studies that reported benefits, SA Olowe. The burden and management of neonatal jaun-
IVIG was used exclusively as a rescue therapy in infants headed dice in Nigeria: A scoping review of the literature. NJCP
for EBT.[15] 2016;19:1-17.
Numerous guidelines for the management of neonatal jaun-
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them. The AAP and Norwegian guidelines all state explicitly the change blood transfusions done for neonatal jaundice in
thresholds for commencing or stopping phototherapy as well Abakaliki, South Eastern Nigeria. Journal of Clinical Neona-
as those for EBT. These guidelines also vary for both gestational tology. 2012;1:34-37
6. Owa JA, Ogunlesi TA. Why are we still doing so many
exchange blood transfusion for neonatal jaundice in Nigeria.
World J Pediatr. 2009; 5:51-5
7. Nigerian consensus statement on newborn jaundice. Man-

agement of jaundice and prevention of extreme hyperbiliru-
binemia in infants ≥ 35 weeks gestation. Nigerian Expert
Committee for severe hyperbilirubinemia (For NISONM)
8. Urbaniak SJ. ADCC (K Cell) lysis of human erythrocytes

sensitized with Rhesus alloantibodies.II. Investigation into
mechanism of lysis. Br J Haematol. 1979;42:315-328
9. Senterre T, Minon JM, Rigo J. Neonatal ABO incompati-

bility underlies a potentially severe hemolytic disease of
the newborn and requires adequate care. Arch Pediatr.
2011;18:279-82
10. Ahlfors CE. Criteria for exchange transfusion in jaundiced

newborns. Pediatrics 1994; 93:488-94
11. Greco C, Arnolda G, Boo NY, Iskander IF, Okolo AA,

Rohsiswatmo R, et al. Neonatal jaundice in low- and
middle- income countries: lessons and future directions
from the 2015 Don Ostrow Trieste Yellow Retreat. Neona-
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12. Cortey A, Elzaabi M, Waegemans T, Roch B, Aujard Y. [Ef-

ficacy and safety of intravenous immunoglobulins in the
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compatibility: a meta-analysis]. Arch Pediatr. 2014; 21:976-
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13. Miqdad AM, Abdelbasit, Shaheed MM, Seidahmed MZ,

Abomelha AM, Arcala OP. Intravenous immunoglobulin G
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14. R Gottstein, RWI Cooke. Systematic review of intravenous

immunoglobulin in hemolytic disease of the newborn. Arch
Dis Chil Fetal Neonatal Ed 2003;88: F6-F10
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Yun boo, Iman F Iskander, Maria Fernanda B de Almeida,
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Paediatrics 2015;15:39
CASE REPORT
SURGICAL MANAGEMENT OF SEVERE TRAUMA

INVOLVING GROUP OF MUSCLES AROUND THE
BRISKET OF DONGOLA BREED OF HORSE: A CASE
REPORT
Olaifa A.K∗,1 , Oguntoye C.O∗ and Olatunji-Akioye A.O∗
∗ Department of Veterinary Surgery and Radiology, University of Ibadan, Nigeria.
ABSTRACT Background: Equine wound management is challenging to the veterinarian in practice. This paper presents
the successful management outcome of a large wound on the brisket of a seven and half years old Dangola Stallion horse
sustained from a road traffic accident. Case Summary: There was traumatic shearing off of the skin including the dermis
and fascia of the thoracic inlet beginning from the ventral caudal end of the neck to the cranial border of both shoulders
and there were two open wounds on the medio-cranial aspect of the right tibial region just below the elbow joint. All
physiological parameters were within normal range. Continuous suture and interrupted patterns were used during the
surgical procedure until all the exposed muscles were apposed. Conclusion: The surgery was successful, with good post
operative care, the wound healed properly.
KEYWORDS Horse, wound, accident, surgery, Dongola
remain a challenging clinical problem, with early and late com-

plications presenting a common cause of morbidity and mor-
tality (Natarajan et al., 2000). A wound is a breakdown in the
Introduction protective function of the skin or loss of continuity of epithelium,
Wounds constitute the most commonly encountered injuries in with or without loss of underlying connective tissues, muscles,
equine practice (Westgate et al., 2010; Pollock, 2011). Due to the nerves, bones following injury to the skin, surgery, a blow, cut,
horse’s nature and the environment in which it lives, wounds chemicals, heat, cold, friction, shear force, pressure or diseases
frequently involve a significant amount of tissue trauma. Legs such as leg ulcers or carcinomas (Velnar et al., 2009; Fernandez
caught in fences, panels, wire, or gates are a frequent occurrence, and Griffiths, 2015). Wound healing is a homeostatic mecha-
as are lacerations from steel siding, trailer accidents, kicks, and nism for restoration of physiological balance and is triggered by
riding accidents. As a result, equine ambulatory practitioners the interruption of the connection between adjacent cells or cell
typically see a relatively large number of cases presenting for death. The healing process consists of a sequence of overlapping
wound care. Enormous variation exists in treatments, medica- events including inflammatory responses, regeneration of the
tions, bandages, and bandaging techniques applied to wounds epidermis, shrinkage of the wound and finally connective tissue
in horses (Theoret, 2008; Gomez, 2008). In pathology, wounds formation and remodelling (Leaper and Harding, 1998; Choucair
and Phillips, 1997).
DOI: 10.5455/IJMRCR.dongola-breed-of-horse-severe-trauma-surgery
Indeed, it seems that of all the species dealt with by veterinary
First Received: May 13, 2017 surgeons, the horse is particularly prone to wounds. Reasons
Accepted: June 01, 2017 for higher incidence of wounds in horses than other species may
Manuscript Associate Editor: George Baytchev (BG) be due to the conditions in which horses are kept, the type of
Editor-in Chief: Cvetanka Hristova (BG) work they are involved in, and the potentially “flighty” equine
Reviewer: Ivan Inkov (BG)
1
[Olaifa A.K, Department of Veterinary Surgery and Radiology, University of Ibadan,
temperament. (Pollock, 2011).Wound infection is a challenge to
Nigeria Email:[email protected] wound management and wound infection and dehiscence can
A.K Olaifa et al./ International Journal of Medical Reviews and Case Reports (2017) 1(2):32-35
occur in both surgically or trauma-induced wounds (Carter et al.,
2003; Westgate et al., 2010). Factors that influence infection in-
clude integrity of the wound and its perfusion, the wound repair
process, bacterial challenge and host immunity. The manage-
ment protocol employed in equine wound may also determine
the outcome of such wounds. Equine wound management is
challenging to the veterinarian in practice (Carter et al., 2003).
This paper presents the successful management outcome of a
large wound on the brisket of a horse sustained from a road
traffic accident involving both the horse and its rider.
Case Presentation
A seven and half years old Dangola Stallion was presented to the
veterinary Teaching Hospital of the University of Ibadan follow-
ing a road traffic accident involving it and its rider. The horse,
owned by the mounting department of the State Police, was
ridden on the highway and had collided with an on- coming ve-
hicle. There was traumatic shearing off of the skin including the Figure 2: Wound repair in progress
dermis and fascia of the thoracic inlet beginning from the ventral
caudal end of the neck to the cranial border of both shoulders suture pattern until all the exposed muscles were apposed. The
and there were two open wounds on the medio-cranial aspect of edges of the skin flap were trimmed and closed with 2-0 nylon
the right tibial region just below the elbow joint (figure 1). The suture (Ethicon, USA), using simple interrupted suture and con-
horse also favoured both hind limbs. The temperature, respira- tinuous patterns. The two open wounds on the medio-cranial
tory rate, heart rate and pulse rate were 38.5º C, 72 breaths/min. aspect of the right tibial region just below the elbow joint were
50 beats/min and 52/min respectively. All were within nor- also sutured using 2-0 nylon suture (Ethicon, USA), with simple
mal range. The mucous membrane was pink. Its weight was interrupted suture pattern.
estimated as 220kg.
Post-operative care
Tetanus toxoid was administered 1500i.u and penicillin- strepto-
mycin antibiotic. (PENSTREP 20/25, Kepro B.V., Holland at a
dosage of 1ml/25kg for 5 days) intramuscularly. Oxytetracycline
spray (Holland) was sprayed on the wound after closure (fig-
ure2). The sutures were removed a week later following wound
dehiscence although significant wound contraction had taken
place (figures 3 and 4).
Figure 1: Wound presented to the Veterinary Teaching Hospital.
Management
The horse was placed on intravenous fluid (15litres Hartman’s
Solution, Danax®) and given xylazine hydrochloride (XYL-M2®,
VMD, Holland) intramuscularly for both sedation and analgesia
at a dosage of 2mg/kg body weight.
Figure 3: Wound repair completed.
Surgery
The wounds were thoroughly irrigated with normal saline to
Discusion:
remove all dirt and all loose and unviable flesh was trimmed.
Following site infiltration with lignocaine hydrochloride, the The wounds of this horse were closed primarily. Primary re-
exposed muscles were closed layer by layer (figure2) using 2-0 pair of the wound is the preferred treatment for wounds that
and 1-0 chromic catgut sutures (Ethicon, USA), using continuous involve detachment of skin with maintenance of an intact blood
Table 1 Timeline
Dates Medical History Diagnostic testing Interventions
No previous case None None
Physical examination,
12/02/2017 Wound from road accident Respiratory rate, Heart Surgical wound repair
rate, Rectal temperature
References
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horse wounds. Wounds should be assessed quickly and care- 11. Singer ER, Saxby F, French NP. A retrospective case-control
fully and aseptic surgical techniques meticulously employed. study of horse falls in the sport of horse trials and three-day
Post-operative care was done conscientiously and appropriate eventing. Equine Vet J. 2003; 35(2):139–145. 7.
antibiotic treatment regimens employed.
12. Hernandez J, Hawkins DL. Training failure among yearling
horses. Am J Vet Res. 2001; 62(9):1418–1422.
COMPETING INTERESTS
13. Collins MN, Friend TH, Jousan FD, Chen SC. Effect of
The authors declare no conflict of interest. Written informed density on displacement, falls, injuries, and orientation
consent obtained from the patients for publication of this article during horse transportation. ApplAnimBehav Sci. 2000;
and any accompanying images. 67(3):169–179.
14. Cruse PJ, Foord R. The epidemiology of wound infection.
A 10-year prospective study of 62,939 wounds. SurgClin
North Am. 1980; 60(1):27–40. 15.
15. PB, Levy J, Dowd E, Eliot J. Surgical wound infection rates

in dogs and cats: Data from a teaching hospital. Vet Surg.
1988; 17(2):60–64.
16. Natarajan S, Williamson D, Stiltz AJ, et al: Advances in

wound care and healing technology. Am J Clin Dermatol
2000; 1: 269–275.
CASE REPORT
A CASE REPORT OF DELAYED DIAGNOSIS OF

UTERINE RUPTURE FOLLOWING VAGINAL DELIVERY
Vanessa Falé Rosado∗, 1 , Sara Rocha∗ , Catarina Vasconcelos∗ , Maria Luisa Martins∗ and Maria José Alves∗
∗ Maternidade Dr. Alfredo da Costa, Rua do Viriato, Lisbon, Portugal.
ABSTRACT Introduction: A uterine rupture is still a rare event, but its incidence appears to be increasing, even in the
unscarred uterus. In our case, the uterine rupture presented itself in an unscarred uterus and after a vaginal delivery.
Case report: A 36 years old women with three previous normal deliveries, comes to our hospital for assistance at 32
weeks with a poor pregnancy surveillance. After diagnosing Gestational Diabetes, she is admitted for therapeutic
adjustment. She is discharged after achieving metabolic control but comes back a few days later with a stillbirth, born by
vaginal delivery. Six days later she presents with: fever and pain; anaemia leukocytosis and a heterogeneous image on
ultrasound. However, was decided to start intravenous antibiotics before choosing for surgery. Her condition worsens,
and an exploratory laparotomy is done: a posterior uterine wall rupture that required a hysterectomy. Conclusion: Risk
factors for uterine rupture were present (maternal age over 35, higher parity, fetal macrosomia) but the absence of any
symptom, the regular examination after delivery, and mostly, an unscarred uterus, resulted in a delay in the diagnosis
of more than one week, leading to catastrophic consequences: hysterectomy. This case reminds us that uterine rupture
happens not only in case of previous uterine surgery, and these cases seem to be increasing because of the increase in
other risk factors: advanced maternal age and diabetes with resulting fetal macrosomia.
KEYWORDS uterine rupture, hysterectomy, postpartum period
maternity). However, in our case, no previous uterine surgery

was reported. This aspect combined with the lack of any symp-
tom, resulted in a delay of more than one week in the diagnosis,
Introduction
which in turn led to disastrous consequences - hysterectomy. Bet-
Uterine rupture represents a breach in the integrity of the uter- ter methods for predicting this disaster complication are needed,
ine muscle, usually during labour although it can also happen especially considering its increasing incidence also in the un-
before. It has been considered an entity with dramatic conse- scarred uterus, possibly due to the growing of other risk factors
quences both for the mother and the fetus, leading to severe like advanced maternal age and gestational diabetes with fetal
haemorrhage on the mother and hypoxia in the fetus. Because macrosomia.
it can have no symptoms, the diagnose may be delayed aggra-
vating the prognosis. This obstetric emergency, considered rare,
seems to be increasing due to the rising rate of uterine surg- Case report
eries like cesareans and myomectomies (due to the delaying in A thirty-six years old woman, melanodermic, natural from
Africa, came to Lisbon, Portugal at 32 weeks of pregnancy
DOI: 10.5455/IJMRCR.uterine-rupture-delayed-diagnosis
to have the remaining pregnancy surveillance and delivery in
First Received: July 07, 2017 Maternidade Dr Alfredo da Costa. Her personal and family
Accepted: July 22, 2017 background were irrelevant. As for previous deliveries, she
Manuscript Associate Editor: George Baytchev (BG) mentioned three eutopic deliveries: in 2002 a healthy baby
Editor-in Chief: Cvetanka Hristova (BG) with 3800grs, in 2004 she referred a late premature delivery
1
Maternidade Dr. Alfredo da Costa, Rua do Viriato, Lisbon, Portugal.,
of a healthy baby with 3400grs and finally in 2014, a pregnancy
[email protected] complicated with gestational diabetes that resulted in a newborn
Vanessa Falé Rosado et al./ International Journal of Medical Reviews and Case Reports (2017) 1(2): 36-40
Table 1 Analysis of patients’ clinical evolution on antibiotic
therapy
Day 0 Day 1 Day 2 Day 3 Day 5
Hb (gr/dL) 9,8 8,3 8,8 7,8 8,1 Table 2 Timeline of the patient.
WC (cells/µL) 26 180 17 500 16 500 13 000 17 200 Dates Relevant Past Medical History and Interventions
Vaginal delivery in Africa, newborn with 3800 grs, without any interferences during
2002
N (%) 91 89 88 84 86 pregnancy, intrapartum or postpartum
Vaginal delivery in Africa, newborn with 3400 grs, without any interferences during
CRP (mg/L) 397 316 315 239 260 2004
pregnancy, intrapartum or postpartum
US image size 105x95x70 116x99x59 Vaginal delivery in Africa, newborn with 3200 grs, diagnosis of Gestational Diabetes
2014
without posterior reclassification test
Summaries from
Dates Diagnostic Testing
Initial and Follow-up Interventions
(in 2016) (including dates)
Visits
The first appointment in
Ultrasound was done in
our hospital; none of the
Portugal on 18 July Analysis with 75grs glucose
22 July previous exams done in
revealing a healthy baby tolerance test was requested
Africa, were available for
with 32+5 weeks
consultation
-Glucose tolerance
test positive: Admission to Maternal-fetal
35+5 weeks 191/238/339 Infirmary for surveillance and
12 August
Asymptomatic -Ultrasound that day: therapeutic adjustment with
fetus on 95th both insulin and metformin
percentile
Adequate metabolic
37+1 weeks control and daily Cesarean scheduled to 6 of
22 August
Asymptomatic cardiotocography September
showing fetal well being
38+2 weeks Breech delivery 10 hours later:
30 August Stillbirth on ultrasound
No fetal movements Dead fetus with 4525 grs
Asymptomatic
Figure 1: Ultrasound image at emergency department, eight
Clinical observation 48hrs after delivery, she is
days after delivery (U – uterus; HI – heterogeneous image) 1 September normal: normal lochia, discharged from Hospital with
normal uterine involution metabolic control
and painless examination
with 3200grs. A reclassification test of gestational diabetes was -Ultrasound: image
not done. All pregnancies were with the same partner. In her with heterogeneous
first appointment in Lisbon at 32+ 5 weeks, it was decided to echogenicity was
repeat a glucose tolerance test (75grs glucose with glycaemia 8th day after delivery
found over the
uterine fundus, with
measurement at 0, 1 and 2 hours). The results were positive: 191, Fever, pelvic pain and Admitted to hospital and
105x95x70 mm
239 and 339 mg/dL. An ultrasound was performed: the fetus 7 September foul smelling vaginal
-Anemia(9,8gr/dL),
initiated ampicillin, gentamicin
was in the 95t h percentile (Hadlock et al) and the amniotic fluid discharge, which was
leukocytosis (26
and clindamycin IV
confirmed by observation
level was normal. She was admitted to the hospital for maternal- 180/µL), neutrophilia
fetal surveillance, and glycemic control was achieved with diet, (91%) and elevated
insulin and metformin. She was released at 37+ 1 with good C-reactive protein
(397mg/L)
metabolic control; a new appointment and elective cesarean for
-Blood test
39 weeks were scheduled. Improvement of
improvements,
symptoms and clinical
At 38 weeks she came to the emergency department because -Ultrasound with
8-10 September observation: pyrexia, no Maintain therapeutic
of the absence of fetal movements for the last 12 hours: she was stable image
pain at fundal uterine
diagnosed with stillbirth. She went into spontaneous labour, palpation
(without increases in
size)
and 10 hours later, after a breech delivery, she had a stillborn
13th day after delivery
with 4525 grs. The family refused an autopsy. Deterioration of both Exploratory laparotomy: vast
5th day with antibiotic
After 48hrs of inpatient observation, she was discharged from 12 September
Clinical deterioration:
blood test and uterine rupture of posterior wall
ultrasound image and a hysterectomy was decided
the hospital. fever, pain
She came back to the emergency department six days later Discharged from hospital after
with complaints of fever, pelvic pain and foul smelling vaginal therapeutic with meropenem,
during three weeks because of
discharge, which was confirmed by observation. On ultrasound,
multiresistant Klebsiella
an image with heterogeneous echogenicity was found over the 10 October
pneumoniae isolated in the
uterine fundus, measuring 105x95x70 mm. Blood analysis re- purulent exudate collected from
vealed low levels of haemoglobin (9,8gr/dL), leukocytosis (26 the abdominal cavity during,
180/µL), neutrophilia (91%) and elevated C-reactive protein surgery
(397mg/L). She was initiated on endovenous antibiotic therapy

with gentamycin, ampicillin and clindamycin. Analytic evolu-
tion is present in table 1.
Because of clinical deterioration, exploratory laparotomy was
Table 3 Medical History of eight cases of rupture (CS – cesarian; Leuco – leucodermic; melano – melanodermic; leucomelano -
leucomelanodermic)
Obstetric and Gynecological History
Dilatation
≥3 Time since last Other uterine
# Race Age & Nulliparous 1 CS ≥ 2 CS
Deliveries cesarian (in months) surgery
Curettage
1 Leuco 31 No No No Yes No 21 No
2 Melano 31 Yes (2) No No Yes No 48 No
3 Melano 29 No No No Yes No 3 No
4 Leuco 44 Yes (1) Yes No No No - No
5 Melano 26 No No No No 15 No
laparotomic
6 Leucomelano 29 Yes (3) No No Yes No 84
cerclage
7 Melano 36 No No Yes No - No
8 Leuco 37 No No No Yes No 72 No
decided. On surgical exploration a large rupture was found on The incidence is somewhat difficult to define due to the
the posterior uterine wall; there was purulent exudate all over scarcity of good quality studies since most of them are case re-
the abdominal cavity. A total hysterectomy was performed with ports. For this reason, uterine rupture rate varies from 1/10,000
unilateral adnexectomy due to technical difficulties. to 1/16,840-19,765, in the unscarred uterus. In the event of a
After surgery, this patient stayed in the hospital for almost previous cesarean, then the rate goes higher as expected: 1/1235-
one month due to the isolation of a multiresistant Klebsiella 4366 and as high as one /100. [3-8]
Pneumoniae, which responded to Meropenem. Intrapartum diagnosis poses many obstacles as Holmgren et
al. showed in a study of 36 cases of rupture.[9] This study shows
The mean age of this small sample was 33 years old (6 STD; which clinical sign led to the decision to perform a cesarean in a
min 26, max 44). Half were melanodermic and six had a previ- woman attempting trial of labor after cesarean (TOLAC): 30,5%
ous cesarean. No case of prior myomectomy. Most went into severe variable decelerations, 19,4% prolonged fetal bradycar-
spontaneous labour and most had a cesarean. dia, 22% maternal symptoms like pain and hypotension with
As for the rupture itself, some situations were identified as no fetal heart rate concerning changes. The maternal pain was
dehiscence and not as a complete disruption of the uterine wall. present in 25% of patients. This means that symptomatology is
This means that the serosa was still intact. The breach location very varied and no symptom appears to be more frequent. This
was mostly on the previous historiography, therefore on the difficulty in intrapartum diagnosis increases time to delivery
anterior uterine wall. with possibly serious consequences for the mother and neonate
Finally, the timing of the diagnosis was mainly intrapartum; According to this group, every additional minute to delivery
only one situation was diagnosed before, during the early third enhances the risk of a neonatal adverse outcome by 8,8%. As
trimester, with inferior segment measurement on ultrasound; for risk assessment, many clinical factors have been pointed
and the already mentioned rupture, that was diagnosed seven out: maternal age equal or higher than 35 years, parity equal or
days after delivery. higher than three, non-Western maternal origin, use of oxytocin,
Using the International Classification of Diseases 9t h Edition prostaglandins or transcervical balloon, scarred uterus (multi-
(ICD 66501 and 6651), we identified eight cases of uterine rupture ple previous cesarean section and type of previous hysterotomy
in our hospital from January 2010 to September 2016 out of a closure), fetal macrosomia (birth weight ≥ 4 kg).[10-12] The one
total of 29 797 (average rate of cesarean of 28,5%). that reunites most agreement is the classical cesarean. In a study
from 2012 by Gyamfi-Bannerman et al., three groups were com-
pared: prior myomectomy, prior classical cesarean and previous
Discussion lower segment transverse cesarean.[13] The main conclusions
The clinical significance of uterine rupture is growing, as its were that classical cesarean increased the risk of uterine rupture
prevalence appears to be rising. However, it represents many (adjusted OR 3,23), while previous myomectomy had no risk.
challenges for modern obstetrics: its risk factors identification, Despite these results, the group of prior myomectomy had some
its timely diagnosis and its prevention. important differences from the groups of classical cesarean and
Many authors consider two types of uterine rupture: dehis- lower segment transverse cesarean: lower rate of induction (1,1%
cence and total rupture. Rupture would represent a disruption of vs. 2,6% for classical and 17,8% lower segment), lower rate of
both uterine muscle and visceral peritoneum while in dehiscence vaginal delivery (0% vs. 5,9% for classical and 44,2% for lower
the peritoneum is intact.[1] Some authors prefer to consider de- segment).
hiscence a partial rupture as opposed to total rupture.[2] What is emerging, as a possibility to predict this disastrous
Table 4 Resume of type of delivery, type of rupture and neonatal outcomes of eight cases (time in labour in hours; CS – cesarian;
N.B.: newborn; AS – Apgar Score)
Labor Rupture Type Delivery
Spontaneous\ Time Site of
Timing of Other NB
# induction/ in Type uterine Type Weeks AS
diagnosis complications weight
cesarian labor disruption
anterior No neonatal
1 spontaneous 4 intrapartum dehiscence CS 38 2755 9//10
wall intercorrence
broad
ligament No neonatal
2 spontaneous 1 intrapartum rupture CS 39 2960 9//9
& lateral intercorrence
wall
3 spontaneous 2 intrapartum dehiscence CS 39 3870 9//10
wall intercorrence
posterior No neonatal
4 induction 6 intrapartum rupture CS 38 3000 9//10
wall intercorrence
elective anterior No neonatal
5 intrapartum dehiscence CS 39 3450 9//10
cesarian wall intercorrence
6 spontaneous 3 intrapartum rupture CS 35 2230 9//10
wall intercorrence
posterior Hysterectomy;
7 spontaneous 2 postpartum rupture Breech 38 4525 0//0
wall stillbirth
8 spontaneous 5 prepartum dehiscence CS 31 1580 9//9
wall intercorrence
complication, is lower uterine segment (LUS) measurement in morbidity (65 vs. 20%, p < 0,001).
women attempting TOLAC.
According to Bujold et al., the measurement should be done
between 35 and 38+ 6 weeks, using both the transvaginal and the As mentioned above, every obstetrician should be more and
transabdominal probes, to reduce interobserver variability.[14] more aware of this condition because recent analysis shows its
Three measurements should be done and the smaller considered. increasing incidence like this Norwegian study from 2015.[12]
However, many debates surround this theme as for what should Deliveries from 21 hospitals from 1967 to 2008 were selected and
be measured and what the right cut-off value to consider. For divided into four groups that correspond to four decades: 67-
some authors, full segment thickness is more predictive of uter- 77, 78-88, 89-99, 00-08. The incidence increased abruptly in the
ine scar rupture. This means measuring both the myometrium last decade: from 0.9/10 000 in the second decade (1978–1988)
and the bladder wall. However in a recent meta-analysis by to 6.1/10 000 in the fourth decade (2000–2008). This increase
Kok et al[15] measuring only the myometrium is as useful in was mostly because of the scarred uterus group: 14.2/10 000 in
predicting rupture. Another issue to debate is cut-off value: it the second decade to 66.8/10 000 in the fourth decade. Indeed,
has varied from 3,5 to 2 mm, and so far no precise value can be scarred uterus and labour augmentation with oxytocin were
recommended.[16] the main contributors to this increase in uterine rupture. In the
In this case report the rupture occurred in an intact uterus, intact uterus, after adjusting for prostaglandins and oxytocin
which makes it even harder to predict and diagnose and there- use, the OR remains almost the same.
fore with worst consequences as shown in a retrospective study
by Gibbins et al.[17] After exclusion of prednisone chronic use,
connective tissue disease and multiple pregnancies, 146 cases Finally, after a uterine rupture should we be tremendously
of uterine rupture were selected; 20 of them in intact uterus. scared of the next pregnancy? In a small study by Fox et al.
After comparing these two groups (intact vs. scarred uterus), with 60 pregnancies (20 after uterine rupture and 40 after uterine
the major difference is the delay in the uterine rupture diagnosis: dehiscence), pregnancy outcomes were very good with no cases
in women with a previous uterine scar, the delivery was more of repeated rupture or hysterectomy. [18] The only dehiscence
often a cesarean (91% vs. 58%), and for this reason the diagnosis repeated itself: 5% in the rupture group and 7,5% in the dehis-
was made intrapartum during the cesarean; as for intact uterus cence group. All pregnancies were ended before 40 weeks: in the
almost half women had a vaginal delivery and the diagnosis rupture group 75% ended between 36 and 37 weeks via cesarean
was delayed resulting in higher blood loss (2000 vs. 800mL, p and in the dehiscence group 93% between 36 and 39 weeks via
<0,001), hysterectomy rates (35 vs. 2,4%, p < 0,001) and maternal cesarean.
Conclusion 9. Holmgren C, Scott JR, Porter TF, Esplin MS, Bardsley T.
Uterine Rupture With Attempted Vaginal Birth After Ce-
Uterine rupture is a serious complication that can be more easily
sarean Delivery Decision-to-Delivery Time and Neonatal
suspected in the case of the previous cesarean; however, it can
Outcome. Obstet Gynecol 2012; 119: 725–31
also happen in the intact uterus and, even though it is a rare
event, factors like labour augmentation and prostaglandins use 10. Kaczmarczyk M, Sparen P, Terry P, Cnattingius S. Risk fac-
for labour induction can eventually lead to an increase in its tors for uterine rupture and neonatal consequences of uter-
prevalence. ine rupture: a population-based study of successive preg-
In this particular case, some risk factors can be identified nancies in Sweden. BJOG 2007;114:1208–14.
as fetal macrosomia, higher parity and maternal age over 35.
Still, she was discharged from the hospital 48hrs after delivery 11. Al-Zirqi I, Stray-Pedersen B, Forsen L, Vangen S. Uter-
with no complaints and after a normal examination. Only one ine rupture after previous caesarean section. BJOG
week later she comes back with pain and fever. This delay 2010;117:809–20
in diagnosis of more than one week after delivery led to very
serious morbidity like mentioned above by Gibbins et al. 12. Al-Zirqi I, Stray-Pedersen B, Forsen L, Daltveit A, Van-
gen S. Uterine rupture: trends over 40 years. BJOG
For all that it was mentioned, uterine rupture should be a
2016;123:780–787.
diagnostic hypothesis to consider, especially if risk factors like
older maternal age and previous uterine scar are present. An 13. Gyamfi-Bannerman et al. Risk of Uterine Rupture and Pla-
earlier clinical suspicion means a better obstetric outcome. centa Accreta With Prior Uterine Surgery Outside of the
Lower Segment. Obstet Gynecol 2012;120:1332–37
Authors’ Statements 14. Bujold E, Jastrow N, Simoneau J, Brunet S, Gauthier RJ.
Competing Interests Prediction of complete uterine rupture by sonographic eval-
The authors declare no conflict of interest. uation of the lower uterine segment. Am J Obstet Gynecol
2009; 201: 320.e1–6
References 15. Kok N, Wiersma IC, Opmeer BC, de Graaf IM, Mol BW, Pa-
jkrt E. Sonographic measurement of lower uterine segment
1. Landon MB, Hauth JC, Leveno KJ, et al. Maternal and
thickness to predict uterine rupture during a trial of labour
perinatal outcomes associated with a trial of labour after
in women with previous Cesarean section: a meta-analysis.
prior cesarean delivery. N Engl J Med 2004;351: 2581-9.
Ultrasound Obstet Gynecol 2014; 42: 132–139.
2. Nahun GG, Pham KQ. Uterine rupture in Pregnancy. Med- 16. Jastrow N, Chaillet N, Roberge S, Morency AM, Lacasse
scape Reference, 25 Mar, 2016 [www.reference.medscape. Y, Bujold E. Sonographic lower uterine segment thickness
com/article/275854-overview]. Accessed 2 March 2017. and risk of uterine scar defect: a systematic review. J Obstet
Gynaecol Can 2010; 32: 321–327.
3. Gardeil F, Daly S, Turner MJ. Uterine rupture in pregnancy
reviewed. Eur J Obstet Gynecol Reprod Biol 1994;56:107- 17. Gibbins KJ, Weber T, Holmgren CM, et al. Maternal and
10.2. Miller DA, fetal morbidity associated with uterine rupture of the un-
scarred uterus. Am J Obstet Gynecol 2015;213:382.e1-6.
4. Goodwin TM, Gherman RB, Paul RH. Intrapartum rupture
of the unscarred uterus. Obstet Gynecol 1997;89:671-3. 18. Fox et al. Pregnancy Outcomes in Patients With Prior Uter-
ine Rupture or Dehiscence. Obstet Gynecol 2014;123:785–9
5. Landon MB. Uterine rupture in primigravid women. Obstet
Gynecol 2006;108:709-10.
6. Zwart JJ, Richters JM, Ory F, de Vries JI, Bloemenkamp

KW, van Roosmalen J. Uterine rupture in The Nether-
lands: a nation- wide population-based cohort study. BJOG
2009;116:1069–78.
7. Landon MB, Hauth JC, Leveno KJ, Spong CY, Leindecker

S, Varner MW, et al.; National Institute of Child Health
and Human Development Maternal-Fetal Medicine Units
Network. Maternal and perinatal outcomes associated with
a trial of labour after prior cesarean delivery. N Engl J Med
2004;351:2581–9.
8. Landon MB, Spong CY, Thom E, Hauth JC, Bloom SL,

Varner MW, et al.; National Institute of Child Health and
Human Development Maternal-Fetal Medicine Units Net-
work. The risk of uterine rupture with a trial of labour in
women with multiple and single prior cesarean delivery.
Obstet Gynecol 2006; 108:12–20.
CASE REPORT
MISDIAGNOSIS OF AN ACARDIAC TWIN IN THE FIRST

TRIMESTER: A CASE REPORT
Catarina Vasconcelos∗, 1 , Vanessa Falé Rosado∗ , Rita Torres∗ , Ana Teresa Martins∗ and Álvaro Cohen∗
∗ Maternidade Dr. Alfredo da Costa - Centro Hospitalar Lisboa Central.
ABSTRACT Background:Twin reversed arterial perfusion (TRAP) sequence is a rare monochorionic twin pregnancy
complication. It should be suspected in the first-trimester ultrasound when one fetus has no cardiac activity in monochori-
onic pregnancies. Doppler study is essential for early diagnosis of acardiac twin. This manuscript shows the importance
of reassessing cases considered as vanishing twins in early pregnancy, to avoid a late diagnosis of acardiac twin, without
timely treatment. Case Summary: In this case, the assumption of the death of one fetus in the first trimester, delayed the
correct diagnosis of TRAP until 25 weeks, when the case was referred to our unit. We opted conservative management
with weekly ultrasound surveillance, looking for features of heart failure in the pump twin. After suspecting fetal
anaemia, at 32 weeks a healthy baby weighing 2100g was delivered along with an acardiac anceps with 518g. This is
a case of expectant management, for lack of another option, to a successful end. Conclusion: Although twin reversed
arterial perfusion (TRAP) sequence being a rare monochorionic twin pregnancies complication, it should be suspected in
the first-trimester ultrasound, when detected monochorionic pregnancies and one embryo/fetus has no cardiac activity.
Follow up in the fetal death of one twin in monochorionic pregnancies must be done with Doppler to look for an acardiac
twin. Because pump twin may develop a high-output cardiac failure and intrauterine fetal demise, for continuing
pregnancies without poor prognostic criteria, it is important to maintain weekly ultrasound surveillance, with attention
to the intervention criteria.
KEYWORDS TRAP sequence, acardiac fetus
through an artery-to-artery placental anastomosis, creating a

“reversed” circulation, which provides perfusion of mixed or
Introduction medium oxygenated blood from the pump twin to the recipient
twin. It results in poor perfusion and tissue necrosis, with the
TRAP sequence is a rare and exclusive complication of mono- evolution of a variety of structural abnormalities in the acardiac
chorionic twin pregnancies, with recent data on the incidence fetus.
of 2.6% in these pregnancies.[1] It is characterised by a fetus
that lacks cardiac structure and activity (acardiac twin), that The heart of the acardiac fetus may be completely absent
is perfused and hemodynamically dependent (parasite) on the (holo-acardius) or represented by a fundamental cardiac struc-
structurally normal co-twin (pump twin). This anomaly occurs ture (pseudo-acardius). Four distinct morphological types have
been described in literature: acardius acephalous: is the most
Copyright © 2019 by the Bulgarian Association of Young Surgeons common type (60-75%), with developed pelvis and lower limbs,
DOI: 10.5455/IJMRCR.acardiac-twin-first-trimester
First Received: July 09, 2017
may have arms, but thoracic organs and head are absent; ac-
Accepted: July 22, 2017 ardius anceps: the most differentiated type of acardiac twins
Manuscript Associate Editor: George Baytchev (BG) with established body and extremities, but only a partially
Editor-in Chief: Cvetanka Hristova (BG) formed head and face (20%); acardius acormus: in which only
Reviewers: Ivan Inkov (BG) cephalic structures were detectable, is a sporadic type (10%);
1
Catarina Vasconcelos, Maternidade Dr. Alfredo da Costa - Centro Hospitalar Lisboa
Central,
acardius amorphous: consists of a shapeless mass of tissue con-
E-mail:catv [email protected] taining no recognizable human structures (5%).[2]
Catarina Vasconcelos et al./ International Journal of Medical Reviews and Case Reports (2017) 1(2):41-45
CASE REPORT: continued EFW in the 50th percentile and at breech presentation.
No signs of high-output cardiac failure like polyhydramnios,
A 37-year-old, caucasian, spontaneous primigravida was re-
cardiomegaly, ascites and fetal hydrops were found. The acar-
ferred to our prenatal unit at 25 weeks when twin reversed
diac mass was 176x90x55mm, less than 25% of the healthy twin
arterial perfusion (TRAP) sequence was diagnosed.
estimated weight (2073g). After inconclusive neurosonogram, a
Early in the pregnancy she had performed the first ultrasound
fetal magnetic resonance imaging (MRI) evaluated the natural
at ten weeks and five days: twin pregnancy was identified, with
brain development of the first twin, at 29 and 32 weeks.
a second embryo without cardiac activity. At the 12 week ultra-
Due to suspected fetal anemia, at 32 weeks and 5 days, she un-
sound, one fetus had a crown rump length (CRL) of 61.3mm,
derwent caesarean section. Delivered a typical female live baby,
described with normal morphology, 1.6mm of nuchal translu-
weighting 2100g, Apgar score of 8 / 9 at one and five minutes re-
cency and low risk in the combined screening test. At this time,
spectively; and another female acardiac anceps: with 518g. The
the second fetus was not described, assuming a vanishing twin.
acardiac fetus had a rudimental structure: incompletely formed
The morphological ultrasound at 20 weeks identified a nor-
skeleton, edematous lower limbs, shortened upper limbs, ab-
mal female fetus and a heterogeneous mass of 71x24mm, with
domen with omphalocele and umbilical cord with two vessels,
suggestive bone tissue, considering possible resorption of the
remnants of the chest and head structures (Figures 2 and 3).
second fetus.
At the time she arrived at our prenatal unit (25 weeks), was
described as a monochorionic monoamniotic twin pregnancy,
with one structurally normal fetus and a second twin anatom-
ically abnormal and acardiac. The first twin presented a fetal
estimation weight (EFW) of 846g, in the 50th percentile, nor-
mal fetal Doppler study and normal amniotic fluid index. The
acardiac mass continued to grow (98x53x57mm), and TRAP
sequence was diagnosed by Doppler study, which identified
interstitial blood flow in the acardiac mass (figure 1).
Figure 2: Acardiac fetus delivered.
The healthy baby had mild anaemia (hemoglobin: 13.4 g/dl,

haematocrit 38.9%), without the need for transfusion. She was
admitted to the intensive/intermediate care unit, with a diagno-
sis of prematurity, hyperbilirubinemia and intra-periventricular
haemorrhage grade I and discharged on the 17th day of life,
clinically and neurologically well. Maintains neurodevelopment
surveillance in our hospital centre.
Figure 1: Ultrasound of the acardiac fetus at 28 weeks, measur-
ing 130x98x67mm. DISCUSSION
Advances in first-trimester ultrasound have enabled diagnosis
Weekly ultrasound was performed with standard Doppler of TRAP sequence at earlier gestational age. TRAP should be
study and no signs of cardiac failure. suspected, when a monochorionic pregnancy is diagnosed with
At 29 weeks, the peak systolic velocity (PSV) in the middle one fetus appearing anatomically normal, and the other lacks
cerebral artery (MCA) of the healthy fetus reached 1.54 mul- cardiac structures and activity. The confirmation is made with
tiples of the median (MoM), with no signs of hemodynamic Doppler study: pulsatile flow in the umbilical artery towards
decompensation. Due to suspicion of fetal anaemia, a course the acardiac fetus or interstitial blood flow within the mass and
of antenatal corticosteroids for fetal lung maturation was ad- supported by the observation of continuous growth of the acar-
ministered. Since then ultrasound surveillance started to be diac twin in serial ultrasound examinations.[3] It may simulate
performed twice a week. The PSV in the MCA maintained un- an intrauterine death of one grossly abnormal monochorionic
der 1.5 MoM until 32 weeks and five days, reaching 1.69 MoM. twin or a placental teratoma. In the described case, the first ultra-
The remaining Doppler studies were normal. The pump twin sound identifies the second embryo without cardiac activity, and
Table 1 Timeline of interventions and outcomes.
From Obstetric Ultrasound to Delivery
Out of our unit In our prenatal unit
Once a Twice a
10w 12w 20w 25w 29w 32w 32w
week week
Caesarean
Healthy fetus: delivery
EFW in because of
the 50t h fetal
Monochorionic
percentile and breech
monoamniotic
at breech presentation.
twin, one
Normal fetus: presentation.
structurally
PSV in PSV in the Healthy
One fetus normal fetus
the MCA MCA = female live
with (EFW - 846g)
One female = 1.54 MoM. 1.69 MoM. baby, 2100g
Two normal and a second
fetus, normal No signs of No signs of AS-8/9. Mild
embryos morphology twin Ultrasound Ultrasound
morphology hemodynamic high-output anaemia
one and low (98x53x57mm) + +
and a decompensation. cardiac failure. (hemoglobin:
without risk in anatomically Doppler Doppler
heterogeneous Fetal MRI: Acardiac mass: 13.4 g/dl,
cardiac the abnormal and study study
mass of normal brain 176x90x55mm, haematocrit
activity. combined acardiac.
71x24mm. development less than 25% 38.9%),
screening Doppler study
of of the standard without the
test. identified
the pump twin estimated need for
interstitial
twin. weight transfusion.
blood flow in
(2073g). Discharged on
the acardiac
Fetal MRI: the 17th day of
mass.
normal brain life, clinically
development of and
the pump twin. neurologically
well.
Suspicion of
fetal anaemia,
a course of
Resorption antenatal Suspicion of
Female
Vanishing of corticosteroids fetal anaemia.
TRAP sequence. acardiac anceps,with
twin? the second for fetal Delivery
518g.
fetus? lung decision.
maturation
was
administered.
AS - Apgar score; EFW - fetal estimation weight; MCA - middle cerebral artery; MRI - magnetic resonance imaging;
MoM - multiples of median; PSV - peak systolic velocity; TRAP - twin reversed arterial perfusion; W- Weeks;
lished. Antenatal intervention, delivery and expectant manage-
ment are possible options. It is controversial regarding elective
versus treatment of TRAP. Some support elective intervention
due to improved outcomes after therapy and low sensitivity of
prognosis factors to predict intrauterine fetal death.[7] It seems
that the results are better if therapy is done before 16 weeks.
Others support expectant management with treatment when
sonographic evidence of compromise is present in the pump
twin (polyhydramnios, cardiac dysfunction, abnormal Doppler
and hydrops), avoiding an intervention that itself can cause
miscarriage.[2]
Current, minimally invasive in utero interventions, to occlude
vascular supply to the acardiac twin, can be performed through
two modalities: extra-fetal (intertwine anastomosis or umbili-
cal cord occlusion) and intra-fetal circulation. Occlusion of the
umbilical cord of the acardiac twin can be performed through
a fetoscope by laser or bipolar coagulation. The intra-fetal in-
tervention modality is performed by radiofrequency ablation
(RFA) or laser therapy, through a needle inserted under ultra-
sound guidance. RFA coagulates the abdominal wall of the
acardiac twin, at the base of the umbilical cord, being preferred
at gestational ages greater than 16 weeks. Laser needle is in-
serted towards fetal abdomen and the target fetal vessels, being
Figure 3: X-ray of Acardiac fetus. preferred until 16 weeks.[3,8]
Current small evidence, suggests ultrasound guided intra-
fetal techniques are associated with higher success rates and
a vanishing twin was erroneously assumed in the first trimester.
lower postoperative complications like preterm premature rup-
The pump twin may develop a high-output cardiac failure
ture of membranes and preterm labour. Laser and RFA are
and intrauterine fetal demise. This high cardiac output also
relatively comparable, with neonatal survival rates of 82% and
increases perfusion of the fetal kidneys, resulting in overpro-
85%, respectively.[7,9] The choice of treatment should be based
duction of fetal urine and polyhydramnios, at risk of premature
on operator experience, gestational age and accessibility.[3]
delivery.[2] The perinatal mortality rate for pump twin without
The ideal timing of in utero intervention remains controver-
treatment is high, around 55%, due to heart failure and preterm
sial; procedures have been reported between 12 and 27 weeks
delivery.[4] After in utero therapy, the survival rate of the pump
of pregnancy, is usually performed at the beginning of the sec-
twin increases to 80-90%.
ond trimester (16 weeks). Knowing that the death of the pump
Indicators of poor prognosis have been described. A higher
twin occurs mostly until 16 weeks, preventive intervention at 12
weight of the recipient twin is more likely associated with cardiac
weeks may be more advantageous. Larger studies are necessary
insufficiency and perinatal mortality of the pump twin.[2]
to examine the best timing for preventive intervention with the
When the ratio of the weight of the acardiac twin to the
higher pump twin survival.[2]
weight of the pump twin is greater than 70%, the risk of preterm
delivery is 90%, and of congestive heart failure in the pump, In our case, the patient was diagnosed late in pregnancy
twin is 30%. In comparison, when the ratio is less than 70% the (25 weeks), at the time she was referred to us we adopted an
corresponding risks are 75% and 10%, respectively. Estimation expectant management with weekly ultrasound surveillance,
of acardiac fetus weight can be calculated using the following because of the higher risk of preterm delivery after treatment
formula: Weight (grams) = (1.2 × longest length 2) - (1.7 × most in late gestational age. No polyhydramnios, signs of cardiac
extended lengths).[4] The alternative prognostic factor is the ra- failure or fetal hydrops in the pump twin were identified. The
tio of the abdominal circumference of acardiac twin to standard acardiac twin continued to grow but with an estimated weight
twin when ≥ 1.0 is considered significant.[5] proportion of less than 25%. Rohilla et al. defends conservative
After diagnosis, the aim of management is to save the pump treatment when the acardiac twin is less than 25% the weight of
twin, as mortality is 100% for an acardiac twin. Exclusion of the pump twin and no signs of heart failure. When the weight
genetic abnormalities (determination of the karyotype) and ul- of the acardiac twin exceeds 70% of the pump twin, invasive
trasound malformations of the pump twin, is advised before the intervention is justified.[10]
management decision. It has been reported that the pump twin The timing of delivery depends on clinical evolution and
has a 9% risk of aneuploidy. To improve the outcome for the gestational age. TRAP sequence without poor prognostic criteria
pump twin, at viable gestational age, fetal monitoring should be should deliver at 34-36 weeks of gestation. Caesarean delivery
done with weekly ultrasound and Doppler fetal study. If there is indicated for general obstetrical indications.[3]
is evidence of pre-hydrops signs, the frequency should be twice When anaemia of the pump twin was suspected in this case,
a week. Fetal surveillance with Doppler studies of middle cere- by Doppler study of middle cerebral artery, a course of antenatal
bral artery help in early diagnosis of anaemia in pump twin.[3,6] steroids was administered. The sign of fetal anaemia was our
Fetal neurosonogram should be performed to evaluate brain criteria to interrupt pregnancy after fetal pulmonary matura-
development of the pump twin if cerebral lesions are suspected tion. We delivered a healthy baby at 32 weeks and five days by
a fetal MRI is advised. caesarean section, for breech presentation.
The correct and appropriate management is not yet estab- Our case is an example of why ultrasound should be per-
formed by an experienced sonographer to avoid failure in early 8. Kilby M, Johnson A, Oepkes D. Fetal Therapy: Scientific
diagnosis of TRAP. In this sequence of late diagnosis, the ap- Basis and Critical Appraisal of Clinical Benefits. Cambridge
proach of expectant management with tight surveillance to de- University Press. 2013; 194:197.
cide when to intervene or deliver allows good outcomes.
LEARNING POINTS/TAKE HOME MESSAGES 9. Cabassa P, Fichera A, Prefumo F, et al. The use of radiofre-
quency in the treatment of twin reversed arterial perfusion
• Although twin reversed arterial perfusion (TRAP) sequence sequence: a case series and review of the literature. Eur J
being a rare monochorionic twin pregnancies complica- Obstet Gynecol Reprod Biol 2013; 166:127.
tion, it should be suspected in the first-trimester ultrasound,
when detected monochorionic pregnancies and one em- 10. Rohilla M, Chopra S, Suri V, Aggarwal N, Vermani N.
bryo/fetus has no cardiac activity. Acardiac-acephalus twins: a report of 2 cases and review of
• Follow up in the fetal death of one twin in monochorionic literature. Medscape J Med. 2008; 10:200.
pregnancies must be done with Doppler to look for an acar-
diac twin.
• The appropriate management is not established: elective
treatment and expectant management with treatment or
delivery, are options. Early in utero therapy reduces the
perinatal mortality rate. Intra-fetal intervention with laser
or radio-frequency ablation is the preferred modality.
• Because pump twin may develop a high-output cardiac
failure and intrauterine fetal demise, for continuing preg-
nancies without poor prognostic criteria, it is important to
maintain weekly ultrasound surveillance.
• Criteria for intervention should be: Onset of Cardiac fail-
ure, hydrops of the pump twin or polyhydramnios and
acardiac/pump twin weight ratio > 70%
Competing Interests
The authors declare no conflict of interest.
References
1. Van Gemert MJ, Van den Wijngaard JP, Vandenbussche FP.
Twin reversed arterial perfusion sequence is more common
than generally accepted. Birth Defects Res A Clin Mol Tera-
tol 2015; 103:641.
2. Pepe F, Teodoro MC, Luca C, Privitera F. Conservative man-

agement in a case of uncomplicated TRAP sequence: a
case report and brief literature review. Journal of Prenatal
Medicine 2015; 9(3/4):29-34
3. Mastrobattista JM, Lucas MJ. Diagnosis and management of

twin revered arterial perfusion (TRAP) sequence. UpToDate
Nov 2016.
4. Moore TR, Gale S, Benirschke K. Perinatal outcome of forty-

nine pregnancies complicated by acardiac twinning. Am J
Obstet Gynecol 1990; 163:907.
5. Wong AE, Sepulveda W. Acardiac anomaly: current is-

sues in prenatal assessment and treatment. Prenat Diagn.
2005;25:796-806.
6. Dubey S, Verma M, Goel P, Punia R. Twin Reversed Ar-

terial Perfusion: To Treat or Not? Journal of Clinical and
Diagnostic Research. 2017 Jan, Vol-11(1): QD05-QD07
7. Pagani G, D’Antonio F, Khalil A, Papageorghiou A, Bhide A,

Thilaganathan B. [13] Intrafetal laser treatment for twin re-
versed arterial perfusion sequence: cohort study and meta-
analysis. Ultrasound Obstet Gynecol. 2013;42:6-14
CASE REPORT
SPIDER PROCEDURE FOR SACRAL PRESSURE SORE

RECONSTRUCTION: A CASE REPORT
Sarah Sabur∗,1 , EL Aissaoui Imane∗ , Raboune Mohamed∗ , Bensaida Lamiaa∗∗ , Mazouz Samir∗ , Gharib Noureddine∗∗ and Abassi
Abdellah∗∗
∗ Department of Plastic and Reconstructive Surgery, Mohammed V University Rabat, Morocco., ∗∗ Plastic Surgeon, Mohammed V Hospital, Tangier, Morocco.
ABSTRACT Background: Many forms of management have been described to treat a sacral pressure sore when the
sacral pressure sore is type 4 the surgical treatment becomes necessary. Myocutaneous and fascio-cutaneous flaps can
provide stable coverage of pressure sores. We propose in this article the use of a fascio-cutaneous flap named Spider
flap in the coverage of sacral pressure ulcer in a young ambulant patient. Case report: A 43-years-old ambulant woman
was addressed to our department to assure the management of a sacral pressure sore developed after an extended
immobilisation in intensive care unit, the coverage was assured with modified 5- fascio-cutaneous flap Z-plasty technique
called the spider procedure, the follow-up was nine months, with no sign of recurrence. Conclusion: The spider
procedure has the advantage of tension-free closure, simple dissection with short operative time and no sacrifice of
muscles function which can make this flap a useful solution in sacral pressure sore reconstruction.
KEYWORDS Spider procedure, Sacral defect pressure sore.
A lot of surgical techniques were proposed to treat this condi-

tion, we present our experience in the coverage of sacral defect
occurred after pressure sore, with a spider procedure, the aim of
INTRODUCTION
this article is to propose the use of this simple technique which
Pressure ulcers, also known as decubitus ulcers or bedsores, uses local tissue with tension-free closure in the coverage of
are defined as localised injuries to the skin and underlying tis- medium to large size sacral pressure sore defect. Until now
sue, usually over a bony prominence, as a result of pressure, there is no references in the medical literature for the use of this
or pressure in combination with shear and friction [1]. The flap in pressure sore reconstruction.
sacral pressure sore is the most frequent form of pressure sores,
and the treatment begins with prevention, but when it is classi-
CASE REPORT:
fied as type 3 or 4 the surgical management is required, and it
can be challenging especially for the ambulatory patients who A 43 years old ambulant woman was addressed to our depart-
developed this condition during an extended immobilisation ment to assure the management of a sacral pressure sore devel-
in intensive care unit. For those patients, we should assure a oped after an extended immobilisation in intensive care unit. At
good coverage of the wound with bulky and well vascularized the time of consultation, the patient was on a physical therapy
healthy tissues to assure a good healing and assure a low rate of program to assure an optimal return of function. The clinical
morbidity and disability. examination found a sacral pressure sore stage 4; the X-ray did
not show any signs of osteomyelitis.
We scheduled surgery after a wound preparation with chemi-
DOI: 10.5455/IJMRCR.Spider-procedure-for-sacral-pressure-sore-reconstruction
First Received: June 10, 2017 cal and mechanical debridement, infection control, and improve-
Accepted: July 02, 2017 ment of nutritional parameters. The surgery was performed
Manuscript Associate Editor: George Baytchev (BG) under spinal anaesthesia. We drew first the pattern of the spider
Editor-in Chief: Cvetanka Hristova (BG) procedure [Figure 1-2]. Then we realised a Surgical debridement
1
[Dr Sarah Sabur, Plastic and reconstructive surgery department, Ibn Sina teaching
until viable tissues were encountered.
Hospital, Rabat-Morocco. Email:[email protected] The debridement consisted of a triangular incision of ulcer,
Figure 1: The pattern of spider procedure.
Figure 3: The final aspect after closure.
Figure 2: Technical drawing of spider procedure.

Figure 4: Nine months after surgery.
underlying bursa, surrounding calcifications, the final defect
was an equilateral triangle each side had the size of 9 cm. In
depth, the defect arrives until contact of the bone. anaesthesia or in sedated patients that remain in the supine
The closure was assured then with the spider procedure position. The pressure wound management demands a mul-
which includes the use of a modified 5-flap Z-plasty technique tidisciplinary approach. For the invasive type pressure sores
to obtain maximum tissue relaxation for the tension-free closure (type 3 and 4), in which there is full thickness tissue loss with
of skin defect [Figure 3] [2] [3]. The skin flaps were elevated as or without exposed muscle, bones or tendons, surgery remains
fascio-cutaneous flaps. To prevent any fluid collection under the best option [4], The goals of surgical management are to
the flaps, we put two drains. The patient was immobilised in prevent progressive osteomyelitis, reduce protein loss, improve
ventral position for two weeks after the surgery. An antibiotic quality of life, improve function and hygiene, and reduce re-
treatment depending on bacterial culture realised in the debride- habilitation and wound care cost [1]. To achieve those goals,
ment tissue was prescript; the dressing was changed three times various methods are proposed to surgically treat the sacral pres-
a week. sure sore, depending on the size and the depth of the defect. The
skin grafting is indicated if the defect is small and secondary
The postoperative period was uneventful for the patient. to acute/short-term disability. However, the recurrence rate
Drains were used for five days. Follow-up of 9 months [Fig- with this method is high (70%) [5]. Gluteus maximus flap re-
ure 4], did not show any recurrence. mains the first choice for sacral pressure sore reconstruction
[6].Theoretically The advantages of using myocutaneous flaps
are elimination of the dead space because they are bulky flaps,
DISCUSSION:
providing a well-vascularized and cushioning tissue over the
Sacral pressure sores are commonly seen in neurologically com- pressure bearing area and their vascularity helps fighting infec-
promised patients and in those who have received prolonged tion at the local site [7], but it compromises muscle function and
Sarah Sabur et al./ International Journal of Medical Reviews and Case Reports (2017) 1(2):46-48
should be avoided, especially in ambulatory individual [1]. It is FUNDING
also may be prone to postoperative skin breakdown because of
This research did not receive any specific grant from funding
tension [8], and the deneurotized muscle becomes atrophic and
agencies in the public, commercial, or not-for-profit sectors
loses its dynamic function and the ability to absorb the pressure
[9].
The fasciocutaneous flaps have the advantage of the reduc- COMPETING INTERESTS
tion in donor site morbidity, minimal blood loss, decreased post- The authors declare no conflict of interest.
operative pain, shorter hospital stays, reduced costs, and preser- Written informed consent obtained from the patients for pub-
vation of muscle function. lication of this article and any accompanying images.
Yamamoto et al. [10] reported that the use of the fasciocuta-
neous flap is expected to provide a better long-term result in the References
surgical reconstruction of pressure sores than the myocutaneous
or muscle flap.Thiessen et al. [7] in their study showed that the 1. Diaz S, Li X, Rodríguez L, Salgado CJ.Update in the Surgical
complications and the recurrence rates were not associated with Management of Decubitus Ulcers. Anaplastology. 2013; 2:
the type of the flap. Local fasciocutaneous flaps like Limber flap, 113.
Dufourmentel flap, are simple to realise and have stable circula-
2. Gemici K, Şentürk S. Surgical management of expansive
tion however they are inappropriate to cover a large skin defect
sacrococcygeal pilonidal sinus with the spider procedure.
in the sacral area because of the distortion and displacement of
Eur J Gen Med.2015; 12(3); 203.207.
the adjacent mobile anatomic structures, so the defect closure
is done with high tension [4].Sapountzis and all proposed the 3. Mutaf.M, Temel M, Günal E .The spider procedure: Anew
use of the reading man flap to cover pressure sores with The Zplasty based local flap procedure Ann Plast Surg.2012; 69
advantage of tension-free closure and the minimal additional (5):555-9.
healthy skin excision, but this flap is not able to fill the dead
space [4]. 4. Sapountzis S, JoonPark H, Ye Ong Heo C.The reading man
Several perforator flaps have been proposed to cover sacral flap for pressure sore reconstruction.Indian J Plast Surg 2011
pressure sores, but they are more tedious to dissect [11] [12] and Sep-dec; 44(3):448-452.
are more prone to venous congestion [13]. The free flap coverage
indications include the absence of local flap tissue especially in 5. Bauer J, Phillips.Pressure sores. Plast Reconstr Surg.2008;
the multiple recurrent pressure sore [1]. 121:1- 10.
In our cases we used the spider procedure based on Z–plasty 6. Sarensen JL, Jorgesen B, Gottrup F. Surgical treatment of
concept to reconstruct a sacral pressure severe defect (size 9 cm). pressure ulcers.AM J Surg. 2004; 188:42-51.
In this procedure, first, the existing defect is surgically converted
to a triangle in shape, then using a modified 5-flap Z –plasty 7. Thiessen FE, Andrades P, Blondeel P, Hamdi M, Roche
pattern. The flaps are outlined by transposing the elevated flaps N, Stillaert F, et al.(2011). Flap surgery for pressure sores:
in a Z-plasty manner. A tension free closure is achieved using should the underlying muscle be transferred or not? J Plast
tissue relaxation provided by opposing Z Plasty [3]. Reconstr Aesthet Surg.2011; 64:84-90.
The advantage of this flap comparing to other local fascio-
cutaneous flaps indicated to cover large sacral defect with big 8. Rubayi S, Chandrase Kharb S. Trunk, abdomen and pres-
cavity like bilateral VY advancement gluteal fascio-cutaneous sure sore reconstruction.Plast Reconstr Surg 2011; 128: 201
flap and rotational gluteal fascio- cutaneous flap is that the ten- e-215 e.
sion in the mid-line is minime because of the extra tissue relax-
9. Lee JL, Pyon JK, Lim SY, Mun GH, Bang SL, Oh KS.
ation provided by opposing Z-plasty.
Perforator- based bilobed flaps in patients with a sacral
sore: Application of a schematic design. J Plast Reconstr
CONCLUSION: Aesthet Surg. 2011; 64:790–5
The spider procedure has the advantage of tension-free closure, 10. Yamamoto Y, Tsutsumida A, Murazumi M, Sugihara T.
simple dissection with short operative time and no sacrifice of Long-term outcome of pressure sores treated with flap cov-
muscles function which can make this flap a useful solution in erage. Plast Reconstru Surg. 1997; 100: 1212–7
sacral pressure sore defect reconstruction, Especially in a no-
paralyzed patient whom we want stable results with less donor 11. Koshima I, Moriguchi T, Soeda S, Kawata S, Ohta S, et
site morbidity. al. The gluteal perforator-based flap for repair of sacral
pressure sores. Plast Reconstr Surg.1993; 91: 678-683.
AUTHOR CONTRIBUTIONS 12. Nojima K, Brown SA, Acikel C, Arbique G, Ozturk S, et al.
Sarah Sabur wrote this article. All authors have read and agreed Defining vascular supply and territory of thinned perfora-
to the final version of this manuscript and have equally con- tor flaps: part I. Anterolateral thigh perforator flap. Plast
tributed to its content and the management of the cases. Reconstr Surg .2005; 116: 182-193.
13. Lee BT, Lin SJ, Bar-Meir ED, Borud LJ, Upton J .Pedicled
DISCLOSURE STATEMENT perforator flaps: a new principle in reconstructive surgery.
There was no financial support or relationships between the Plast Reconstr Surg.2010; 125: 201208
authors and any organisation or professional bodies that could
pose any conflict of interests.
REVIEW ARTICLE
META-ANALYSIS OF ANTIEPILEPTIC DRUGS INDUCED

CHOREOATHETOSIS IN PAEDIATRIC PATIENTS
Sriram Shanmugam∗∗ , Lidhu Daniel∗∗ and Jaleel Ahamed∗
∗ Consultant paediatrician, Sri Ramakrishna Hospital, Coimbatore, India., ∗∗ Department of Pharmacy Practice, College of Pharmacy, SRIPMS, Coimbatore,
India.
ABSTRACT
BACKGROUND The safety profile of anti-epileptic drugs (AEDs) is an essential consideration for the regulatory
bodies, owners and prescribing clinicians. Meta-analysis has increasingly been used to identify adverse effects of drugs.
Efficacy studies are often too small to reliably assess risks that become important when a medication is in widespread
use, so meta-analysis, which is a statistically efficient way to pool evidence from similar studies, seems like a natural
approach. The safety profile of drugs is an important consideration, and it affects clinicians’ decisions to prescribe
specific AED(s), as serious adverse effects can lead to chronic complications or even death. Less serious, but significant,
adverse effects can significantly impact quality of life, leading to systematic illness which may increase the overall
cost of treatment. METHODS An electronic search was performed in using Pubmed,Paediatric journals of Neurology,
MEDLINE. RESULTS The literature search identified 30 unduplicated papers. Of these 11 papers were excluded by
reading the abstracts and titles. Another ten papers were excluded from reading their complete text. We selected
nine papers which comprised of case studies and observational studies. CONCLUSION The combination of different
antiepileptic drugs has resulted in drug-induced choreoathetosis. A mainly increased risk was seen with combinations
that have phenytoin and lamotrigine. This could be due to an additive or a synergistic effect on central dopaminergic
pathways.
KEYWORDS: choreoathetosis, antiepileptics, meta-analysis
Morbidity and mortality due to ADR are becoming a challenge

to the healthcare system. Approximately 25% of ADRs have
been reported in inpatients admitted to the hospital. This could
Introduction be attributed to a multitude of factors like polypharmacy, drug
interactions, lack of awareness, easy accessibility of drugs and
Adverse Drug Reaction (ADR) is defined as "Any noxious increased co-morbid disease conditions. The unexpected ADRs
change, which is suspected to be due to the drug, occurs at for the new drugs are yet to be well documented; hence the ADR
doses normally used in man, requires treatment or decrease in monitoring system will be beneficial for the treating physician.
dose or indicates caution in the future use of the same drug”. Some adverse drug reactions have been identified after use by
a large number of people in the phase IV clinical trial, so the
DOI:10.5455/IJMRCR.ANTIEPILEPTIC-DRUGS-INDUCED-CHOREOATHETOSIS documentation of ADR is more emphasised. In India, ADR mon-
First Received: July 31, 2017 itoring system is still primitive due to lack of awareness and
Accepted: August 31, 2017 interest in reporting by the healthcare professionals. A voluntary
reporting system could do active ADR monitoring in a hospital
Editor-in Chief: Cvetanka Hristova (BG)
Reviewers: Ivan Inkov (BG) set-up. Pharmacovigilance plays an essential role in providing
1
Department of Pharmacy Practice,college of Pharmacy,Sri Ramakrishna Hospital information about adverse drug reactions and drug safety in a
Campus,SRIPMS,Coimbatore. hospital. The safety of drug prescribing has become a highly
Email: [email protected]
Sriram Shanmugam et al./ International Journal of Medical Reviews and Case Reports (2017) 1(3):49-52
visible topic in medicine, due in part to research suggesting a computerised literature search of electronic databases like
that there are essential ADRs caused by commonly used medi- PubMed (www.ncbi.nlm.nih.gov./entrez/query.fcgi), ScienceDi-
cations.Paediatric patients constitute a vulnerable group about rect (www.sciencedirect.com), Scirus (www.scirus.com/srsapp ),
rational drug prescribing since many new drugs are released ISI Web of Knowledge (http://www.isiwebofknowledge.com),
onto the market without the benefit of even limited experience in Google Scholar (http://scholar.google.com).
this age group. This deficiency causes paediatricians to prescribe
children drugs in an ’off-label’ manner often, thereby increasing Individual or Aggregated Data
the risk of drug toxicity. Adequate controlled clinical trials in
children lack, mainly because of issues of cost and responsibility, A 3-year-old boy was admitted for treatment of recurrent mi-
and to regulations that frequently act as significant obstacles. nor motor seizures complicating a recent otitis media. At this
Moreover, until recently, the few clinical trials that had been admission, he was still having seizures on a combination of
performed involving children focused on the efficacy of drugs lamotrigine, clonazepam, felbamate and a ketogenic diet. He
and rarely monitored their safety. Chorea (Latin for “dance”) is was started on phenytoin and discharged. Within a few weeks,
a hyperkinetic movement disorder usually due to basal ganglia he developed generalised choreoathetoid movements, which
injury or dysfunction. Movements are brief, irregular, unpre- increased with activity and disappeared during sleep. On dis-
dictable, and flow from one body part to another in a random continuation of phenytoin, there was a significant improvement
fashion. Occasionally, they may be incorporated into a more in his chorea. Chorea as a result of lamotrigine therapy was
purposeful movement to avoid social embarrassment. Chorea noted as a rare side effect in trials. Published reports describing
can occur in isolation, but usually appears in conjunction with new onset choreoathetosis in two young patients treated. With
slow, writhing, distal movements called athetosis (i.e., choreoa- lamotrigine.
thetosis).athetosis is considered to be a hyperkinetic movement A study describes three patients with new-onset choreoa-
disorder characterised by involuntary writhing movements of thetosis that developed while receiving lamotrigine and pheny-
the distal extremities and perioral muscles. Most athetosis is toin in combination therapy. The first patient developed tran-
secondary to lesions in the basal ganglia, whether from cerebral sient chorea when her usual lamotrigine and carbamazepine,
palsy, ischemia, or trauma. In cerebral palsy, athetosis commonly were supplemented with phenytoin, which was loaded intra-
coexists with chorea and is called choreoathetosis. Meta-analysis venously shortly before chorea development. At the time chorea
is already a well-established methodological approach for evalu- developed, her phenytoin levels were in the normal range (16 µ
ating the effectiveness of therapies. However, in contrast to the ml-1 ).The third patient experienced the onset of choreoathetosis
published experience of using meta-analysis to assess drug effi- on a combination of anticonvulsants which included phenytoin
cacy, the use of this method to also quantify the risk of treatments and lamotrigine, in addition to felbamate and topiramate. The
remains limited to date. A recently published meta-analysis on abnormal movements started shortly after adding phenytoin
the incidence of ADRs in hospitalised patients shows that ADRs and significantly improved when phenytoin was discontinued
represent a significant public health issue, making these reac- since this patient used phenytoin in monotherapy before this
tions between the fourth and sixth leading cause of death in the without any abnormal movements. Similar movement disor-
USA, even when the drugs are used in proper doses and for ders developed in two 8-year-old mentally disabled children
approved indications. while they were receiving phenytoin. Seizures after a diphtheria-
Although paediatric pharmacotherapy has recently come to pertussis-tetanus immunisation had improved in each child at
the fore, so far no meta-analytical review has been performed to 1 to 2 months of age. A static encephalopathy ensued, charac-
assess the risk of drugs in the paediatric population. Recently terised by mental retardation, ataxia, spasticity, and a mixed
published drug surveillance studies allow an estimation of the seizure disorder. Intermittent dystonia and choreoathetosis de-
overall incidence of ADRs in different child health care settings. veloped insidiously while serum phenytoin concentrations were
In this study, we systematically conduct a meta-analysis on in the therapeutic range. Sustained dystonia and choreoathetosis
drug-induced choreoathetosis in the paediatric population and developed 2 hours after an oral provocation with phenytoin. The
provide a summary quantitative estimate of their occurrence. baseline abnormalities on the electroencephalogram remained
unchanged during the choreoathetosis. Recognizable metabolic
abnormalities known to be associated with similar movement
Material and Method disorders were excluded.
INCLUSION CRITERIA A study describes three patients with severe myoclonic
• Age(neonates-14years), epilepsy in infancy (SME) who suffer from choreoathetosis due
• Seizure disorder, to the adverse effect of phenytoin. Choreoathetosis appeared
• Combination of ant-epileptic drugs. when these patients were 8, 19, and 21 years old, two days to 6
months after increasing the phenytoin dosage. Choreoathetosis
disappeared when the phenytoin dosage was decreased. The
EXCLUSION CRITERIA
two elder patients experienced the episodic and rather paroxys-
• Comorbid disease, mal onset of long-lasting choreoathetosis, requiring the differen-
• Pseudoseizure, tial diagnosis of degenerative disease. In one of the patients, an
• Age above 14 years. ictal SPECT revealed decreased perfusion in the basal ganglia
contralateral to the unilateral choreoathetosis. Polypharmacy,
LITERATURE RESEARCH including carbamazepine and zonisamide, may have facilitated
A sound meta-analysis is characterised by a thorough and the onset of choreoathetosis. Phenytoin-induced choreoatheto-
disciplined literature search. A clear definition of hypothesis in the patients with SME is a vital differential diagnosis of
ses investigated provides the framework for an investiga- degenerative disorders involving involuntary movements. The
tion.Typically, published papers and abstracts are identified by episodic and paroxysmal nature of this movement disorder can
delay its diagnosis and effective treatment. GABAergic mechanism.
Paroxysmal kinesigenic dyskinesia Paroxysmal kinesigenic The most consistent biochemical lesion in patients with Hunting-
dyskinesia (PKD) described three cases of brief dyskinetic ton chorea appears to be a loss of neurons in the basal ganglia
episodes induced by sudden voluntary movements. Further that synthesise and contain GABA. The significance of this re-
descriptions followed and named the clinical entity paroxysmal mains unknown. A variety of pharmacologic techniques have
kinesigenicchoreoathetosis (PKC). As a subtype of the primary been attempted to increase CNS GABA levels. Valproic acid,
dystonias, it is also called DYT10. PKC is a rare neurologic con- which acts in part via a GABAergic mechanism, has, in a limited
dition, and most cases are sporadic. Only 27% have a family number of uncontrolled cases, ameliorated not only the agitation
history with an autosomal dominant inheritance. The attacks sometimes seen in persons with HD but also the movement prob-
last seconds to minutes and can start between 1 and 40 years of lem. [17] However, no systematic studies have been conducted
age. Up to 100 attacks per day of dystonic posturing, choreoa- on the use of GABAergic agents to treat HD.
thetosis, and ballism can occur. About 42% of patients have
additional afebrile seizures in childhood. Infantile convulsions Pathophysiology
and paroxysmal choreoathetosis- age onset less than one-year
A simple model of basal ganglia function states that dopamin-
Childhood –chromosome 16p12-q12. The rare adverse event
ergic and GABAergic impulses from the substantia nigra and
of antiepileptic therapies includes choreoathetosis.polytherapy
motor cortex, respectively, are funnelled through the pallidum
with antiepileptic drugs induces choreoathetosis.The commonly
into the motor thalamus and motor cortex. These impulses
involved antiepileptic drugs include phenytoin, Phenobarbi-
are modulated in the striatum via two segregated, parallel, di-
tal, valproic acid, carbamazepine and also benzodiazepines.
rect and indirect loops through the medial pallidum and lateral
Non-psychotic drugs that produce choreoathetosis arePheno-
pallidum/subthalamic nucleus. Subthalamic nucleus activity
barbital, carbamazepine, benzodiazepines, valproic acid and
drives the medial pallidum to inhibit cortex-mediated impulses,
phenytoin.A study report three patients who developed choreoa-
thereby inducing parkinsonism. Absent subthalamic nucleus
thetoid movements on anticonvulsants. All those patients were
inhibition enhances motor activity through the motor thalamus,
using phenytoin and lamotrigine in combination as part of their
resulting in abnormal involuntary movements such as dysto-
anticonvulsant regimen when they developed chorea. No pa-
nia, chorea, and tics. VPA-induced chorea seems to require
tient had these movements while on either phenytoin or lamot-
both high or toxic VPA serum concentrations and pre-existing
rigine monotherapy at normal, or toxic, concentrations.Drugs
brain injury. This case report highlights the importance of an
are causing chorea: There are many drugs which may cause
accurate pharmacological history when approaching a patient
choreoathetosis. Drug-induced chorea may be seen during
with subacute onset of movement disorders because of the po-
the acute phase of treatment or may appear after some time.
tential for recovery upon drug discontinuation.Moreover, the
Antiparkinsonian and antiepileptic drugs are most important
VPA, singly or in combination with phenytoin or carbamazepine
causes of chorea. Levodopa-induced chorea is the most common
should be used with caution in those with pre-existing basal
cause of chorea in adults. Treatment includes withdrawal of the
ganglia injury.Pharmacokinetic interaction leading to increased
offending drug. It may take days to months before patiently
free phenytoin level was suggested as a plausible explanation.
is free from symptoms In this patient, it is also reasonable to
Both generalised and focal movements have been described. Du-
conclude that neither lamotrigine nor phenytoin alone produced
ration of dyskinesia has been variable but has often responded
chorea, but the combination did. The third patient experienced
to discontinuation of the anticonvulsants. Chorea is most likely
the onset of choreoathetosis on a combination of anticonvul-
due to the combination therapy. This patient has similar risk
sants which included phenytoin and lamotrigine, in addition
factors to other children described with drug-induced chorea. In
to felbamate and topiramate. The abnormal movements started
combination, the effects of lamotrigine and phenytoin appear to
shortly after adding phenytoin and significantly improved when
be additive or synergistic, resulting in sufficient enhancement
phenytoin was discontinued since this patient used phenytoin
of dopaminergic activity to provoke clinically apparent choreoa-
in monotherapy before this without any unusual movements
thetosis, this is rather a pharmacodynamic interaction between
. Chorea is most likely due to the combination therapy. This
both anticonvulsants.the movement disorder is secondary to
patient has similar risk factors to other children described with
phenytoin and can occur at therapeutic serum concentrations.
drug-induced choreoathetosis. The mechanism by which these
Phenytoin is a central anticholinergic agent and a central stim-
patients developed chorea is unknown. Pharmacokinetic interac-
ulant of serotonin and may induce movement disorders as a
tion between anticonvulsants causing elevated high phenytoin
result of altering these neurotransmitters in the brain. The vari-
levels is unlikely in our patients as lamotrigine is known not to
able expression of these movement disorders may relate to the
elevate phenytoin levels.
nature of the preexisting striatal insult. Combined lamotrig-
ine and phenytoin effects on central dopaminergic pathways
might explain why our patients developed chorea when both
anticonvulsants were present in combination. Neither direct
Discussion enhancement of dopaminergic activity induced by phenytoin
or indirect enhancement by lamotrigine alone was sufficient to
Anticonvulsant-induced choreoathetosis was first reported in provoke abnormal movements. In combination, the effects of
1962 with phenytoin. A literature search reveals about 80 cases. lamotrigine and phenytoin appear to be additive or synergis-
Reported patients are frequently young and have organic brain tic, resulting in sufficient enhancement of dopaminergic activity
abnormalities including mental retardation. More than one-half to provoke clinically apparent choreoathetosis, this is rather a
of the cases have occurred in association with toxic drug levels. pharmacodynamic interaction between both anticonvulsants.
The use of phenytoin with other medications was reported to Removal of one of the medications was sufficient to ameliorate
increase the risk of developing abnormal movements. or eliminate the clinical symptoms. These reported cases sug-
gest that patients treated with a combination of lamotrigine and 7. Phenytoin-induced dystonia and choreoathetosis in two
phenytoin may have an increased risk of developing dyskinetic retarded epileptic children Elias G. Chalhub.M.D. Darryl
movement disorders. C. DeVivo, The official journal of American Academy of
Neurology May 1976 vol. 26, issue no. 5, pg no 494.
Conclusion 8. Adverse motor effects induced by antiepileptic drugs G.
We conclude that drug-induced choreoathetosis may be seen Zaccara et al., US National Library of MedicineNational
during the acute phase of treatment or may appear after some Institutes of Health, September 2004; vol6: issue 3, pg no
time Chorea is a rare side effect of anticonvulsants.The combina- 153-68.
tion of different antiepileptic drugs has resulted in drug-induced
9. Chorea Sanjay Pandey Journal of the association of physi-
choreoathetosis.Polytherapy with certain anticonvulsants may
cians of India, July 2013, volume 61, pg no 35-47.
predispose patients to drug-induced choreoathetosis.It is essen-
tial for clinicians to evaluate both AEDs’ effectiveness and safety 10. Movement Disorders II: Chorea, Dystonia, Myoclonus, and
on an individual basis before the selection of the appropriate Tremor Jonathan W. Mink, Samuel H. Zinner, US national
monotherapy or adjunctive AED therapy. library of medicine-national institute of health, Vol.31, issue
no.7 July 2010, pg no 287-94.
Acknowledgement
11. Chorea and related disorders R Bhidayasiri, D DTruong,
We are very much delighted to connote our vehement in- post graduate medical journal 2004 Sep;80(947):527-34.
debtedness to Thiru. R.Vijayakumhar, our Managing Trustee,
Dr.P.Sukumaran, Dean, Sri Ramakrishna Hospital & Dr T.K. 12. Study of Chorea in Children Debabrata Ghosh MD; Stephen
Ravi, Principal, College of Pharmacy, Sri Ramakrishna Institute Sreshta; Kohilavani Velayudam. International Parkinson
of Paramedical Sciences and all the teaching staff of the Depart- and Movement Disorder Society,8 July 2016, Volume 4, Issue
ment of Pharmacy Practice, College of Pharmacy, SRIPMS for 2, Pages 231–236.
the support, guidance and encouragement throughout the study.
13. Lotze T, Jankovic J. Paroxysmal kinesigenicdyskinesias.
Seminars in pediatric neurology 2003; 10: 68-79.
14. Drug-induced movement disorders Denes Zadori, et al.
Competing Interests
, US National library of medicine –national institute of
Written informed consent was obtained from the patient for health,pubmed.gov, 2015 Jun, volume 6, pg no 877-890.
publication of this case report and any accompanying images.
There were no financial support or relationships between the 15. Valproate-Induced Generalized Choreoathetosis Alfonso
authors and any organization or professional bodies that could Giordano, Marianna Amboni, and Alessandro Tessitore,
pose any conflict of interests. The International Parkinson and Movement Disorder Soci-
ety,28 JUL 2014, Volume-1-Issue-3.
References 16. Phenytoin-induced chorea in a pediatric patient: An in-
1. Choreoathetosis as a side effect of gabapentin therapy teraction between phenytoin, phenobarbital and clobazam
in severely neurologically impaired patients.Chudnow Manish Barvaliya et al., Indian Journal of Pharmacology,
RS, Dewey RB Jr, Lawson CR, Archives of Neurol- Vol. 43, No. 6, November-December, 2011, pp. 731-732.
ogy,1997Jul;volume54, issue 7pg.no:910-2.
17. Evidence for Efficacy of Combination of Antiepileptic Drugs
2. Movement disorders in patients taking anticonvulsants C in Treatment of Epilepsy Ehsan M. Sarhan, Matthew C.
Zadikoff et al.Journal of Neurology, Neurosurgery and Psy- Walker, Caroline Selai, journal of neurology research, Vol-
chiatry,2007 Feb; 78(2): 147–151. ume 5, Number 6, December 2015, pages 267-276.
3. Reversible valproate-induced choreiform movements Dilek

Ince Gunal1, MelihaGuleryuz, CananAykutBingol, Journal
of Neurology, April 2002, Pages 205–206, VOLUME 11, IS-
SUE 3.
4. Valproate-Induced Chorea and Encephalopathy in Atypi-

cal Nonketotic Hyperglycinemia Peter F. Morrison, Raman
Sankar, W. Donald Shields, Paediatric Journal of Neurology,
Volume 35, Issue 5, November 2006, Pages 356–358.
5. Anticonvulsants-induced chorea: a role for pharmacody-

namic drug interaction? MegdadZaatrehet al., US National
Library of MedicineNational Institutes of Health, Volume
10, Issue 8, December 2001, Pages 596–599.
6. Transient chorea induced by phenytoin F. Filloux, J.A.

Thompson, Journal of Paediatrics, Volume 110, Issue 4,
April 1987, Pages 639-641.
CASE REPORT
CASE REPORT OF DENGUE ENCEPHALOPATHY IN

PREGNANCY MIMICKING ATYPICAL ECLAMPSIA – AN
INSIGHT OF NEUROLOGICAL MANIFESTATIONS OF
DENGUE IN PREGNANCY
Sasirekha Rengaraj∗,1 and Suthrasika Thiyagalingam∗
∗ Department of Obstetrics & Gynaecology, JIPMER, Puducherry, India.
ABSTRACT
Dengue is a global public health problem. Dengue in pregnancy carries a high maternal and perinatal morbidity and even
mortality especially when the diagnosis is delayed. Often dengue with hemorrhagic shock syndrome is misinterpreted
as HELLP syndrome and unnecessary obstetric interventions have to be avoided in such situations. There are case
reports and guidelines on dengue hemorrhagic syndrome in pregnancy. However, rare atypical forms like neurological
manifestations of dengue in pregnancy are rarely studied. Neurological events of dengue are increasingly observed
nowadays. We are presenting a case report of atypical dengue (dengue encephalopathy) in pregnancy with a review of
the literature. It was initially thought as meningitis then atypical eclampsia and HELLP syndrome. Whenever dengue
infection presents in atypical form, strong clinical suspicion and early intervention improve the outcome.
KEYWORDS Dengue, dengue encecphaolopathy, atypical HELLP
rhage, preterm labour and stillbirths. Apart from that, Dengue

infection as such in pregnancy can be a significant diagnostic
challenge. This is very true in atypical presentations like dengue
Introduction
encephalitis /encephalopathy. Pregnancy-specific conditions
Dengue fever is a significant global health problem worldwide, like preeclampsia, HELLP syndrome can mimic such atypical
and 2/5th of world’s population is at risk[1]. Dengue fever, presentations which can delay or misguide the clinician which
an Aedes aegypti mosquito-borne infection has an expanded often leads to poor maternal and perinatal outcome.
clinical spectrum of asymptomatic infection to fatal dengue hem- However, atypical forms such as neurological manifestations
orrhagic fever and shock syndrome. are not uncommon even in pregnancy. It was first reported
The impact of dengue on pregnancy needs to be better un- in 1976, and worldwide literature search shows the various
derstood primarily regarding the maternal and perinatal out- incidence of dengue encephalopathy from 0.5% to 6.2% with
come. The complications of dengue in pregnancy have been a mortality rate up to 40% [2]. Neurological manifestations of
scarcely studied. There are increased risks of maternal haemor- dengue are poorly studied especially in pregnancy.

DOI: 10.5455/IJMRCR.dengue-encephalopathy-in-pregnancy-mimicking-atypical- Case Report
eclampsia
First Received: May 20, 2017 Twenty-six years Mrs A, G2P1L1 monoamniotic twin pregnancy
Accepted: August 31, 2017 presented at 36+2 weeks to the emergency department with
Manuscript Associate Editor: George Baytchev (BG) history of leaking P/V.
Editor-in Chief: Cvetanka Hristova (BG) It was spontaneous conception; she was booked at a private
1
Associate Professor, Department of Obstetrics & Gynaecology, JIPMER,
hospital. Her antenatal period was uneventful. There was no
Puducherry,605006. [email protected] history of preeclampsia or anaemia. She was taken up for emer-
Sasirekha Rengaraj et al./ International Journal of Medical Reviews and Case Reports (2017) 1(3):53-55
gency LSCS given monoamniotic twin. Her previous Obstetric
history was routine. There was no significant past and family
history.
Emergency LSCS was done under SA. She one recording
of 150/100 intraoperative, settled on its own. There were no
other intraoperative complications. Both female babies weighed
2.2 & 2.1kg, and they were by M/S. She did not receive any
antihypertensive drugs.
Within 6 hours of surgery, her dressing was soaked which
required exploration of subcutaneous bleed under anaesthesia.
There was a brisk bleeder from the subcutaneous plane, and
the same was ligated. She had one high spiking fever on a
postoperative day 1. No clinical signs of puerperal sepsis. Her
breasts were soft. Fundamental investigations were sent, and
urine culture was sterile. Two days later she developed diffuse
abdominal pain associated with vomiting which was non-bilious
and non-projectile. There was no diarrhoea. Her Vitals were
stable except for minimal dehydration.
The abdominal examination did not reveal any abnormal-
ity. It was soft, and there was minimal gaseous distension. On
a postoperative day four, she started having signs of cerebral
irritation with up drawing plantar. Her pupils were equal and re-
active. Initial neuroimaging (CT) did not reveal any abnormality.
She was transferred to ICU and received supportive measures.
Her Bp was 110/60mmHg. The fluid imbalance was corrected.
With the correction of intra volume depletion, she developed
altered mental status with signs of decerebrate rigidity. Hence Figure 1: Timeline for case report.
further imaging was undertaken to rule out any cerebrovascular
accidents but showed only diffuse cerebral oedema; received
brain cells by IHC in CSF of a patient with encephalitis. It could
fluid therapy under close monitoring. Platelets and packed cell
be due to direct viral infection of CNS or autoimmune reac-
transfusion were given. Cerebral symptoms showed improve-
tion following dengue infection or due to metabolic/underlying
ment with anti-cerebral oedema measures. Two days later her
hemorrhagic complication.
dengue serology report came and showed a positive result for
Atypical manifestations of dengue fever are associated with
NSN1 antigen and IgM Ab. Recovered well, had secondary su-
more severe grades of disease. Various case series have shown
turing for wound infection. The patient was discharged from
the importance of early and prompt proactive management ob-
the hospital on Post op day 14 without any sequelae.
served nowadays and carried a high mortality. Few case series
have been reported which shows different presentations like en-
Discussion cephalopathy, encephalitis, neuropathy, and optic neuritis and
Dengue fever, which is caused by four similar but antigenically Guillain Barre syndrome [5].
different serotypes of flavivirus has 50% mortality rate, espe- Even though rare, they have been increasingly reported in
cially if untreated [3]. There are 50-100 million new cases added dengue epidemic regions. Classically it can occur in patients
every year. Even though India is an endemic area for recent with few or no signs of previous dengue infection like this pa-
dengue statistics have shown a case fatality ratio of <0.5% es- tient. Moreover, it can occur within 2-30 days after the onset
pecially after the implication of national guidelines on clinical of fever. There can be myelitis, myositis, radiculoneuritis and
management of dengue fever. Even in India some areas has a neuropathy. A headache, seizures and altered consciousness
case fatality rates between 3-5%. are the usual manifestations of dengue encephalitis [6]. How-
Dengue fever in pregnancy can be confused with HELLP ever, these typical symptoms are seen in only less than 50% of
syndrome, SLE, thrombotic thrombocytopenic purpura (TTP), patients with dengue encephalitis. Whenever patient presents
sepsis and DIC especially when it presents in atypical form [4]. with encephalitis, it should be one of the differential diagnosis
Following viral infection, there is lifelong protection against in patients from dengue-endemic areas
similar subtype. However, subsequent infection with differ- Diagnostic criteria for dengue encephalitis:
ent subtype can be dangerous because it increases the chance of • Fever
dengue hemorrhagic fever. The heterotypic antibodies form com- • Acute signs of cerebral involvement
plexes, which attacks mononuclear phagocytes with enhanced • Presence of anti-dengue IgM/ dengue genomic material
efficiency. This antibody-dependent enhancement results in en- in serum/CSF MRI/CT findings may vary from cerebral
hanced viral load replication which results in the severe form of oedema, haemorrhages to focal abnormalities in basal gan-
dengue fever like dengue hemorrhagic fever and dengue shock glia, hippocampus and thalamus.
syndrome. Similarly, it may also contribute to the development
of atypical forms of dengue called dengue encephalopathy. It takes time to make an exact diagnosis of atypical manifesta-
These neurological manifestations are associated explicitly tions of dengue fever like in our patient. It was initially thought
with DENV-2 & 3. These serotypes were found in encephali- of postoperative ileus, warranted surgery opinion later meningi-
tis, meningitis and myelitis. Even Den-4 was also detected in tis (complication following spinal anaesthesia) and then HELLP
syndrome. The most crucial presentation was encephalopathy 3. Rajagopala L, Satharasinghe RL, Karunarathna M BMC Res
like features which include a headache, seizure, altered senso- Notes. 2017; 10: 79. Published online 2017 Feb 2. doi:
rium and behavioural disturbances. Various pathogenesis has 10.1186/s13104-017-2391.
been described. It could be due to cerebral oedema or cerebral
haemorrhage. Even microcapillary haemorrhage and cerebral 4. Lavanya R, Ravindra L. Satharasinghe, and Madhava
hypoxia play a role. Electrolyte disturbances like hyponatremia, Karunarathna A rare case of dengue encephalopathy com-
hepatic failure or release of toxic products might be one of the plicating a term pregnancy J Community Med Health Educ
reasons for neurological manifestations. DOI: 10.4172/2161-0711.C1.019.
There is no convincing evidence for a demonstration of direct 5. Carod-Artal FJ, Wichmann O, Farrar J, Gascon J. Neurologi-
invasion to the central nervous system (CNS) however recent cal complications of dengue virus infection. Lancet Neurol.
evidence shows dengue virus is capable of direct invasion onto 2013;12:906–19.
CNS. The entry of the virus into brain seems to occur through
infiltrates of infected macrophages7. Few animal studies have 6. Solomon T, Dung NM, Vaughn DW, et al. Neu-
shown the cytokines which are released in dengue plays a role rological manifestations of dengue infection. Lancet.
in creating a breach in BBB which result in CNS invasion. The 2000;355(9209):1053–9.
neurological manifestations can vary from altered consciousness
(most common), seizure, mental confusion; limb spasticity to 7. Puccioni-Sohler M, Rosadas C, Cabral-Castro MJ. Neurolog-
focal neurological deficits (least familiar).The IgM antibody was ical complications in dengue infection: a review for clinical
even isolated from CSF. Usually, it is self-resolving. Supportive practice. Arq Neuropsiquiatr. 2013;71 (9B):667–71.
treatment is important. There is no specific treatment as such.
Fluid support and intense monitoring play a significant role in
the outcome. Involvement of other systems such as hepatic fail-
ure, respiratory distress, hyperkalemia, acute kidney injury and
metabolic acidosis may occur if untreated which increases the
morbidity rate. Secondary infections like ventilator-associated
pneumonia (VAP) may complicate the situation.
Even though misdiagnosis happened in our case, early in-
tensive monitoring and fluid therapy along with ante cerebral
oedema measures improved the outcome.
Conclusion
Dengue in pregnancy is a real threat to both obstetrician and
patient. The classical presentation of dengue is unlikely to be
missed. However, it is a significant diagnostic challenge when
it presents in atypical form, especially during pregnancy. It can
be easily misinterpreted with many other clinical conditions of
pregnancy. Dengue should be the differential diagnosis even
a pregnant women presents with fever and altered sensorium
especially in endemic areas like India. A high index of clinical
suspicion and prompt intervention in such situations improves
the maternal and fetal outcome. Further studies are needed to
evaluate dengue neurotropism in pregnancy.
Disclosure Statement
There were no financial support or relationships between the
authors and any organization or professional bodies that could
pose any conflict of interests.
Competing Interests
Written informed consent obtained from the patient for publica-
tion of this case report and any accompanying images.
References
1. Gupta E, Ballani N. Current perspectives on the spread of
dengue in India. Infect Drug Resist 2014 ;7:337-42.
2. WHO. Dengue and Dengue haemorrhagic fever. Factsheet

No. 117. Geneva: World health organization. 2008.

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Table of Cont ent s - Int J Med Rev Case Rep.

Year: 2 0 1 7 , Volume: 1 , Issue: 1 -3 , pp: 1 -5 5

ISSN: 2 5 3 4 -9 8 2 1 – Edit or-in-Chief: Dr. Ivan Inkov

Review Art icle

3 . Pancreas Divisum Causing Recurrent Pancreat it is: A Case Report

Review Art icle

7 . Test icular Descend, How and Why: A Review Art icle

9 . Surgical management of severe t rauma involving group of muscles around t he brisket of

1 1 . Misdiagnosis of an Acardiac Twin in t he First Trimest er, A Case Report .

Review Art icle

1 4 . Case report of Dengue encephalopat hy in pregnancy mimicking at ypical eclampsia – An

LOS HOMBRES Y LOS ACCIDENTES DE TRÁNSITO:

Los accidentes de tránsito son un grave y complejo problema de

23. Andrade SSCA, Mello Jorge MHP. Estimate of physical

Herpes virus type 6 and 8[3], Human Immunodeficiency virus

Figure 3:Axillary lymph node blade, representing necrotizing

16. Kucukardali Y, Solmazgul E, Kunter E, et al. Kikuchi-

22. Smith KG, Becker GJ, Busmanis I. Recurrent Kikuchi’s dis-

31. Kishimoto K, Tate G, Kitamura T, et al. Cytologic features

36. Rezai K, Kuchipudi S, Chundi V, et al. Kikuchi-Fujimoto

PANCREAS DIVISUM CAUSING RECURRENT

Figure 2: ERCP showing presence of minor papilla about 5

Post procedure, the patient had a smooth recovery with sig-

This report emphasizes the need of early suspicion of disease, in

VULVAR NON-FILARIAL ELEPHANTIASIS

Copyright © 2017 by the Bulgarian Association of Young Surgeons

Figure 2: Posterior view of the vulva.

PITUITARY PROLACTIN MICROADENOMA WITH,

Pituitary adenomas are benign tumours developed at the ex-

Evolution and prognosis

SURGICAL MANAGEMENT OF TRAUMATIC

letter from the Institutional Ethical Review Committee was ac-

Discusion: 2. Lemaitre J, Koriche C, Massard G, Wihlm J. Manubrioster-

7. Chou SS, Sena MJ, Wong MS. Use of SternaLock plating

8. Abdul-Rahman MR, Seong NK, Hee TG, Aljada ME, Reda

9. Al-Qudah A. Operative treatment of sternal fractures. Asian

10. Severson EP, Thompson CA, Resig SG, Swiontkowski MF.

TESTICULAR DESCEND, HOW AND WHY: A REVIEW

The urinary system consists of Kidney, Ureter, Urinary blad-

14. Nightingale SS, Al Shareef YR, Hutson JM. Mythical ‘Tails

15. Attah AA, Hutson JM. The role of intra-abdominal pressure

16. Ramareddy RS, Alladi A, Siddappa OS. Ectopic testis in

17. Fentener van Vlissingen JM, Koch CA, Delpech B, Wens-

EFFICACY OF INTRAVENOUS IMMUNOGLOBULIN AND

phototherapy (PT) have traditionally been used to treat jaun-

7. Nigerian consensus statement on newborn jaundice. Man-

8. Urbaniak SJ. ADCC (K Cell) lysis of human erythrocytes

9. Senterre T, Minon JM, Rigo J. Neonatal ABO incompati-

10. Ahlfors CE. Criteria for exchange transfusion in jaundiced

11. Greco C, Arnolda G, Boo NY, Iskander IF, Okolo AA,

12. Cortey A, Elzaabi M, Waegemans T, Roch B, Aujard Y. [Ef-

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14. R Gottstein, RWI Cooke. Systematic review of intravenous

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SURGICAL MANAGEMENT OF SEVERE TRAUMA

remain a challenging clinical problem, with early and late com-

Figure 1: Wound presented to the Veterinary Teaching Hospital.

2. Leaper PJ, Harding KG (1998). Wounds, Biology and man-