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Green teeth resulting from neonatal hyperbilirubinemia: Report of a case

Article  in  Pediatria Polska · March 2015

DOI: 10.1016/j.pepo.2014.12.004

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Case report/Kazuistyka

Green teeth resulting from neonatal

hyperbilirubinemia: Report of a case

Norma Suely Falcão de Oliveira Melo 1, Regina Paula Guimarães

Vieira Cavalcante da Silva 2, Antonio Adilson Soares de Lima 1,*
1
Department of Stomatology, School of Dentistry, Universidade Federal do Paraná – UFPR, Curitiba-PR, Brazil
2
Department of Pediatrics, School of Medicine, Universidade Federal do Paraná – UFPR, Curitiba-PR, Brazil

article info abstract

Article history: In general, cosmetic changes or painful disorders often lead parents to seek dental care
Received: 05.11.2014 for their children. Several systemic disorders in pediatric patients can produce dental
Accepted: 11.12.2014 alterations. One of the manifestations of these disorders is the elevated serum level of
Available online: 23.12.2014 bilirubin (hyperbilirubinemia), a product of hemoglobin degradation, which is deposited
in mineralized and soft tissues. The aim of this article is to report a case of green teeth
Keywords: affecting all deciduous teeth in a 3-year-old girl. The patient was taken to see the dentist
 Hyperbilirubinemia by their parents due to the presence of green pigmentation in the teeth. During birth, the
 Dental enamel patient suffered a traumatic injury that resulted in hyperbilirubinemia. This kind of ena-
 Deciduous teeth mel pigmentation is permanent and occurred during the period of dental development.
 Tooth discoloration © 2015 Published by Elsevier Sp. z o.o. on behalf of Polish Pediatric Society.

substances and bacteria, which represent deposits, adhered

to the enamel [2].
Introduction The intrinsic dental pigmentation is associated with
a chemical active change in the tooth structure and it
Changes in the normal color of deciduous and permanent cannot be removed without changing the structure of the
teeth may occur due to factors intrinsic or extrinsic. The tooth. It can be caused by erythropoietic porphyria, fetal
intrinsic stains are changes in the color of the tooth related erythroblastosis, amelogenesis imperfecta, dental fluorosis,
to systemic factors such as: genetic factors, birth defects, hyperbilirubinemia, decomposition of red blood cells and
metabolic defects, prenatal care, infectious diseases, neurolo- medications (tetracycline) [3].
gical disorders, endocrine, kidney, liver, nutritional deficien- Bilirubin, a breakdown product of heme, is normally
cies and poisoning that usually occurred during odontogene- excreted in the bile or further catabolized before excretion
sis [1]. Moreover, the formation of the extrinsic stains on in the urine. Approximately 80% of bilirubin is derived
teeth occurs primarily from the remains of food, medicinal from free hemoglobin resulting from hemocaterese. In this

* Corresponding author at: School of Dentistry – Department of Stomatology, Universidade Federal do Paraná – UFPR, Rua Prefeito
Lothário Meissner 632, Jardim Botånico, 80170-210 Curitiba-PR, Brazil. Tel.: +55 41 33604050; fax: +55 41 33604134.
E-mail address: [email protected] (A.A.S. de Lima).
http://dx.doi.org/10.1016/j.pepo.2014.12.004
0031-3939/© 2015 Published by Elsevier Sp. z o.o. on behalf of Polish Pediatric Society.
156 pediatria polska 90 (2015) 155–160

physiological process, senescent red cells with approxima-

tely 120 days are destroyed by macrophages in the spleen,
liver and bone marrow. Hyperbilirubinemia is a clinic condi-
tion characterized by an abnormal increase of bilirubin in
the blood, which may result in jaundice. The jaundice is the
clinical sign caused by the elevation of serum bilirubin
levels and its deposition in the skin, sclera, mucous mem-
branes and some organs (liver and kidneys). The deposition
of this pigment in these tissues and organs produces a color
ranging from yellow to black, passing through various
shades of green [4].
All healthy neonates develop unconjugated hyperbilirubi-
nemia during the first week of life, but 50% of these patients
have jaundice, benign and transient in character [5]. Jaun-
dice occurs when the liver fails to eliminate bilirubin Fig. 2 – Lateral incisor, canine, and first primary molar with
produced by the body [6]. Severe neonatal hyperbilirubine- enamel hypoplasia and green pigmentation
mia carries a potential for permanent neurological impair-
ment [7]. Furthermore, hyperbilirubinemia is associated with
the development of changes in color of the teeth, known as
green teeth [8]. These teeth do not show changes in their
morphology. However, the green coloration considerably Furthermore, the tip of the cusp canine was also pigmented.
affects the esthetics of the affected tooth. The appearance The clinical crown of all teeth showed hypoplasia areas
of the dentition is of concern to a large number of people between the middle and incisal thirds (Fig. 2). The teeth
seeking dental treatment and the color of the teeth is of of the lower arch also showed a green coloration on the
particular cosmetic importance [9]. The purpose of this crown (Fig. 3). At this time, the child has not had the eruption
report is to present a case of green teeth of the primary of the primary second molars. The upper and lower canines
dentition in a 3-year-old Brazilian girl. had green pigmentation in the incisal half of the crown. The
pigmentation of the first primary molars reached ¾ of the
crown. According to the presence of this green pigmentation
Report of a case in the crown of teeth, a diagnosis of green teeth associated
with a systemic disorder has been established.
A patient of 2 years was brought for consultation by the The past medical history revealed that the child's mother
mother due to the presence of green teeth in the mouth. The was 28 years old and that she had consulted 4 times during
intraoral examination showed the presence of anterior pri- the prenatal period. During pregnancy, the mother had
mary teeth with a green aspect. All deciduous teeth were a urinary tract infection that was treated with cephalexin.
affected. No other changes involving the oral soft tissues The mother was admitted in premature labor and prema-
were observed. According to the story told by the mother, the ture rupture occurred an hour earlier. The child was born by
teeth were erupting in the mouth with a discoloration. The cesarean section with difficult extraction. The indication for
pigment involved about 5/6 of the crown of the incisor teeth this type of delivery was due to dystocia progression.
(Fig. 1). At 3 years of age, the child presented with the first At birth, the child had been in gestation for 31 weeks
primary molars with half of the crown pigmented (Fig. 2). and was weighing 1660 g. The cause of preterm birth was

Fig. 1 – Upper incisors showing discoloration and areas of

enamel hypoplasia Fig. 3 – Clinical appearance of green teeth in the lower arch
 pediatria polska 90 (2015) 155–160 157

premature rupture of membranes. After birth, the child

proved to be hypotonic, cyanotic and did not cry. A cycle of Discussion
positive-pressure ventilation and cardiac massage for reco-
very were made. In addition, she was intubated in the Bilirubin is produced mainly by degradation of the hemoglo-
delivery room. The newborn had neonatal asphyxia and bin of red blood cells, the final product of the catabolism of
slow infusion. In addition, the child had tocotraumatism, heme [6]. The clinical jaundice in newborns occurs when
anemia, bloating, and early sepsis. The child was treated bilirubin levels in blood plasma reaches values between
with ampicillin, gentamicin, dopamine, furosemide, pipera- 5 and 7 mg/dL. Severe neonatal hyperbilirubinemia, when
cillin, tazobactam, dopamine, dobutamine, fentanyl and unmonitored or untreated, can progress to acute bilirubin
metronidazole. A diagnosis of large lesion in subcapsular encephalopathy [10].
hepatic lobe was established and it was classified as grade II Neonatal jaundice is one of the most common problems
splenic laceration that was treated expectantly. Infection of for newborn infants during the first weeks of life, affecting
the child was treated for 52 days and the child remained approximately 60–70% of term babies and almost all prema-
intubated (intubation) for 12 days due to apnea and surfac- ture babies [11]. According to Shrestha et al. [13], the risk
tant administration. factors associated with preterm birth are: inadequate ante-
During her stay in the Intensive Care Unit, volume natal checkup (52%), maternal age <20 years (34.7%), ante-
globular and bilirubin levels were investigated (Table I). partum hemorrhage (23.4%) and pregnancy induced hyper-
Jaundice was diagnosed on the second day of life by tension (13.1%). Morbidities more commonly observed in
yellowish discoloration of the skin during routine physical preterm infants are: clinical sepsis (66.7%), hyperbilirubine-
examination. The pathological jaundice was treated with mia (58.8%), birth asphyxia (26.8%) and hyaline membrane
phototherapy. The child had the same blood type (A disease (23.5%) [12]. The patient in this case report showed
positive) as the mother and received packed red blood cells. the major health problems related to prematurity, especially
The child presented with hyperbilirubinemia for about 2 sepsis, neonatal jaundice, and apnea. In this case, bilirubin
months. was considered pathologic due to lesion in the subcapsular
The mother was instructed on the importance of diet lobe resulting from tocotrauma. The levels of total bilirubin
and oral hygiene was reinforced. As the green pigmentation reached 30.58 mg/dL in the 18th day of life.
affected the deciduous dentition and there was a possibility According to Watanabe et al. [12], total bilirubin levels
it could also occur in the permanent dentition, the child's between 1.2 and 1.8 mg/dL are already capable of inducing
mother was advised to return for follow-up dental. No pigmentation tooth. These authors have identified levels of
cosmetic treatment has been established in affected tooth. bilirubin in deciduous teeth obtained from 2 patients with
As the child was premature, we observed a delayed eruption a history of severe liver dysfunction and hyperbilirubinemia.
of deciduous teeth and consequently also in the permanent The teeth were histologically analyzed and bilirubin was
dentition. extracted and quantified by spectrophotometry. Histological

Table I – Values of globular volume and levels of bilirubin of the patient

Date Globular volume Direct bilirubin Indirect bilirubin Total bilirubin
June 18 16% N N N
June 19 32.9% N N N
June 20 33.6% 3.27 mg/dL 9.29 mg/dL 12.56 mg/dL
June 21 37.5% 3.27 mg/dL 9.29 mg/dL 12.56 mg/dL
June 22 35.2% Absence of bilirubin in the
partial examination of urine
June 23 38.3% 11.97 mg/dL 8.11 mg/dL 20.08 mg/dL
June 25 32.2% Jaundice++/4+
June 28 30.1% 11.55 mg/dL 23.25 mg/dL 11.70 mg/dL
June 30 41.0% N N N
July 1 37.4% N N N
July 2 36.0% N N N
July 3 34.4% N N N
July 4 34.0% 20.25 mg/dL 8.08 mg/dL 28.33 mg/dL
July 5 46.0% 21.12 mg/dL 9.46 mg/dL 30.58 mg/dL
July 9 43.9% N N N
July 11 34.9% N N N
July 13 N 10.6 mg/dL 4.02 mg/dL 14.68 mg/dL
July 17 33.2% N N N
July 20 30.9% N N N
July 21 N 9.14 mg/dL 1.67 mg/dL 10.81 mg/dL
July 27 26.0% 10.46 mg/dL 5.35 mg/dL 15.18 mg/dL

N = sample not collected.

158 pediatria polska 90 (2015) 155–160

analysis revealed a green line parallel to the incremental bilirubin showed clinically the time of injury to the tooth
lines of dentin. These authors analyzed the deciduous germ by the incorporation of these substances to the
canine of a child with a history of biliary atresia and enamel [12, 31].
congenital hyperbilirubinemia at birth that showed that the In this case, the patient report presented different levels
tooth were colored brown in the middle of the crown to half of pigmentation of deciduous teeth. The crowns of the
of the root. The other case was a boy of 7 years of age with central and lateral incisors were more affected as half of
a history of hemolytic anemia and hyperbilirubinemia at 18 their crowns were affected. The pigmentation was observed
months of life showed total bilirubin levels 20–60 mg/dL. involving only half of the crown of the molars. Moreover,
The upper central incisors and lateral incisors had a change canine teeth were the least affected. As calcification of the
of color of green. Furthermore, this color change reached permanent dentition has its beginning from birth, the
half incisal of the dental crown. These findings were similar possibility of dental pigmentation affecting the permanent
to those observed in primary teeth of children of this report. teeth, especially the first molar, has not been ruled out.
The normal color of teeth is determined by the shades of High serum bilirubin levels favor the deposition of the
blue, green and pink of the enamel and is reinforced by product of hemoglobin degradation in many tissues, inclu-
shades of brown or yellow dentin below. Metabolic diseases ding soft tissue and mineralized tissues. When hyperbiliru-
and systemic factors can affect the development of teeth binemia occurs during dental development, the teeth can
and cause tooth discoloration, such as: alkaptonuria, eryth- develop a green color due to the bilirubin deposition. This
ropoietic porphyria congenital, amelogenesis imperfecta, intrinsic pigment keeps teeth permanently pigmented. The
dentinogenesis imperfecta, tetracycline stain, and congenital blood vessels of the dental follicle are the source of
hyperbilirubinemia. However, the teeth do not show morp- nutrients for the ameloblasts to synthesize the tooth
hological changes [9]. The patient in this case report had enamel [32]. Thus, the pigmentation of the tooth is most
green teeth and areas of enamel hypoplasia. Despite these likely the result of the pigment passing through the
changes, the teeth remained preserved in its form. bloodstream to ameloblasts and, then, to the tooth enamel.
The areas of enamel hypoplasia observed in the crown of Bilirubin is a yellow pigment. However, when this pigment
the anterior teeth may be associated with systemic factors undergoes oxidation it becomes a green pigment otherwise
and intubation, because the child remained intubated for 12 called biliverdin. In the past, it was believed that the
days. The literature reveals that tracheal intubation in the accumulation of this pigment was responsible for the green
neonatal period can cause enamel hypoplasia [14, 15]. teeth associated with hyperbilirubinemia. However, a study
The occurrence of hyperbilirubinemia in childhood is an developed by Shibata et al. [33] proved that the green color
important fact because the consequences of this systemic comes from the accumulation of bilirubin and not of
disorder can seriously compromise the color of teeth. The biliverdin.
first report of case about green teeth associated with The correct diagnosis for the cause of dental discolora-
jaundice was performed by Weyers [16] in 1956. Since then, tion is important as, invariably, it has a profound effect on
the dental literature shows some reports of patients who treatment outcomes. According to Shibata et al. [33], the
developed green teeth associated to neonatal hyperbilirubi- diagnosis of bilirubin pigmentation is usually based on
nemia [17–28]. a clinical history of jaundice combined with the green
It is known that the location and extent of pigmentation coloration. Therefore, it would seem reasonable that health
tooth corresponds to the period of tooth formation [28]. In care providers have an understanding of the etiology of
this case report, the patient presented dental pigmentation tooth discoloration in order to diagnose and recommend
in the crown of all deciduous teeth. These dental changes a more appropriate treatment.
corresponded to the period when she was admitted to the Actually, cosmetic dentistry is presented as a source to
Intensive Care Unit. Normally, deciduous tooth calcification meet the esthetic and functional wishes of patients and
process begins in the 4th month of intrauterine life and professionals. The tooth whitening is a technique used to
finishes 11 months after birth. The dental crown of incisors solve the esthetics of discolored teeth. It can be performed for
is completely formed 1 month after birth and the canines both vital and non-vital teeth. Various chemicals such as 35%
and molars after 6 months [29]. According to current hydrogen peroxide, carbamide peroxide and sodium perbo-
knowledge, enamel mineralization proceeds in 2 steps. At rate and others are employed [34]. In general, the esthetic
first, ameloblasts secrete a protein matrix in which hydro- result of whitening of natural teeth has a better result when
xyapatite crystals are deposited up to a concentration of compared to the restoration or resin crowns. In this case, the
about 25%. In a second step, the enamel maturation, enamel natural appearance of the enamel is more esthetic in relation
matrix proteins are degraded and resorbed almost comple- to restorative materials. The first reports of tooth whitening
tely, while hydroxyapatite crystals grow in thickness, until in primary teeth indicate the use of carbamide peroxide 10%
the enamel attains a mineral content of about 95%. Conse- for treatment of dental fluorosis. This technique requires
quences of this formation in 2 stages are also evident in the a high level of adherence of the patient and the esthetic
wisdom teeth showing crown pigmentation [30]. result does not always guarantee long treatment time due to
Enamel formation can be disrupted during the course of the limited life of these teeth [35].
a systemic disorder, and all the teeth that form in this Moreover, some adverse effects of the tooth whitening
period may be marked by bands of enamel malformed. may occur, such as: the external root resorption and dentin
After healing of illness, the formation of normal enamel sensitivity [36]. According to Lee et al. [37], the use in
is restored again. Disorders induced by tetracycline or children and adolescents of some dental bleaching agents
 pediatria polska 90 (2015) 155–160 159

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