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Case report

DOI: 10.5958/2319-5886.2015.00188.5
Open Access

PRIMARY GASTRIC ACTINOMYCOSIS: A RARE CASE REPORT

1

Mohit Bhatia , Archana Thakur , Bibhabati Mishra , Vinita Dogra

ARTICLE INFO
th

Received: 26 July 2015

th
Revised: 20 Aug 2015
th
Accepted: 20 Sep 2015
1

Authors details: Senior Resident,

2,4
3
Director
Professor,
Director
Professor & Head,
Department of
Microbiology, Govind Ballabh Pant
Institute of Post Graduate Medical
Education and Research, New Delhi,
India
Corresponding author: Mohit Bhatia
Department of Microbiology, Govind
Ballabh Pant Institute of Post
Graduate Medical Education and
Research, New Delhi, India.
Email: [email protected]

ABSTRACT
Actinomycosis is a chronic disease characterized by abscess formation, tissue
fibrosis, draining sinuses and ulcers caused by the filamentous, gram-positive
anaerobic or microaerophilic bacterial species of the genus Actinomyces.
Actinomycosis mainly presents in three forms namely cervicofacial (31-65%),
abdominopelvic (20-36%) and thoracic (15-30%) respectively. Primary gastric
actinomycosis is extremely rare, with only 24 cases reported till date. We
present a case report of a thirty five years old female patient who was admitted
in a super specialty hospital with complaints of low grade intermittent fever,
abdominal pain and two discharging sinuses on anterior abdominal wall.
Clinical, radiological, microbiological and pathological evaluation revealed
findings suggestive of primary gastric actinomycosis with underlying gastric
adenocarcinoma in this patient. To the best of our knowledge, this is the first
ever report of primary gastric actinomycosis from India.

Keywords:
Actinomyces
spp.,
Primary gastric actinomycosis, Gastric
adenocarcinoma
INTRODUCTION
Actinomycosis is a chronic disease characterized by
abscess formation, tissue fibrosis, draining sinuses and
ulcers. It is caused by the filamentous, gram-positive
anaerobic or microaerophilic bacterial species of the
[1]
genus Actinomyces.
Actinomycosis mainly presents in
three forms: cervicofacial (31-65%), abdominopelvic (20[2,3]
36%) and thoracic (15-30%).
Abdominal actinomycosis
has often been called one of the greatest imitators in
[4]
clinical practice. It often presents as an indolent chronic
suppurative process with atypical symptoms that are
misdiagnosed as neoplasms and other inflammatory
[5]
diseases like tuberculosis or Crohns disease. There is a
predilection for appendix and ileocecal region of the bowel
and thus, it can easily mimic colonic adenocarcinoma,
intestinal tuberculosis, chronic appendicitis or regional
[6]
enteritis.
When outside the intestine, abdominal
actinomycosis generally grows by local spread with rare
incidences
of
haematogenous
or
lymphatic
[7]
dissemination.
Primary gastric actinomycosis is
[8-14]
extremely rare, with only 24 cases reported till date.
The rarity of gastric involvement by Actinomyces spp. has
been attributed to the high luminal acidity of stomach, as a
result of which, the organisms are either killed or their
[11]
growth is inhibited.
We hereby present a case of
primary gastric actinomycosis, which to the best of our
knowledge, is the first ever report from India.

patient revealed that one month prior to the appearance of

discharging sinuses, she had abdominal swelling. There
was also history suggestive of loss of appetite and
significant weight loss. Although, there was no significant
past history suggestive of use of Intra Uterine
Contraceptive Device (IUCD), however, this patient had
undergone cholecystectomy for cholelithiasis at a private
nursing home six months ago. On examination, the patient
was febrile and had pallor. No icterus, cyanosis, clubbing,
lymphadenopathy or edema were observed. Systemic
examination revealed no abnormality except for the
presence of two sinuses discharging frank pus on anterior
abdominal wall. Sinus openings were raised, inflamed and
flared up [Figures 1a & 1b].

A thirty five years old female patient was admitted in a

super specialty hospital with complaints of low grade
intermittent fever, abdominal pain and discharging sinuses
on anterior abdominal wall of one week duration. The

Fig 1:(a & b). Discharging sinuses on anterior

abdominal wall
The following investigations were carried out, the results of
which are as follows: Complete haemogram revealed
anaemia (hemoglobin: 5.5 gram%) and leucocytosis (total
leucocyte count: 20,400/cu.mm of blood) respectively.

Mohit et al.,

Int J Med Res Health Sci. 2015;4(4):921-924

CASE REPORT

921

Ultrasonography of abdomen revealed eccentric

asymmetric wall thickening of gastric antro-pyloric region
of maximum thickness 3 cm with loss of mural
stratification. Few enlarged perigastric lymph nodes of size
12 mm10 mm each were also seen. Contrast enhanced
computerized tomography (CECT) of abdomen revealed
thickening of gastric mucosa with formation of a mass
extending to anterior abdominal wall and present up to
transverse colon. Upper gastro intestinal endoscopy
revealed the presence of a large area of elevated surface
of size 4 cm5cm near lesser curvature of stomach. Two
openings each of size 1cm1cm with smooth margins
probably containing some necrotic material were
visualized in this area. The overlying mucosa of elevated
surface appeared to be slightly nodular and abnormal.
Gastric biopsy samples collected during this procedure
were subjected to histopathological examination which
revealed the presence of neoplastic glands (present in
lamina) lined by cells showing nuclear pleomorphism,
stratification, high Nuclear: Cytoplasmic ratio and
moderate amount of eosinophilic cytoplasm suggestive of
moderately differentiated gastric adenocarcinoma.
Pus and necrotic material collected from sinuses was
subjected to Gram stain, Ziehl-Neelson stain and aerobic
culture and sensitivity. On macroscopic examination, the
pus was yellow to brown in color, thick, fowl smelling with
presence of numerous granules which were yellow to
white in color and approximately 0.5-1mm in size. Gram
stain of pus sample revealed the presence of numerous
pus cells, occasional Gram negative bacilli and numerous
Gram positive non-fragmented, non-branching, thin
filamentous bacteria [Figure 2].

Fig 2: Gram stain of pus sample showing Gram

positive, non-branching, thin filamentous bacteria and
Gram negative bacilli (1000X)
No acid fast bacilli were seen on Ziehl-Neelson stain using
3% acid-alcohol as decolorizer. Taking all sterile
precautions, the granules present in pus sample of this
patient were transferred to several glass slides and
crushed with the help of cover slips and wooden handle of
inoculating wire. Potassium hydroxide mount examination
of crushed granules revealed the presence of numerous
aseptate filamentous structures approximately 0.5 to 1m
in size [Figure 3]. Gram stain and modified Ziehl-Neelson
stain (using 1% sulfuric acid as decolorizer) of crushed
granules respectively revealed the presence of numerous
Gram positive and non-acid fast, non-fragmented, nonbranching, thin filamentous bacteria without any chains of
spores suggestive of Actinomyces spp [Figures 4 & 5].

Fig 3: KOH mount of crushed granules showing

aseptate, non-branching, thin filamentous structures
suggestive of Actinomyces spp. (400X)

Fig 4: Gram stain of crushed sulfur granules revealing

Gram positive non-branching, thin filamentous
bacteria (1000X)

Fig 5: Modified Ziehl-Neelson stain showing non acid

fast
structures
morphologically
resembling
Actinomyces spp. (1000X)
Escherichia coli sensitive to amikacin, imipenem,
meropenem and ertapenem was isolated after twenty four
hours of aerobic incubation of pus sample. In lieu of the
clinical picture, aforementioned macroscopic and
microscopic findings, special requisition was sent from the
laboratory to the clinicians for sending some more pus and
necrotic debris, collected from sinuses of this patient, for
performing anaerobic culture for confirmation of
actinomycosis. However, by the time this requisition was
received by the clinicians, the patient had deteriorated

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Int J Med Res Health Sci. 2015;4(4):921-924

clinically and finally expired due to cardiac arrest owing to

which a second sample could not be collected.
Suggestive history, predisposing factors in the form of
surgery and presence of gastric adenocarcinoma,
endoscopic findings and presence of discharging sinuses,
macroscopic and microscopic microbiological findings
suggest that it was a case of primary gastric
actinomycosis.
DISCUSSION
Actinomyces spp. are often found as saprophytes in the
oral cavity, gastrointestinal and female genital tract. The
destruction of the muscular barrier by trauma in the form
of
surgeries,
endoscopic
manipulation,
immune
suppression as in leukemia, lymphoma and other
malignancies, renal insufficiency, renal transplant,
diabetes and chronic inflammatory diseases are
recognized as predisposing factors for penetration
[15]
by these organisms.
Upon penetration, these
organisms in turn result in characteristic clinical
manifestations in different parts of the body at varying
frequencies. Primary gastric actinomycosis frequently
presents as low-grade fever, epigastric pain, weight loss,
upper GI bleeding and rarely symptoms of gastric outlet
[3,12,14]
obstruction.
The patient under study presented with
similar clinical features suggestive of actinomycosis. She
had undergone cholecystectomy six months ago and was
diagnosed on histopathological examination as a case of
gastric adenocarcinoma later on, both of which are
predisposing
factors
for
acquiring
abdominal
actinomycosis.
There are no specific radiological or endoscopic findings
suggestive of this condition. CT findings mostly
demonstrate an infiltrative lesion with diffuse gastric wall
thickening suggestive of gastric adenocarcinoma or
[2,14]
lymphoma.
Endoscopic findings of the disease may
simulate a gastric neoplasm and include submucosal
tumor-like or infiltrative lesions and occasionally, mucosal
ulceration. In the present case, endoscopic findings
revealed elevated surface and two openings containing
necrotic material near the lesser curvature of stomach.
Because of the submucosal localization of the
inflammatory process, endoscopic biopsy specimens
[14]
usually reveal nonspecific inflammatory changes. Out of
the twenty four reported cases of primary gastric
actinomycosis, only three cases have been diagnosed
pre-operatively by histopathological examination of
[9,10,14]
endoscopic biopsy specimens.
In a case reported by
Khaleel Al-Obaidy et al, the patient was subjected to upper
gastrointestinal endoscopy twice, carried out one week
apart. Actinomycosis was detected by histopathological
examination only in the second biopsy which was obtained
from an area containing brownish fibrinopurulent
[14]
inflammatory exudate.
In the present case also it is
possible that biopsy samples may not have been obtained
from the necrotic area and therefore, the diagnosis of
actinomycosis was missed. However, histopathological
examination revealed gastric adenocarcinoma as the most
probable underlying cause of acquiring this infection in
contrast to most reported cases of gastric actinomycosis,

in which it was impossible to trace the mechanism by

[14]
which Actinomyces spp. had reached the gastric wall.
Actinomycosis is often but not always characterized by the
presence of sulfur granules which occur in 50% of the
cases. Although the presence of sulfur granules strongly
suggests a diagnosis, these are not pathognomonic for the
[16,17]
disease.
The differential diagnosis of sulphur granules
includes nocardiosis, streptomycosis, chromomycosis,
[18]
eumycetoma and botryomycosis.
In the case under
study, preliminary microbiological examination of pus and
necrotic material obtained from discharging sinuses
revealed the presence of organisms morphologically
resembling Actinomyces spp and largely ruled out the
aforementioned conditions.
Culture is negative in >76% cases of gastric
[14]
actinomycosis.
In our case, aerobic culture of pus and
necrotic material obtained from discharging sinuses
yielded Escherichia coli. Generally typical actinomycotic
lesions contain one to ten bacterial species in addition to
the pathogenic actinomycetes. These bacteria are
responsible for early symptoms of the disease and
treatment failures in addition to acting as probable
synergistic pathogens that strengthen the comparatively
[19]
low
invasive
power
of
Actinomyces
spp.
Whether Escherichia coli, which is known to cause
[20]
botryomycosis , had contributed to the development of
mycetoma in our case remains unclear. Unfortunately,
anaerobic culture for isolation and speciation of
Actinomyces spp. could not be attempted due to early
death of the patient.
CONCLUSION
Primary gastric actinomycosis is an extremely rare
disease and often missed by routine diagnostic tests if the
sample is not specifically collected from the affected area.
A high level of suspicion is required both by
gastroenterologists and pathologists in order to correctly
diagnose this condition. Actinomycosis should be
considered in the differential diagnosis of radiological and
upper gastrointestinal endoscopic findings of gastric wall
thickening, particularly in patients with history of
abdominal surgery, trauma or immune compromised
status. In order to avoid the possibility of missing the
diagnosis, the pathologists should be more vigilant and
employ appropriate staining techniques when gastric
endoscopic biopsy samples reveal the presence of subtle
changes such as inflammatory exudates.
ACKNOWLEDGEMENTS
Department of Gastroenterology, Govind Ballabh Pant
Institute of Post Graduate Medical Education and
Research, New Delhi
Conflict of Interest: Nil
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