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Indian Pediatr 2013;50: 699-701 |
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Neonatal Zygomycosis with Gastric Perforation
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NB Mathur and Aashima Gupta
From Referral Neonatal Unit, Department of
Pediatrics, Maulana Azad Medical College, New Delhi, India.
Correspondence to: Dr NB Mathur, Dir Prof
(Pediatrics), MAM College, New Delhi 110 002, India.
Email: [email protected]
Received: October 23, 2012;
Initial review: November 08, 2012;
Accepted: March 14, 2013.
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Zygomycosis is a rare infection in
neonates. The clinical presentation is non-specific and diagnosis most
often is made at autopsy. Surgical debridement performed early improves
survival. We report a case of neonatal zygomycosis with gastric
perforation.
Keywords: Neonate, Zygomycosis, Intestinal
perforation.
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Neonatal zygomycosis occurs rarely
with only 59 cases described in English literature till 2007
[1,2]. Skin is the most common point of entry for these
patients. Premature infants represent 72% of cases and the
overall mortality rate varies from 64-75% [1,2].
Gastrointestinal tract (GIT) involvement results from
ingestion of fungal spores and prematurity is an important
predisposing factor [1,3]. Early initiation of therapy is
crucial in maximising outcomes and optimal management
strategies have not been defined [4]. We present a case of
neonatal zygomycosis with gastric perforation.
Case Report
A four-day-old male baby weighing 1880
grams presented with abdominal distension for 30 hours. He
was born vaginally at 34 weeks in a private nursing home and
had cried immediately after birth. He received oxygen,
intravenous fluids and intravenous amikacin and
piperacillin-tazobactum for tachypnea, and formula feeds by
nasogastric tube by day 2 of life. The baby developed
abdominal distension on day 3 and feeds were stopped. The
baby was referred to us in view of no improvement. At
admission, he was hypothermic (axillary temperature 35.5 o
C), heart rate was 160/min, respiratory rate was 62/min and
capillary refill time was normal. The oxygen saturation on
room air was 84%. Examination revealed tense distended
abdomen with absent bowel sounds. Rest of systemic
examination was normal. Investigations revealed hemoglobin
17.9 g%, total leucocyte count (TLC) 5050 cells/mm3
(Polymorphs 79%, Lymphocytes 17%), and platelet count 1.83
lac/mm3. The
immature: total neutrophil ratio was 20% and micro
erythrocyte sedimentation rate was 10 mm at the end of first
hour. Cerebrospinal fluid examination was normal. Baby was
administered oxygen by nasal prongs, intravenous fluids and
antibiotics (piperacillin- tazobactum and amikacin) were
continued. X- ray abdomen revealed free gas under
diaphragm. At 12 hours of admission, exploratory laparotomy
was performed, which revealed gangrenous perforation (1 × 2
cm) of anterior wall of stomach with loculated fluid
collection in peritoneal cavity. Excision of the gangrenous
portion and feeding jejunostomy was done. Baby continued to
have bilious nasogastric tube aspirates in the post
operative period. He developed leucopenia (TLC 4500/mm3)
and thrombocytopenia (platelet count- 78,000/mm3)
by fifth post-operative day. Blood urea was high (58mg/dL).
Blood cultures were negative. Gastric wall biopsy received
on eighth post-operative day revealed gangrenous segment
with fungal colonies of zygomycetes with angioinvasion (Fig.
1). Zygomycetes was also isolated from gastric aspirate
samples on eight post-operative day. The baby was started on
intravenous fluconazole on day 8 of hospitalisation.
Jejeunostomy feeds (expressed breast milk) were initiated by
day 8 of antifungal therapy. Repeat gastric aspirates were
negative. Both leucopenia and thrombocytopenia began to
normalise by day 14 of therapy. The baby started gaining
weight and was transited to full jejunostomy feeds by day 16
of therapy. However, parents took the baby home against
medical advice for personal reasons on day 21 of treatment.
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Fig.1 Ulceration of the
gastric mucosa with ulcer bed showing broad
branching fungal hyphae (arrow) entrapped in the
inflammatory granulation tissue (HCE×40). Inset
shows hyphae stained black.
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Discussion
Zygomycosis is the new taxonomic
classification which includes organisms in the order
Entomophthorales and Mucorales [1]. The fungi in the order
Mucorales have been associated with human disease; and are
collectively referred to as zygomycetes [5]. The
predisposing factors for zygomycosis include prematurity,
asphyxia, umbilical catheterization and assisted ventilation
[1,6]. Gastrointestinal tract is the most commonly involved
site in neonates followed by cutaneous involvement [2]. Skin
is the most common portal of entry in neonates with adhesive
tape, monitor leads or central venous access sites
predominating. Gastrointestinal infection results from
ingestion of fungal spores either from environment or from
colonised upper airways. Occasionally hematogenous or direct
extension route can result in intestinal involvement [1].
All sites in GIT can be involved with stomach and colon
being most preferred sites [1]. Premature infants with
gastrointestinal mucormycosis may experience necrotising
enterocolitis with or without pneumatosis intestinalis.
Perforation after bowel wall necrosis is a common occurrence
[1,3]. The baby is generally sick with low counts and may
show a poor response to conventional chemotherapy [2]. The
diagnosis is made on histological examination which may
reveal thrombosis of the small gut vessels with angio-invasion
[1,2,5]. Microbiological cultures are generally negative for
fungi. 56% neonates were diagnosed as zygomycosis on
histology and 44% were diagnosed on histology and culture
[2]. The identification of the zygomycete species is
generally not performed in most clinical laboratories due to
inexperience in the recognition of microscopic differences
among species, unavailability of tester strains to perform
mating studies, or a lack of access to materials needed to
perform molecular diagnostics. Microscopic evidence of a
zygomycete in clinical material from a deep tissue biopsy is
sufficient for the confirmed diagnosis of zygomycosis.
The case fatality is high, especially in
premature neonates [1]. In an in vitro study of 37
isolates of zygomycosis, the mean inhibitory concentration
of various antifungals were reported [7]. Amphotericin B has
been used in most trials, though occurrence of side effects
limits its use [4, 8]. Liposomal amphotericin is less toxic
but often not affordable due to its high cost. Azoles have
been tested and have proved efficacious. Funada, et al.
[9] reported improvement in a patient with pulmonary
mucormycosis with fluconazole. Similarly, Selcen, et al.
[10] reported complete recovery with fluconazole
in a diabetic child with mucormycosis. There is a role of
newer therapies like hyperbaric oxygen, immunotherapy and
iron chelation [4,8].
Mucor is angioinvasive hence surgical
debridement is necessary for radical cure [1,2,4]. Most
patients need repeated surgical debridement. Outcome was
seen to be adversely affected in neonates where surgical
debridement could not be performed [4,8].
To conclude, zygomycosis is a rare life
threatening infection in neonates. It should be suspected in
neonates presenting as acute abdomen. Early diagnosis
combined with medical and aggressive surgical approach is
the mainstay of therapy.
Contributors: NBM: case management,
finalisation of the manuscript and will act as guarantor;
AG: case management and drafting of manuscript.
Funding : None; Competing interest
: None stated.
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