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Year : 2011  |  Volume : 54  |  Issue : 3  |  Page : 664-665
Gastrointestinal mucormycosis in an infant


Department of Paediatric Surgery, Chacha Nehru Bal Chikitsalaya and Maulana Azad Medical College, Geeta Colony, Delhi, India

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Date of Web Publication20-Sep-2011
 

How to cite this article:
Mohta A, Neogi S, Das S. Gastrointestinal mucormycosis in an infant. Indian J Pathol Microbiol 2011;54:664-5

How to cite this URL:
Mohta A, Neogi S, Das S. Gastrointestinal mucormycosis in an infant. Indian J Pathol Microbiol [serial online] 2011 [cited 2020 Nov 29];54:664-5. Available from: https://www.ijpmonline.org/text.asp?2011/54/3/664/85149


Sir,

Gastrointestinal zygomycosis is an uncommon condition and carries a very high mortality. We report an infant who presented with features suggestive of malrotation with volvulus but was diagnosed to have intestinal zygomycosis which proved fatal.

A one-year-old girl was brought to the emergency department with history of fever, lethargy and refusal to feed and loose stools. She had an episode of bleeding per rectum followed by obstipation and bilious vomiting. On examination, she had pulse rate of 160 per minute and respiratory rate of 25 per minute. Abdomen was distended with an ill-defined abdominal mass with marked rigidity, tenderness and guarding. A clinical diagnosis of malrotation with volvulus was made. The investigations revealed hemoglobin of 9 g/dL; total leukocyte count of 7300/mm 3 ; blood urea level at 133.4 mg/dl; serum creatinine - 2.61 mg/ dl and normal serum electrolytes. X-ray abdomen in the erect position revealed a gas-less abdomen with a fundal gas shadow. Exploratory laparotomy revealed 100 mL of serous non-hemorrhagic peritoneal fluid; no evidence of malrotation or volvulus; but there was gangrene of terminal 45 cm of ileum [Figure 1]. Resection of the gangrenous ileum with divided ileostomy was done in view of doubtful viability. Despite uneventful initial recovery, she continued to remain lethargic. The pre-operative blood culture was reported as sterile. On post-operative day 2, she developed altered sensorium and decerebrate rigidity. Examination did not show signs of anastomotic leak. She was shifted to the intensive care unit (ICU), where blood and cerebro-spinal fluid (CSF) cultures were taken and were found to be sterile. Clinical condition of the child kept deteriorating despite intensive care and ventilatory support and she expired. Histopathology was suggestive of intestinal necrosis with mucormycosis [Figure 2] and [Figure 3].
Figure 1: Operative photograph showing regional gangrene of the intestine along with thinning of the intestinal wall

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Figure 2: Transmural gangrene small intestine with marked hemorrhage and vascular congestion (H and E, ×100). Submucosa (inset) shows large number of broad, thin-walled hollow appearing fungal hyphae invading the vessel wall and forming thrombi in the vessels (angiotropism) (H and E, ×400)

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Figure 3: PAS stain showing broad fungal hyphae with right-angled branching (periodic Schiff stain, ×600)

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Zygomycosis (mucormycosis) is an opportunist mycotic infection caused by fungi of the order Mucorales. Gastrointestinal mucormycosis is extremely uncommon in neonates and infancy and less than 20 cases have been described from India. [1],[2],[3] In the pediatric age group, the mechanism of entry is thought to be ingestion. [4] Although any part of the gastrointestinal tract may be involved, stomach and colon are most commonly involved. [1],[4] It occurs in the presence of lowered host resistance, and based on clinical presentation and the involvement of a particular anatomic site, it has been classified into at least six clinical categories. [1] Zygomycosis of the gut in neonates is rare, difficult to recognize and usually fatal. It carries a mortality rate of about 70% and very few survivors have been reported in the neonatal age group. [5]

There are no reliable, serologic, polymerase chain reaction (PCR)-based or skin tests for zygomycosis. Therefore, the diagnosis is usually made by biopsy of infected tissues. [1] The biopsy demonstrates the characteristic wide, ribbon-like, aseptate hyphal elements that branch at right angles, as seen in the present case. A hallmark of zygomycosis infections is the virtually uniform presence of extensive angioinvasion with resultant vessel thrombosis and tissue necrosis.

A sterile blood culture does not rule out the infection, and waiting for the results of the fungal culture may delay the institution of appropriate therapy. In the absence of suitable diagnostic tests, knowledge of this condition as a possible cause of intestinal ischemia and high index of suspicion is required to make an early diagnosis and management. It is important to initiate vigorous supportive medical management and antifungal agents after adequate surgical excision with clear margins.


   Acknowledgement Top


We wish to thank Prof Nita Khurana of Department of Pathology, Maulana Azad Medical College for providing histopathology slides.

 
   References Top

1.Spellberg B, Edwards J Jr, Ibrahim A. Novel perspectives on mucormycosis: Pathophysiology, presentation, and management. Clin Microbiol Rev 2005;18:556-69.  Back to cited text no. 1
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2.Dhingra KK, Mandal S, Khurana N. Unsuspected intestinal mucormycosis in a neonate presenting as necrotizing enterocolitis (NEC). Eur J Pediatr Surg 2008;18:119-20.  Back to cited text no. 2
[PUBMED]  [FULLTEXT]  
3.Agarwal K, Sharma M, Singh S, Jain M. Antemortem diagnosis of gastrointestinal mucormycosis in neonates: Report of two cases and review of literature. Antemortem diagnosis of gastrointestinal mucormycosis in neonates: report of two cases and review of literature. 2006;49:430-2.  Back to cited text no. 3
[PUBMED]    
4.Karanth M, Taniere P, Barraclough J, Murray JA. A rare presentation of zygomycosis (mucormycosis) and review of the literature. J Clin Pathol 2005;58:879-81.  Back to cited text no. 4
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5.Siu KL, Lee WH. A rare cause of intestinal perforation in an extreme low birth weight infant-gastrointestinal mucormycosis: A case report. J Perinatol 2004;24:319-21.  Back to cited text no. 5
[PUBMED]  [FULLTEXT]  

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Correspondence Address:
Anup Mohta
28-B, Pocket-C, S.F.S. Flats, Mayur Vihar Phase-III, Delhi - 110 096
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.85149

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    Figures

  [Figure 1], [Figure 2], [Figure 3]

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