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ABSTRACTS: Poster Session Abstracts

P0533 DISSEMINATED HISTOPLASMOSIS ASSOCIATED WITH SCLEROSING CHOLANGITIS

Kirkham, S. E.1; Heuschkel, R. B.1; Murch, S. H.1; Thomson, M. A.1

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Journal of Pediatric Gastroenterology and Nutrition: June 2004 - Volume 39 - Issue - p S259
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Introduction: A case is presented of a 12 year old boy with a history of recent travel to a histoplasmosis endemic area presenting with epigastric pain. During admission the patient developed fever and severe right upper quadrant pain associated with an elevation in transaminases and inflammatory markers (peak ALT 206, peak CRP 218). Six weeks prior to presentation he had returned from a trip to the Blue Ridge region of Virginia, an area known to have a high incidence of histoplasmosis. The fungus histoplasma capsulatum is increasingly recognised as an important pathogen in patients with immunocomprise, particularly in endemic areas. Although involvement of the gastrointestinal tract is common in disseminated histoplasmosis, gastrointestinal manifestations are considered rare, occurring in 2–12% of patients (1). Granulomatous inflammation of the liver is not an uncommon manifestation (2). Sclerosing cholangitis associated with histoplasmosis has not been previously described.

Methods: Extensive investigation was conducted to define or exclude a pathogen in this patient with a presumed atypical presentation of inflammatory bowel disease.

Results: Endoscopy demonstrated numerous volcano like ulcers throughout the oesophagus, and microaphthoid ulceration of the stomach and duodenum. Histology revealed a focal acute and chronic granulomatous oesophagitis. Mild chronic inflammation was described in the gastric, duodenal and colonic biopsies. The histology was interpreted as suggestive of Crohn’s disease. Histoplasma antibody was elevated at a titre of 1:16 rising to 1:64. Complement fixation and histoplasma antigen were also positive. Serology to a variety of other pathogens including leptospira, blastomyces and cryptosporidium were negative. Fungal culture of liver and oesophageal tissue is negative to date. Magnetic resonance cholangiopancreatography showed marked irregularity of the biliary tree in the left lobe of the liver suggestive of sclerosing cholangitis. Subsequent ERCP confirmed these findings. Autoimmune markers including anti-LKM were negative.

Conclusion: Histoplasmosis is an unusual cause of granulomatous inflammation in non-endemic areas and rarely considered. This case illustrates the importance of caution in instituting immunosuppressive treatment in patients with suspected inflammatory bowel disease where immunosuppression is suspected or there is a history of foreign travel. A possible association of histoplasmosis with sclerosing cholangitis has not been previously described.

Reference(S):

1. Cappell S, Mandell W, Grimes MM, Heu HC. Gastrointestinal histoplasmosis. Dig Dis Sci 1988 33: 353–60
2. Lamps LW, Molina CP, West AB, Haggit RC, Scott MA. The pathologic spectrum of gastrointestinal and hepatic histoplasmosis. Am J Clin Path 2000 Jan 113 (1) 64–72
© 2004 Lippincott Williams & Wilkins, Inc.