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Microsporidial AIDS cholangiopathy due to Encephalitozoon intestinalis: case report and review.
Am J Gastroenterol. 2000 Sep; 95(9):2364-71.AJ

Abstract

Microsporidia are increasingly recognized as opportunistic infections in immunodeficient patients, predominantly patients with AIDS. The two microsporidia most commonly associated with disease in AIDS patients are Enterocytozoon bieneusi and Encephalitozoon intestinalis (previously known as Septata intestinalis). The most common clinical presentation of microsporidiosis in AIDS patients is diarrhea, most commonly caused by the Enterocytozoon bieneusi species. Encephalitozoon intestinalis is a recently described species that has been reported to cause disseminated human infection including cholangitis. We report a case of AIDS cholangiopathy that presented with abdominal pain and cholestatic liver tests. Ultrasound examination and ERCP revealed a picture of sclerosing cholangitis. Bile samples obtained at ERCP were negative for microsporidia; stool studies for microsporidia and cryptosporidia were also negative. No organisms were identified on routine light microscopy of the biopsy specimens from the duodenum, ampulla, and bile duct. E. intestinalis spores were demonstrated in the bile duct biopsies, by methylene blue and azure 11 staining and confirmed by electron microscopy. Albendazole therapy was successful in eradicating E. intestinalis with clinical improvement and improvement in CD4 count. However, the cholangiographic picture did not improve and repeat cholangiography revealed progressive bile duct injury. Albendazole therapy was delayed and may have been too late to prevent bile duct damage; the drug had to be approved by the US Food and Drug Administration for compassionate use. This is an unusual case of sclerosing cholangitis caused by an unusual organism and requiring biliary sphincterotomy and stent placement for progressive stricturing despite eradication of the infection.

Authors+Show Affiliations

Department of Internal Medicine, University of California Davis, Sacramento 95932, USA.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article
Review

Language

eng

PubMed ID

11007244

Citation

Sheikh, R A., et al. "Microsporidial AIDS Cholangiopathy Due to Encephalitozoon Intestinalis: Case Report and Review." The American Journal of Gastroenterology, vol. 95, no. 9, 2000, pp. 2364-71.
Sheikh RA, Prindiville TP, Yenamandra S, et al. Microsporidial AIDS cholangiopathy due to Encephalitozoon intestinalis: case report and review. Am J Gastroenterol. 2000;95(9):2364-71.
Sheikh, R. A., Prindiville, T. P., Yenamandra, S., Munn, R. J., & Ruebner, B. H. (2000). Microsporidial AIDS cholangiopathy due to Encephalitozoon intestinalis: case report and review. The American Journal of Gastroenterology, 95(9), 2364-71.
Sheikh RA, et al. Microsporidial AIDS Cholangiopathy Due to Encephalitozoon Intestinalis: Case Report and Review. Am J Gastroenterol. 2000;95(9):2364-71. PubMed PMID: 11007244.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Microsporidial AIDS cholangiopathy due to Encephalitozoon intestinalis: case report and review. AU - Sheikh,R A, AU - Prindiville,T P, AU - Yenamandra,S, AU - Munn,R J, AU - Ruebner,B H, PY - 2000/9/28/pubmed PY - 2000/10/14/medline PY - 2000/9/28/entrez SP - 2364 EP - 71 JF - The American journal of gastroenterology JO - Am J Gastroenterol VL - 95 IS - 9 N2 - Microsporidia are increasingly recognized as opportunistic infections in immunodeficient patients, predominantly patients with AIDS. The two microsporidia most commonly associated with disease in AIDS patients are Enterocytozoon bieneusi and Encephalitozoon intestinalis (previously known as Septata intestinalis). The most common clinical presentation of microsporidiosis in AIDS patients is diarrhea, most commonly caused by the Enterocytozoon bieneusi species. Encephalitozoon intestinalis is a recently described species that has been reported to cause disseminated human infection including cholangitis. We report a case of AIDS cholangiopathy that presented with abdominal pain and cholestatic liver tests. Ultrasound examination and ERCP revealed a picture of sclerosing cholangitis. Bile samples obtained at ERCP were negative for microsporidia; stool studies for microsporidia and cryptosporidia were also negative. No organisms were identified on routine light microscopy of the biopsy specimens from the duodenum, ampulla, and bile duct. E. intestinalis spores were demonstrated in the bile duct biopsies, by methylene blue and azure 11 staining and confirmed by electron microscopy. Albendazole therapy was successful in eradicating E. intestinalis with clinical improvement and improvement in CD4 count. However, the cholangiographic picture did not improve and repeat cholangiography revealed progressive bile duct injury. Albendazole therapy was delayed and may have been too late to prevent bile duct damage; the drug had to be approved by the US Food and Drug Administration for compassionate use. This is an unusual case of sclerosing cholangitis caused by an unusual organism and requiring biliary sphincterotomy and stent placement for progressive stricturing despite eradication of the infection. SN - 0002-9270 UR - https://www.unboundmedicine.com/medline/citation/11007244/Microsporidial_AIDS_cholangiopathy_due_to_Encephalitozoon_intestinalis:_case_report_and_review_ L2 - https://Insights.ovid.com/pubmed?pmid=11007244 DB - PRIME DP - Unbound Medicine ER -