A Case of Adult Hirschsprung's Disease with Obstructive Colitis

Kotaro Ozawa, Tetsuhisa Yamamoto, Toshihiko Yagyu, Hideki Ueno, Manabu Kinoshita, Takashi Ichikura and Hidetaka Mochizuki

Department of Surgery I, National Defense Medical College

A case of adult Hirschsprung's disease with obstructive colitis is reported. A 28-year-old woman hed been suffering from severe constipation and abdominal distension since childhood. Although constipation tendency continued, she was able to have a bowel movement without cathartics after several days. Recently constipation gradually worsened, and she was admitted to our hospital complaining of abdominal distension and vomiting. Barium enema examination revealed a typical change in caliber and dilatation of the sigmoid colon with partial stenosis. Colonscopy revealed a narrowed rectum, a dilatated sigmoid colon with segmental stesosis and a map-like ulcer, and normal mucosa lying between the narrow rectum and the ulcer. Manometry revealed loss of anorectal reflex. Microscopic findings of the map-like ulcer showed a nonspecific inflammation and ganglion cells were present in the dilatated sigmoid colon, but absent in the rectum. In addition, acetylcholine esterase staining revealed the presence of hypertrophied nerve fibers in the rectal biopsy specimen.

Key words
adult Hirschsprung's disease, acetylcholine esterase staining, obstructive colitis

Reprint requests
Kotaro Ozawa Department of Surgery I, National Defense Medical College 2-3 Namiki, Tokorozawa, 359-8513 JAPAN

Accepted
December 9, 1998

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