1992 Volume 15 Issue 3 Pages 290-296
In this report, we described a case of atypical Crohn's disease associated with Sjögren's syndrome (SjS) who suffered from reccurent pleuritis of unknown origin. A 59 year-old woman started to have episodes of acute polyarthritis from 1970. She suffered from left pleuritis of unknown origin in 1978. In 1987, she had hematemesis and anal bleedings, and then was diagnosed as atypical Crohn's disease lacking colonic lesion, because of oral, esophageal and anal ulcers with a pathological finding of non-caseous granulomatous lesions in the anal ulcer. In March, 1988, she was admitted to our hospital with exertional dyspnea. On admission, multiple oral ulcers, polyarthritis and right pleuritis were found. Laboratory data revealed elevated ESR, hypergammaglobulinemia, positive fluorescent antinuclear antibodies and weakly positive anti-ds-DNA antibody. SjS was diagnosed with the existence of keratoconjunctivitis sicca, positive lip biopsy and positive sialography. After treatment with 3 g/day of salicylazosulfapyridine, pleural effusion and polyarthritis rapidly disappeared and oral lesions became improved.
Crohn's disease is rarely asscciated with SjS. We discussed here possible relationship between these diseases.