E. Bourrat et al., ANITIS, VULVAR EDEMA AND MACROCHEILITIS REVEALING A CROHNS-DISEASE INA CHILD - EFFICACY OF METRONIDAZOLE, Annales de dermatologie et de venereologie, 124(9), 1997, pp. 626-628
Background We report a case of oro-genital and perianal Crohn's diseas
e which progressed for 4 years with no digestive involvement in a 12-y
ear-old girl. Case report At the age of 8 years, a young girl develope
d recurrent anal fissures. Voluminous vulvar edema developed at 12 yea
rs with fissurar macrocheilitis. There were no digestive signs and the
diagnosis of Crohn's disease was obtained on the basis of granulomato
us epithelioid infiltration of biopsy specimens (Bauhin valve, anus, v
ulva). Metronidazole given per os at the dose of 25 mg/kg/day for 6 mo
nths led to partial significant symptomatic remission. Discussion Unil
ateral or bilateral vulvar edema is highly suggestive of Crohn's disea
se even if the classical digestive inflammatory signs are absent. Demo
nstration of perianal lesions (erythema, pseudocondylomatous formation
s) must not mislead the diagnosis (sexual abuse). Simultaneous granulo
matous lesions in the genital and labial regions is exceptional in Cro
hn's disease. Ileocolonoscopy is indicated in such cases and alone can
demonstrate latent digestive inflammatory processes. Different agents
have been proposed for treatment of local Crohn's disease skin lesion
s, IL is difficult to evaluate their efficacy due to the spontaneous v
ariability of disease expression.