[Crohn's disease affecting only vulvoperineal area].

INTRODUCTION: Vulvar involvement in Crohn's disease is uncommon. We report here a rare case of Crohn's disease affecting only the vulva and perineum.

CASE: A 55-year-old women had been followed at another hospital since 1995 for histology-proved Crohn's disease affecting only the vulvoperineal area. Treatment with infliximab led to a relapse in 2001. The patient was hospitalized because of a new vulvar and perineal flare-up, with major vulvar edema, aphthoid vulvar and perineal erosions and fissures. Findings from upper endoscopy and colonoscopy were normal. A biopsy sample of the ulcerated tissue showed inflammatory infiltration including histiocytes and macrophages. No microorganisms were found. The initial course was favorable, with systemic corticosteroid therapy and azathioprine. Clinical relapse during the corticosteroid tapering necessitated infliximab.

DISCUSSION: Vulvar localizations of Crohn's disease are uncommon. They may precede gastrointestinal involvement by many years or very rarely be isolated, as here. Typical clinical appearance includes edema and ulcerations. Other causes of granulomatous vulvar and perineal lesions must be ruled out. There is no consensus for its treatment. This case indicates that infliximab, which is used in fistulized Crohn's disease, can be useful for vulvar and perineal involvement. Physicians must recognize that on rare occasions vulvar involvement is possible without any gastrointestinal localization.