Abstract

Crohn’s disease (CD) is a type of chronic inflammatory bowel disease (IBD) which is characterized by the histologic demonstration of granulomas. Besides the gastrointestinal tract, CD may have mucocutaneous manifestations. We report a 58-year-old woman with CD who presented with a 6-month history of painful asymmetric nodular swelling of her right inferior vulva and perineum. A skin biopsy from the perineum revealed scattered dermal noncaseating granulomas, some of which showed a perivascular distribution. In addition, clusters of histiocytes were noted within a few dilated small caliber vessels and lymphatic channels in superficial and deep dermis. The most common clinical condition associated with intravascular/intralymphatic histiocytosis is rheumatoid arthritis. The associations of intravascular/intralymphatic histiocytosis with Crohn’s disease as well as anti-TNF? treatment have also been reported in the literatures. Our patient’s CD is treated with vedolizumab (Entyvio), a monoclonal antibody to integrin ????, rather than a TNF? inhibitor. Therefore, the presence of intravascular/intralymphatic histiocytosis is most likely related to her CD rather than monoclonal antibody treatment. Here, we report a rare event of concurrent cutaneous Crohn’s disease and intravascular/intralymphatic histiocytosis in the same biopsy.

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