A Case of "Intractable Ulcerating Enterocolitis" of Infant

ABSTRACT

Intractable ulcerating enterocolitis of infancy is uncommon, inhereditary disease characterized by ulcerating stomatitis, severe perianal disease, affecting the whole gastrointestinal tract, mainly colon with flask shaped large ulcer. It was first described by Sanderson et al in 5 cases of infant with intractable diarrhea having above clinical manifestation. It should be differentiated with Crohn's disease and Behcet's disease. We experienced a case of intractable ulcerating enterocolitis in an infant. A 17 month old patient was admitted because of intractable diarrhea since 2 months of age. Radilogical and endoscopic examination revealed chronic ulcerative inflammation with pseudopolyps involving ileum and entire colon. Ileocolectomy was performed because of its unresponsiveness to medical theraphy. The histology of resected specimen showed large flask shaped ulcer with underlying edge in the colon, terminal ileum. No evidence of granuloma suggesting Crohn's disease or vasculitis suggesting Behcet's colitis were noted. We report this case as an example of Intractable ulcerating enterocolitis (Sanderson et al).