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Tuberculosis cutis orificialis (TCO) is a rare manifestation of cutaneous tuberculosis. Due to its variable clinical features, the diagnosis may be missed at the onset of the disease. Here we describe, TCO as well as intestinal tuberculosis in a patient with recurrent non-healing perianal ulcer. First TCO was detected, later on intestinal T.B was also detected. The perianal lesions appeared as multiple outbreaks of ulcer which resolved totally after combined anti T.B treatment. The clinical course of intestinal lesions and tuberculosis cutis orificialis appeared interrelated. Ó 2014 Production and hosting by Elsevier B.V. on behalf of King Saud University. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/3.0/).