Introduction: Cytomegalovirus (CMV) is a herpes-type virus. Most of the time, the virus produces mild or no symptoms in healthy people. However, in immunocompromised patient, serious CMV infections can occur. It can develop at any level along the gastrointestinal tract, but perianal ulcer is one of rare form of CMV manifestation. Case description: We presented case of a 33 years old gentleman presented with painless per rectal bleeding for 3 months’ duration. Physical examination revealed a near- circumferential perianal ulcer extending into the rectum. Colonoscopy showed no synchronous proximal lesion. During examination under anesthesia, the ulcer was shown to be extended until distal to dentate line. Wedge skin biopsy was performed with subsequent immunohistochemistry test was consistent with CMV infection. HIV serology test later was positive. Conclusion: Although it is rare, all immunocompromised patients with perianal ulcer should be suspected for CMV infection. Tissue biopsy is mandatory for confirmation of diagnosis. Antiviral therapy is the mainstay of treatment.
 Keywords
 Cytomegalovirus, Immunocompromised Patient, Ulcer, Anal