Cutaneous leishmaniasis is a neglected tropical disease affecting mostly the exposed skin, causing severe and disfiguring lesions in Ethiopia. In this report, we present two cases of atypical mucocutaneous leishmaniasis; one HIV positive and one HIV negative patient. Cases. A 32-year-old male HIV patient presented with 40 days of bleeding per-rectum and a perianal lesion of 5 years. An erythematous nontender plaque measuring 5 cm by 5 cm was observed over the right perianal area with circumferential constricting firm swelling of the rectum. The patient was cured with AmBisome and miltefosine after an incisional biopsy revealed leishmaniasis. A 40-year-old presented with bleeding per-rectum and stool incontinence of 3 months, generalized body swelling of 2 months, and mass around his anus for ten years. A 6 by 3 cm indurated ulcerating mass surrounding the anus and a fungating circumferential mass of 8 cm were seen above the proximal anal verge. An excisional biopsy revealed leishmaniasis, and the patient was treated with AmBisome but passed away due to complications with colostomy diarrhea. Conclusion. Clinicians should consider atypical mucocutaneous leishmaniasis as a possible diagnosis in patients with chronic skin lesions resembling hemorrhoids and colorectal masses, especially in endemic areas such as Ethiopia, regardless of their HIV status.