| Summary: | Introduction: Anal canal melanoma is a rare form of both anal cancer and skin melanoma, representing 1% of all anal cancers. Diagnosis is often made at an advanced disease stage. Because of late diagnosis and aggressive clinical course, anal canal melanoma has a very poor prognosis. Case Presentation: We present the case of a 61-year-old female with anal canal melanoma and lung, liver, pelvic and central nervous system (CNS) metastasis, BRAF V600E mutated. Combined BRAF/MEK inhibition and CNS radiation therapy were able to achieve excellent local and distant disease control. Conclusion: Anal canal melanoma is a rare and aggressive form of melanoma. Multidisciplinary management in experienced centers is mandatory for good clinical outcomes.
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