A 59 year-old Caucasian male with a history of hepatitis C infection, presented in October 2003 with a painless skin lesion on his back. Physical examination revealed a 3 cm lesion on the back that resembled a sebaceous cyst, with similar lesions on the anterior aspects of both legs. A right inguinal lymph node was palpated as well, measuring approximately 2 cm. A wide surgical resection was performed on the back lesion due to its extensive nature at the time of surgery. Pathologic evaluation revealed a primary cutaneous anaplastic large cell lymphoma, measuring 12 cm x 5 cm in size. Immunohistochemical staining showed reactivity for CD30, CD3, UCHL-1, CD43 and CD45. Despite being asymptomatic, the patient had widespread disease on CT/PET scans, including diffuse lymphadenopathy, bony disease, and lung lesions. The patient was treated with weekly high-dose methotrexate at 2 grams/m2 followed by leucovorin rescue for cycle 1 and dose-reduced for cycles 2 through 5 to 1 gram/m2 secondary to myelosuppression. For cycles 6 through 10, the patient was able to tolerate the original dose of 2 grams/m2. After eight cycles, the patient had complete resolution of all disease by clinical examination and by CT/PET scans, and has remained in complete remission since February 2004. The patient tolerated the chemotherapy well with myelosuppression being the most significant toxicity. He also had a transient increase in his liver enzymes after cycle 1 that resolved spontaneously. He is currently being treated for reactivation of hepatitis C with pegylated interferon and ribavirin. To the best of our knowledge, this is the first case of primary cutaneous anaplastic large cell lymphoma with systemic involvement being treated successfully with weekly high-dose methotrexate and leucovorin rescue with prolonged complete remission. Weekly high-dose methotrexate with leucovorin rescue is an active and well-tolerated regimen with limited side effects.

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