Levamisole is an immunomodulatory agent which is used in the adjuvant therapy of certain malignancies. Agranulocytosis is the most commonly reported hematologic side effect associated with this drug. We report here a patient who developed thrombocytopenia nearly 2 years after starting adjuvant levamisole therapy for malignant melanoma. In this case, thrombocytopenia was shown to be levamisole-related by rechallenge with the drug. Levamisole-induced thrombocytopenia (LIT) has been rarely diagnosed, but may be seen more frequently as increasing numbers of patients receive adjuvant therapy for colon cancer and melanoma.