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Imatinib, a tyrosine kinase inhibitor, is the current preferred agent for the treatment of unresectable or metastatic dermatofibrosarcoma protuberans (DFSP). We present the first case report with exacerbated psoriasis during imatinib treatment for recurrent DFSP. The condition of a 63-year-old man with a history of psoriasis vulgaris for 17 years was controlled well by topical calcipotriol. He suffered from a recurrent DFSP, and the molecular pathology analysis showed OL1A1–PDGFB fusion transcript. Imatinib (400 mg daily) was commenced after surgical excision, and 4 weeks later he developed generalized psoriasis that was poorly controlled by regular treatment. After discontinuation of imatinib, combined with systemic antihistamine and topical corticosteroid, the lesions improved in 8 weeks. We further reviewed the literature to characterize the imatinib-induced psoriasis.