Purpose.To report a case of dermal and ocular changes after prolonged use of high-dose chlorpromazine hydrochloride therapy.Method.This case report includes clinical history, clinical findings, and photographic images of ocular and dermal changes.Results.Chlorpromazine therapy in a cumulative dosage exceeding 1,100 g resulted in dramatic skin discoloration and multiple crystalline deposits in both corneas. Anterior capsular opacities were binocularly present. These changes were sufficient to cause reduction in visual acuity.Conclusions.Chlorpromazine deposition at high levels can cause reduction in visual acuity and significant skin discoloration.