A syndrome of severe central nervous system toxicity (confusion, cortical blindness, quadriplegia, seizures, and coma) associated with cyclosporine therapy and a low serum cholesterol level in patients with liver transplants has been described. We present a case history of a patient who demonstrated several similar features after heart-lung transplantation. Possible cyclosporine neurotoxicity should be considered in any patients with hypocholesterolemia receiving this drug.