Immunosuppressive drugs have been used for many years in the prevention of graft failure in transplant recipients. Although they improve morbidity and mortality after transplantation, these medications carry a significant risk of adverse mucocutaneous and systemic effects. We describe a patient receiving 4 immunosuppressive drugs who experienced persistent facial dysmorphism along with striking follicular disturbances. On histopathologic examination, the follicular structures were dilated and hyperplastic with a peculiar dysplasia of the pilar matrix. Based on a review of the clinical, microscopic, and investigational findings of the skin previously reported in association with her immunosuppressive drugs, we conclude that cyclosporine was the most likely causative agent. Moreover, hypertrichosis, dysmorphic facies, and tissue hyperplasia have all been observed in patients during cyclosporine administration. (J Am Acad Dermatol 2000;43:118-22.)