Elastosis perforans serpiginosa (EPS) is a rare complication of D-penicillamine therapy. EPS has been reported in patients with Wilson disease, cystinuria, and rheumatoid arthritis after many years of high-dose therapy. We report a case of D-penicillamine-induced EPS with coexisting acquired cutis laxa in a patient with cystinuria. Although both EPS and acquired cutis laxa can be associated with D-penicillamine therapy, few cases have been reported with overlapping clinical presentations, and previously only in patients with Wilson disease. We review the characteristic clinical and histologic features of EPS and discuss the potential dermatologic manifestations of D-penicillamine therapy.