We report a case of epidermodysplasia verruciformis (EV)-like lesions in a patient with graft-versus-host disease after peripheral blood stem cell transplantation from his HLA-matched brother. The patient presented with a diffuse papular eruption that was clinically consistent with graft-versus-host disease; however, histopathology demonstrated viral cytopathic changes and polymerase chain reaction confirmed EV human papillomavirus types 8 and 20. Repeated biopsy specimen showed both human papillomavirus cytopathic effect and graft-versus-host disease, and further workup revealed ocular and hepatic involvement. This progressed to a lupuslike syndrome with lichenoid, violaceous, flat-topped papules in a malar distribution and positive antinuclear autoantibodies. Although EV-like lesions have been reported in patients who are immunocompromised, the incidence is low, and may be linked to EV-related haplotypes.