A patient with extensive cutaneous chronic graft-versus-host disease (cGVHD) affecting 100% of the body surface, with painful ulcerations that involved 20% of it, was treated unsuccessfully during 9 months with steroids, cyclosporine-A (CSa), sirolimus, tacrolimus, mychophenolate mofetil (MMF), infliximab, and rituximab. Twenty-one months after the allograft the patient was started on alemtuzumab, 10 mg/day subcutaneously, for 6 consecutive days every 4 weeks. Seven months after starting the treatment, 100% of the ulcers had disappeared, as had the pain. To our knowledge, there are no reports of the use of alemtuzumab in the treatment of extensive, ulcerated, refractory cutaneous cGVHD. The data presented here suggest that this agent may be useful in some patients with refractory forms of cGVHD.