Alopecia areata is a relatively common nonscarring alopecia that may progress to complete loss of scalp hair (alopecia totalis) or scalp and body hair (alopecia universalis) in a subset of patients. This extreme loss of hair results in cosmetic disfigurement and is notoriously difficult to treat. We report the ongoing successful treatment of a patient with long-standing alopecia totalis using combined methotrexate and prednisone. Our case report adds to the published evidence of the efficacy of this regimen in treating patients with refractory alopecia totalis or alopecia universalis. We conclude that combined methotrexate and prednisone is a promising treatment for long-standing alopecia totalis in patients for whom the potential benefits outweigh the risks.