Two children, 19 months of age, were independently treated with a compassionate protocol of etretinate therapy for recalcitrant, debilitating pustular psoriasis. Laboratory test results, roentgenograms of the spine, and bone age were periodically monitored. Over a 31/2-year period of intermittent treatment with a maximum dosage of 1.5 mg/kg/d, both children showed remarkable improvement with no apparent drug effect on growth and development. Side effects included xerosis, skin fragility and transient, minimal elevations of aspartate aminotransferase, lactic dehydrogenase, and triglyceride levels. Etretinate therapy may prove to be a viable treatment option for the child with intractable pustular psoriasis that seriously impairs quality of life.