This case report describes the sustained symptomatic and hematologic improvement in a 21-year-old woman with homozygous sickle cell (ss) disease during treatment with pentoxifylline, 400 mg three times daily after meals. Pain crises decreased from six to zero per year, hemoglobin level rose from 8.4 g/dL to 11.4 g/dL, hematocrit rose From 24.8% to 34.8%, lactate dehydrogenase level decreased from 375 IU/L to 322 IU/L, and total bilirubin level decreased from 1.8 mg/dL to 1.6 mg/dL. Mean corpuscular hemoglobin increased From 21.6 pg to 30 pg and mean corpuscular hemoglobin concentration increased from 24.1 g/dL to 34.5 g/dL. These changes were sustained for seven years except for a brief self imposed hiatus in therapy during which period a pain crisis occurred. Further increase in pentoxifylline dosage to 400 mg four times daily did not result in any further improvement in these hematologic parameters. These results suggest that pentoxifylline reduces hemolysis in SS patients with a resulting improvement in anemia and a reduction in or elimination of pain crises.