While laboratory and clinical benefits of hydroxyurea for patients with sickle cell disease (SCD) are wellestablished, few data describe the extent and implications of non-adherence. We sought to assess adherence to hydroxyurea among patients with SCD and investigate associations between adherence and clinical and economic outcomes. Insurance claims of North Carolina Medicaid enrollees (6/2000-8/2008) with SCD were analyzed. Inclusion criteria included age <65 years, continuous Medicaid enrollment 12 months before and following hydroxyurea initiation, and 2 hydroxyurea prescriptions. Three hundred twelve patients, mean age 21 (±12.2) years, met inclusion criteria and 35% were adherent, defined as a medication possession ration (MPR) 0.80; mean MPR was 0.60. In the 12 months following hydroxyurea initiation, adherence was associated with reduced risk of SCD-related hospitalization (hazard ratio [HR] 5 0.65, p 5 .0351), all-cause and SCD-related emergency department visit (HR 5 0.72, p 5

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