The attitudes of patients with sickle cell disease (SCD) toward the use of hydroxyurea (HU) therapy may contribute to the underutilization of HU in the United States, yet our understanding of these attitudes is limited. We examined the attitudes and beliefs of 94 adult SCD patients, comparing those who never used HU (n 5 37), formerly used HU (n 5 23), and were currently using HU (n 5 34). Seventy percent of current HU users reported some level of improvement from the drug ("average" or "very much") and 80% reported little or no trouble from side effects. Fifty-seven percent of former users reported taking HU for less than 6 months, with "doctor"s recommendation," or "not liking the way it made me feel" given as the most commonly reported reasons for stopping HU. Fifty percent of the never users reported receiving no information about HU from any source, and 85% of the never users thought that they would receive no improvement if they were to take HU. A deeper understanding of patient perspectives toward HU utilization is required as part of multipronged efforts to combat its underutilization in the treatment of SCD.