Sickle cell disease (SCD) is a significant contributor to the global disease burden, particularly in developing countries where access to care is limited, and coordinated multidisciplinary care is often unavailable. Hydroxyurea is the only readily available disease-modifying-drug therapy in many resource-limited settings, such as Jamaica where it is subsidized through the National Health Fund (NHF). This cross-sectional study assessed barriers to healthcare access and hydroxyurea use in Jamaica among adults with SCD and parents or caregivers of children with SCD from four regional health authorities and a specialized SCD centre. Participants completed an interviewer-assisted questionnaire collecting information on sociodemographics, healthcare access, hydroxyurea utilization and associated barriers. All analyses were exploratory. The study recruited 173 adults (mean age 34.0{+/-}11.4 years, 73.6% female, 48.6% urban, 75.1% with secondary education) and 189 parents/caregivers (mean age of parents/caregivers 37.2{+/-}10.2 years and children 7.1{+/-}4.8 years, 55.0% of children female, 50.3% urban, and 64.3% of parents/caregivers with secondary education). Overall, emergency care was most commonly accessed at public hospitals (87.3%), with the SCU (21.5%) and private hospitals (7.7%) less commonly reported. Routine care was accessed at the SCU by 85.9%, with 28.6% also attending hospital outpatient clinics. Enrolment in the National Health Fund (NHF) drug subsidy programme was reported by 50.9% of adults and 36.0% parents/caregivers, while insurance coverage was reported by 34.7% and 31.8%, respectively. Common healthcare access barriers included not being seen quickly enough when in pain (77.8% adults), worry or fear (47.2% overall), long wait times (39.2% overall), frustration or anger (29.3% overall), and high transportation costs (22.4% overall). Hydroxyurea awareness was reported by 62.4% adults and 67.6% parents/caregivers, while 45.0% and 55.0% reported receiving hydroxyurea education, respectively. Awareness and education were both higher among adult females and parents/caregivers of older children, while awareness was also greater among tertiary-educated adults. Overall, 30.2% of adults and 39.4% of children reported initiating hydroxyurea (p=0.18). Among patients advised to take hydroxyurea, 68.4% adults and 77.1% children initiated (p=0.18), and 50.0% adults and 73.4% children (p=0.001) were currently using it. Among adults, lower initiation was associated with reporting insufficient knowledge about hydroxyurea (odds ratio, OR=0.1), disinterest in another medication (OR=0.1), and having neither NHF coverage nor other health insurance (OR=0.1). Among parents/caregivers, initiation and current use were lower among those who were disinterested in another medication (OR=0.1 and 0.02, respectively) and concerned about side effects (both OR=0.2). Poor adherence was most commonly attributed to forgetting to take the medication. Adherence was lower among adults who reported inadequate social support and healthcare being too expensive, including co-pay. Among children, adherence was greater among females, younger children and those with tertiary-educated parents. Findings suggest hydroxyurea awareness and education are inadequate, and initiation and continued use remain suboptimal, highlighting the need for improved education strategies and NHF enrolment. Strategies should be tailored to better engage males, parents and patients of different age groups, and those with lower educational attainment. Greater support is needed to address mental health and transportation challenges, alongside protocols aimed at reducing wait times.