Impact of telehealth on hydroxyurea adherence and clinical outcomes in sickle cell disease management: A systematic review and meta-analysis.

BACKGROUND: Sickle cell disease (SCD) is one of the most common chronic blood diseases found among individuals of African heritage. Hydroxyurea (HU) has remained the mainstay pharmacological therapy for SCD, given its disease-modifying effects. Still, its use is limited by a lack of adherence, particularly among adolescents and young adults. Telemedicine has been beneficial in addressing patient nonadherence to chronic conditions but underexplored in the SCD population. OBJECTIVE: To assess the impact of telehealth-based interventions on HU adherence and clinical outcomes in patients with SCD. METHODS: We conducted a comprehensive literature search from January 2014 to March 2025 across PubMed, Web of Science, PsycINFO, and Embase. The included studies were randomized controlled trials and quasi-experimental studies assessing the effect of telemedicine intervention on SCD and clinical outcomes. A random effects model was used to conduct the meta-analysis in R Statistical Software, version 4.4.2. RESULTS: Five studies (n = 353 participants) met the inclusion criteria: 1 cluster randomized controlled trial and 4 quasi-experimental designs. The pooled effect of telehealth for HU adherence (standardized mean difference, 0.91; 95% CI, 0.62-1.20) with moderate heterogeneity (17.8%) was observed alongside increased mean corpuscular volume and fetal hemoglobin in some studies. CONCLUSIONS: Telehealth programs improved HU adherence in the SCD population. However, findings should be interpreted cautiously, given the small number of studies and reliance on quasi-experimental studies. Prospective investigations to evaluate the effect of these telehealth interventions on the diverse SCD genotypes are needed to individualize and optimize strategies to improve HU adherence. TRIAL REGISTRATION: PROSPERO International Prospective Register of Systematic Reviews CRD420251037623.