Sickle cell disease (SCD) is the most common inherited hematologic disease. Studies have shown that comprehensive medical care can decrease adverse outcomes associated with SCD, but such care is not easily accessed. It is estimated that less than 70% of children with SCD receive comprehensive care, and only 20% experience effective care coordination. Additionally, comprehensive sickle cell centers are most commonly located in major metropolitan areas, making it difficult for families in rural or medically underserved areas to access comprehensive care. Telemedicine, the remote provision of medical care using modern information technology, is a potential strategy to address the barriers experienced by patients with SCD living in rural and medically underserved areas. Not only have studies demonstrated that telemedicine is safe and effective, but the current SARS-CoV2 pandemic has shown how it can increase local access to subspecialty care. My preliminary work in preparation for this resubmission demonstrated the feasibility and acceptability of telemedicine for SCD care in a rural part of Indiana (AHRQ K12 HS026390). My long- term goal is to improve access and adherence to quality care for children with SCD. The objective of this application is to investigate the use of various telemedicine models to provide comprehensive care to children with SCD in rural and medically underserved communities without ready access to specialized services. We will use the results gathered from our preliminary K12 work to adapt, refine, and pilot an existing hub-and-spoke model and a direct-to-consumer model. I hypothesize that both telemedicine models will be a feasible means of providing evidence-based comprehensive pediatric SCD care to patients in rural or medically underserved areas, with caregivers and patients having decreased barriers to care and improved caregiver/patient-centered outcomes with the direct-to-consumer model. The specific research aims of this proposal are: (1) Adapt two telemedicine models for use with children with SCD using caregiver input from our preliminary K12 work; (2) Demonstrate the feasibility of the telemedicine models developed in Aim 1 as they undergo successive stakeholder refinement during use in actual clinical care; (3) Evaluate the effectiveness of the refined models from Aim 2 in a pre/post study by assessing (a) process of care measures, (b) provider satisfaction, (c) caregiver/patient-centered outcomes, and (d) clinical outcomes and healthcare utilization. I will apply for R21/R03 funding to scale-up the models to additional communities statewide based on how each performs in different environments. I will also apply for R01 funding to assess, through a randomized controlled trial, their effect on health care utilization, disease outcomes, and caregiver/patient-centered outcomes. This work can be adapted to evaluate the impact of telemedicine on issues like hydroxyurea use and adherence. The proposed research will also h...