Craniofacial microsomia: Accelerating Research and Education (CARE)

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Project Summary/Abstract Craniofacial microsomia (CFM) is the third most common congenital craniofacial condition and typically involves underdevelopment of the facial structures, including the ear (e.g., microtia) and jaw (e.g., mandibular hypoplasia). Individuals with CFM have complex healthcare needs and frequently require a coordinated evaluations and interventions to optimize hearing, neurodevelopment, breathing, and minimize the impact of extra-cranial malformations. We have established an international community of individuals affected by CFM, healthcare providers, and advocacy leaders to facilitate research in CFM. We are currently conducting a large, international study in which we are exploring the lived experiences of individuals with CFM and exploring the healthcare and psychosocial experiences of this population. We are collecting multi-perspective data from English and Spanish speaking individuals with CFM and their caregivers across the treatment pathway. Early in 2023, NIH introduced new data sharing requirements for NIH-funded studies. Data sharing allows investigators to maximize the usefulness of existing data to answer important research questions. However, we lack information from the craniofacial community regarding stakeholder preferences about sharing of clinical phenotypic data used for research. This is an important issue especially for participants with rare and visible conditions. From an ethical perspective, it’s critical to develop guidelines that reflect participants lived experiences and values. To our knowledge, this has not been done in individuals with craniofacial conditions for sharing of phenotypic data. We propose to advance knowledge regarding data sharing in clinical craniofacial research by addressing the following specific aims: Aim 1. Explore patient and caregiver perspectives on research data sharing for craniofacial research. Aim 2. Synthesize empirical data and ethical norms to produce recommendations for data sharing in patient-oriented craniofacial research. As part of this proposal, we have established a CARE Ethics of Data Sharing Committee composed of multiple stakeholders, and this group will produce a manuscript addressing considerations for data sharing for patient-oriented craniofacial research. This bioethics research proposal will add to the existing evidence base regarding participant preferences for data sharing in individuals with visible conditions.