Craniofacial microsomia: Accelerating Research and Education (CARE)

颅面微小症：加速研究和教育 (CARE)

• 批准号: 10791256
• 负责人: Carrie Lyn Heike
• 金额: $ 34.4万
• 依托单位: SEATTLE CHILDREN'S HOSPITAL
• 依托单位国家: 美国
• 财政年份: 2023
• 资助国家: 美国
• 起止时间: 2023-09-11 至 2025-02-28
• 项目状态: 未结题
• 来源: https://reporter.nih.gov/project-details/10791256
• 关键词: Acceleration; Address; Advocacy; Affect; Benefits and Risks; Bioethics; Biomedical Research; Breathing; Caregivers; Cephalic; Child Care; Clinical; Clinical Research; Collaborations; Communities; Complex; Consent; Data; Data Collection; Development; Ear; Education; Ethics; Evaluation; Face; Funding; Future; Genetic; Grant; Guidelines; Health Personnel; Health Status; Healthcare; Hearing; Holistic Health; Individual; International; Intervention; Interview; Jaw; Knowledge; Language; Learning; Live Birth; Mandible; Manuscripts; Mental Health; Participant; Pathway interactions; Patients; Policies; Population; Privacy; Recommendation; Recording of previous events; Registries; Research; Research Personnel; Research Proposals; Resources; Risk; Structure; Surveys; Trust; United States National Institutes of Health; Work; care burden; care outcomes; clinical phenotype; craniofacial; craniofacial microsomia; data sharing; evidence base; experience; feeding; improved; malformation; member; microtia; neurodevelopment; nutrition; parent grant; patient oriented; phenotypic data; preference; psychologic; psychological distress; psychosocial; research study; stakeholder perspectives

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.

项目摘要/摘要 颅面微粒细胞（CFM）是第三大常见的先天性颅面状况，通常是 涉及面部结构不发达的，包括耳朵（例如Microtia）和下巴（例如，下颌 发育不全）。 CFM患者有复杂的医疗保健需求，经常需要协调 评估和干预措施以优化听力，神经发育，呼吸并最大程度地减少 颅外畸形。我们已经建立了一个受CFM影响的个人的国际社会， 医疗保健提供者和倡导领导者促进CFM的研究。我们目前正在进行大型 我们正在探索CFM个人的现场经验并探索国际研究 该人群的医疗保健和社会心理经历。我们正在从 英语和西班牙语的人在治疗途径中使用CFM及其护理人员。 2023年初，NIH引入了NIH资助研究的新数据共享要求。数据共享允许 研究人员最大程度地提高了现有数据的有用性来回答重要的研究问题。但是，我们 缺乏有关颅面社区的信息，内容涉及利益相关者关于共享临床的偏好 用于研究的表型数据。这是一个重要的问题，特别是对于稀有和可见的参与者而言 状况。从道德的角度来看，制定反映参与者生活的准则至关重要 经验和价值观。据我们所知，这尚未在具有颅面状况的个体中进行 用于共享表型数据。我们建议提高有关临床数据共享的知识 颅面研究通过解决以下特定目的：目标1。探索患者和护理人员 针对颅面研究的研究数据共享的观点。目标2。综合经验数据和道德 在面向患者的颅面研究中提出建议共享数据的规范。作为其中的一部分 提案，我们建立了由多个利益相关者组成的数据共享委员会的护理伦理， 该小组将产生一个手稿，以解决针对患者的数据共享的注意事项 颅面研究。该生物伦理学研究建议将增加有关的现有证据基础 参与者对具有可见条件的个体的数据共享的偏好。