Health and Psychosocial Outcomes in Young Children with Cleft Palate

项目概要/摘要 口面裂是美国最常见的先天性疾病之一，发生在大约一个 在每800个新生儿中腭裂伴或不伴唇裂（CP±L）的儿童需要跨学科护理， 从婴儿期到成年期，以改善语言，面部外观以及身体和心理健康。裂- 相关的治疗对受影响的儿童及其家庭来说是沉重的负担， 据估计，每年近7亿美元。儿童CP± L的治疗指南主要是 在评估和干预的类型和时机方面提供广泛的自由度。 尽管美国腭裂颅面协会建议所有的腭裂治疗团队都应该包括一个 心理社会专业人员，以解决心理健康，生活质量，情感，行为和社会需求 对于患有CP±L的儿童，不同中心的服务类型差异很大。此外，尚不清楚哪些 患有CP±L的儿童健康状况不佳和心理社会后果的风险最大，而哪些人口统计数据 (e.g.,种族），医疗（例如，裂缝类型、诊断时机）和环境因素（例如，照顾者压力） 助长了这些风险。因此，只有有限的证据表明， 干预措施，以支持CP±L儿童及其家庭的长期身心健康。这 该项目将通过与腭裂登记处/研究成果网络合作来解决这一差距 （CORNET; NIDCR R01 DE02749）。CORNET是迄今为止美国最大的唇腭裂护理前瞻性研究， 重点关注婴儿队列（N = 10）的手术和言语结果（腭修复前至3岁）。 1514）与CP±L在16个地理上不同的裂中心。利用CORNET和跨学科 一个在唇腭裂护理结果方面具有专业知识的调查团队，我们建议评估心理社会和健康状况， 通过两项新的研究，对CP±L幼儿及其照顾者的结局进行了研究：1）一项横断面研究 [N = 500]预测健康和心理社会结局的医学和人口统计学因素;以及2）a 纵向、混合方法研究[N = 200]，以评价心理社会和健康的背景预测因素 结果随着时间的推移。具体目标包括：1）检查医疗和人口因素之间的关联 和健康（生长/BMI）和心理社会结果（儿童健康相关的生活质量，调整） 年龄2-5岁入组CORNET; 2）前瞻性评估环境风险和保护性 随着时间的推移，健康和心理社会结果的因素（基线/腭手术前、24个月和36个月） 月龄）;和3）定性评估照顾者对健康和心理社会结果的看法， 治疗经验随着时间的推移，通过叙事访谈40照顾者。本研究的数据将 阐明不良和最佳结局轨迹的早期预测因素，并告知 干预措施，以确保所有出生时患有CP±L的儿童获得最佳的健康和福祉。

PROJECT SUMMARY/ABSTRACT Orofacial clefts are among the most common congenital conditions in the U.S., occurring in approximately one in every 800 births. Children with cleft palate with or without cleft lip (CP±L) require interdisciplinary care from infancy through adulthood to improve speech, facial appearance, and physical and psychosocial health. Cleft- related treatments are burdensome for affected children and their families, and lifetime healthcare costs are estimated at nearly $700 million U.S. dollars annually. Treatment guidelines for children CP± L are primarily based on clinical expertise and provide wide latitude in the types and timing of assessments and interventions. Although the American Cleft Palate-Craniofacial Association recommends that all cleft teams include a psychosocial professional to address the mental health, quality of life, emotional, behavioral, and social needs of children with CP±L, the types of services vary greatly across centers. In addition, it remains unknown which children with CP±L are at greatest risk for poor health and psychosocial outcomes and which demographic (e.g., race), medical (e.g., cleft type, timing of diagnosis) and contextual factors (e.g., caregiver stress) contribute to these risks. Consequently, there is limited evidence to inform the provision of timely and effective interventions to support the long-term mental and physical health of children with CP±L and their families. This project will address this gap by partnering with The Cleft Palate Registry/Research Outcomes Network (CORNET; NIDCR R01DE02749). CORNET is the largest U.S. prospective study in cleft care to date and focuses on surgical and speech outcomes (pre-palate repair through 3 years of age) in a cohort of infants (N = 1514) with CP±L across 16 geographically diverse cleft centers. Leveraging CORNET and an interdisciplinary team of investigators with expertise in cleft care outcomes, we propose to evaluate psychosocial and health outcomes in young children with CP±L and their caregivers through two new studies: 1) a cross-sectional study [N = 500] of medical and demographic factors predictive of health and psychosocial outcomes; and 2) a longitudinal, mixed methods study [N = 200] to evaluate contextual predictors of psychosocial and health outcomes over time. Specific aims include: 1) examine associations between medical and demographic factors and health (growth/BMI) and psychosocial outcomes (child health-related quality of life, adjustment) in children ages 2-5 years enrolled in CORNET; 2) prospectively assess the impact of contextual risk and protective factors on health and psychosocial outcomes over time (at baseline/pre-palate surgery, 24 months, and 36 months of age); and 3) qualitatively evaluate caregiver perceptions of health and psychosocial outcomes and treatment experiences over time through narrative interviews with 40 caregivers. Data from this study will elucidate early predictors of poor and optimal outcome trajectories and inform both the type and timing of interventions to ensure that all children born with CP±L achieve optimal health and well-being.