Health and Psychosocial Outcomes in Young Children with Cleft Palate

项目摘要/摘要 口腔裂隙是美国最常见的先天性疾病之一，大约发生在 每800个新生儿中。唇裂伴或不伴唇裂的儿童(CP±L)需要从 婴儿期到成年期，以改善语言、面部外观以及身体和心理社会健康。裂隙- 相关治疗对受影响的儿童及其家人来说是负担，一生的医疗费用是 估计每年近7亿美元。儿童CP±L治疗指南主要是 以临床专业知识为基础，并在评估和干预的类型和时间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唇裂团队都包括一名 心理社会专业人员，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，目前还不清楚是哪一种 患有CP±L的儿童健康和心理社会结局不良的风险最大， (例如，种族)、医疗(例如，唇裂类型、诊断时机)和环境因素(例如，照顾者的压力) 加剧了这些风险。因此，只有有限的证据来提供及时和有效的信息 支持慢性阻塞性肺疾病±L患儿及其家属长期身心健康的干预措施。这 该项目将通过与腭裂登记/研究成果网络合作来解决这一差距 (Cornet；NIDCR R01DE02749)。Cornet是迄今为止美国最大的唇裂护理前瞻性研究 重点研究一组婴儿(N=3岁)的手术和语音结果(3岁前的腭前修复) 1514年)，Cp±L横跨16个地理上不同的裂隙中心。利用Cornet和跨学科 具有唇裂护理结果专业知识的调查团队，我们建议评估心理社会和健康 两项新研究对患有CP±L的幼儿及其照料者的结果：1)一项横断面研究 [n=500]预测健康和心理社会结果的医疗和人口因素；以及2)a 评估心理社会和健康的背景预测因素的纵向、混合方法研究[N=200] 随着时间的推移而产生的结果。具体目标包括：1)检查医疗和人口因素之间的联系 儿童的健康(生长/体重指数)和心理社会结果(与儿童健康有关的生活质量、适应) 2)前瞻性评估背景风险和保护措施的影响 随时间推移对健康和心理社会结果的影响因素(基线/前腭裂手术，24个月和36个月 3)定性地评估照料者对健康和心理社会结果的看法，以及 通过对40名照顾者进行叙述性采访，获得随时间推移的治疗经验。这项研究的数据将 阐明不良和最佳结果轨迹的早期预测因素，并告知类型和时机 干预措施，确保所有出生时患有CP±L的儿童实现最佳健康和幸福。

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.