CFM: Longitudinal Outcomes in Children pre-Kindergarten (CLOCK)

CFM：学前班儿童的纵向结果 (CLOCK)

• 批准号: 8221064
• 负责人: Carrie Lyn Heike
• 金额: $ 82.87万
• 依托单位: SEATTLE CHILDREN'S HOSPITAL
• 依托单位国家: 美国
• 财政年份: 2012
• 资助国家: 美国
• 起止时间: 2012-09-01 至 2017-08-31
• 项目状态: 已结题
• 来源: https://reporter.nih.gov/project-details/8221064
• 关键词: 3 year old; Address; Affect; Age; Anatomy; Area; Behavior assessment; Behavioral; Child; Child Care; Cohort Studies; Collaborations; Complex; Congenital neurologic anomalies; Cranial nerve diseases; Data; Deformity; Development; Diagnosis; Disease; Ear; Early treatment; Emotions; Enrollment; Etiology; Evaluation; External Ear; Face; Facial asymmetry; Facial nerve structure; Foundations; Future; Goals; Healthcare; Hearing; Individual; Infant; Infant Development; Intervention; Investigation; Labyrinth; Lead; Link; Longitudinal Studies; Mandible; Maps; Maxilla; Measures; Mediator of activation protein; Methods; Movement; Neuraxis; Outcome; Outcome Assessment; Participant; Pathway interactions; Patients; Performance; Production; Protocols documentation; Randomized Clinical Trials; Randomized Controlled Trials; Research; Research Design; Risk; School-Age Population; Schools; Series; Severities; Socialization; Speech; Structure; Testing; Time; Toddler; Tongue; Unilateral Hearing Loss; base; craniofacial; craniofacial microsomia; hearing impairment; improved; kindergarten; malformation; meetings; middle ear; neurobehavioral; neurodevelopment; neuropsychological; orofacial; social; soft tissue; standard measure; standard of care

DESCRIPTION (provided by applicant): Craniofacial microsomia (CFM) is a complex, congenital condition associated with underdevelopment of the facial structures. This condition is frequently associated with malformations of the outer, inner and middle ear, mandible, soft tissue, and facial nerve, resulting in varying degrees of facial asymmetry. As a result, children with CFM have elevated risk of hearing loss, impaired speech production, neurodevelopmental delays, poor academic and social outcomes, and behavioral maladjustment. Our prior studies in school-age children with CFM demonstrate that children with CFM do not perform as well on neuropsychological and social outcome assessments as children without craniofacial conditions. However, no studies have been performed in infants and toddlers and nor have the specific mechanisms contributing to these outcomes been investigated. The long term goals of our research consortium are to improve neurodevelopmental, social, and behavioral outcomes for children with CFM. The proposed research includes a longitudinal cohort study in children with and without CFM. The rationale underlying the research plan is that better understanding of the direct and indirect effects of CFM malformations on development will lead to the identification of more specific and effective early interventions. This proposal outlines a multi-center, longitudinal cohort study of 125 infants with CFM and 100 infants without craniofacial anomalies. Participants will undergo a series of evaluations between ages 0-3 years of age to comprehensively evaluate the developmental status of infants and toddlers with CFM. This research design will also explore specific pathways by which CFM may lead to certain outcomes. Specifically, the proposal explores (1) the longitudinal relations between facial asymmetry and emotion-related facial movements and poor socialization; and (2) associations among ear malformations, hearing and speech deficits and neurobehavioral outcomes. Results of this research will ultimately lead to future investigations that assess new interventions and corresponding changes in current standards of care for children with CFM. PUBLIC HEALTH RELEVANCE: We propose to develop a research collaboration with five craniofacial centers to enroll 125 children with craniofacial microsomia. We will perform a longitudinal study to investigate developmental outcomes in children with craniofacial microsomia during the first 3 years of age, and evaluate the relationship between these outcomes and the severity of craniofacial asymmetry, as well as hearing. The results of this study will enable further research on the etiology and health care outcomes in patients with craniofacial disorders.

描述（由申请人提供）：颅面矮小症（CFM）是一种复杂的先天性疾病，与面部结构发育不全有关。这种情况通常与外耳、内耳和中耳、下颌骨、软组织和面神经的畸形有关，导致不同程度的面部不对称。因此，患有CFM的儿童听力损失、言语产生受损、神经发育迟缓、学业和社会成绩差以及行为适应不良的风险增加。我们先前对学龄儿童CFM的研究表明，CFM儿童在神经心理和社会结果评估方面的表现不如无颅面条件的儿童。然而，尚未在婴儿和幼儿中进行研究，也没有研究导致这些结果的具体机制。我们研究联盟的长期目标是改善CFM儿童的神经发育，社会和行为结果。拟议的研究包括一项纵向队列研究，在儿童和没有CFM。研究计划的基本原理是，更好地了解CFM畸形对发育的直接和间接影响将导致确定更具体和有效的早期干预措施。该提案概述了一项多中心、纵向队列研究，纳入125名CFM婴儿和100名无颅面畸形婴儿。参与者将在0-3岁之间进行一系列评估，以全面评估患有CFM的婴幼儿的发育状况。本研究设计还将探索CFM可能导致某些结果的具体途径。具体而言，该提案探讨了（1）面部不对称和情感相关面部运动与不良社会化之间的纵向关系;以及（2）耳部畸形，听力和语言缺陷与神经行为结果之间的关联。这项研究的结果将最终导致未来的调查，评估新的干预措施和相应的变化，目前的标准照顾儿童CFM。 公共卫生相关性：我们建议与五个颅面中心合作，招募125名颅面矮小儿童。我们将进行一项纵向研究，调查颅面矮小儿童在前3岁的发育结果，并评估这些结果与颅面不对称的严重程度以及听力之间的关系。本研究的结果将使颅面疾病患者的病因学和医疗保健结果的进一步研究成为可能。