Congenital cytomegalovirus: Infant developmental trajectories and parent experiences

项目摘要 先天性巨细胞病毒（cCMV）婴儿出生的风险增加，长期后遗症，如 智力残疾、感音神经性听力损失和脑瘫。大多数先前的工作已经检查了 使用出生时疾病严重程度预测广泛定义的长期cCMV婴儿的发育 结果（例如“正常”与“异常”发展）在很大程度上忽略了环境或心理社会 这些影响可能会影响长期结果。一般来说，母亲的压力和较差的应对技能是两个 这些影响与高危婴儿的不良发育结果有关。检查 患有cCMV的婴儿的发育轨迹可能允许对cCMV的出现进行更细致的评价， 或发育迟缓的减轻，沿着相关的母亲经历。更好地了解 cCMV婴儿的发育、母亲的压力源、经历和感知的需求可能产生近端目标， 干预，如在其他条件下，以改善婴儿与cCMV的结果。检查cCMV婴儿 发展轨迹，他们与母亲的压力，幸福和养育经验的关系是 对建立针对具体疾病的预期指导、干预和支持系统至关重要， 婴儿的结果。因此，我们将招募并保留100个母婴配对的出生队列， 在婴儿期的前12个月内纵向研究婴儿发育，以实现以下目标： 确定cCMV婴儿的不同发育轨迹，并确定这些轨迹是否 与婴儿和母亲的基线特征相关;目的2）确定是否发育 轨迹成员与自我报告工具测量的母亲压力和福祉有关; 和目的3）通过序贯、重复和随机抽样的方法， 从出生到12个月大的非结构化定性访谈。研究PI，Pesch博士，是一个发育和 行为儿科医生，其最近的研究和临床工作集中在诊断和 治疗cCMV。在跨学科导师和顾问的支持下，Pesch博士将获得 使用虚拟平台测量婴儿发育的经验，招募和保留 罕见疾病队列研究，内部研究方法，以尽量减少偏见和家庭弹性理论。K23 支持的研究培训将增加对婴儿发育轨迹的理解， cCMV，以及这些轨迹与母亲压力和幸福感的关系。这些发现将导致 未来对cCMV婴儿的母亲进行母亲赋权干预，以改善婴儿 发展成果。

PROJECT SUMMARY Infants born with congenital cytomegalovirus (cCMV) are at heightened risk long-term sequelae such as intellectual disability, sensorineural hearing loss and cerebral palsy. Most prior work has examined development in infants with cCMV using disease severity at birth to predict broadly defined long-term outcomes (e.g. “normal” vs. “abnormal” development) largely ignoring environmental or psychosocial influences that may shape long-term outcomes. Maternal stress and poorer coping skills, in general, are two such influences that have been associated with poorer developmental outcomes in at-risk infants. Examining developmental trajectories in infants with cCMV may allow for a more nuanced appreciation of the emergence or lessening of developmental delays, along with correlating maternal experiences. A better understanding of cCMV infant development, mothers’ stressors, experiences and perceived needs may yield proximal targets for intervention, as in other conditions, to improve outcomes of infants with cCMV. Examining cCMV infant developmental trajectories, their relationship with maternal stress, well-being and parenting experiences are critical to creating disease-specific anticipatory guidance, interventions and support systems that may improve infant outcomes. Therefore we will recruit and retain a birth cohort of 100 mother-infant dyads, and measure infant development longitudinally across the first 12 months of infancy to address the following aims: Aim 1) To identify distinct trajectories of development in infants with cCMV, and to determine whether trajectories are associated with baseline characteristics of the infant, and mother; Aim 2) To determine whether developmental trajectory membership is associated with maternal stress and well-being measured by self-report instruments; and Aim 3) To examine mothers’ experiences of parenting an infant with cCMV through sequential, repeated unstructured qualitative interviews from birth to 12 months of age. Study PI, Dr. Pesch, is a developmental and behavioral pediatrician whose recent research and clinical work has centered around the diagnosis and treatment of cCMV. With the support of an interdisciplinary tem of mentors and an advisor, Dr. Pesch will gain experiences in the measurement of infant development using a virtual platform, the recruitment and retention of a rare disease cohort, insider research methods to minimize bias and family resilience theory. This K23 supported research training will increase the understanding of the developmental trajectories of infants with cCMV, and the relationships of those trajectories with maternal stress and well-being. These findings will lead to a future maternal empowerment intervention for mothers of infants with cCMV to improve infant developmental outcomes.