Garg Shruti, Wan Ming Wai, Begum-Ali Jannath, Kolesnik-Taylor Anna, Green Jonathan, Johnson Mark H, Jones Emily
Division of Neuroscience and Experimental Psychology, Manchester Academic Health Science Centre, University of Manchester, Manchester, United Kingdom.
Royal Manchester Children's Hospital, Central Manchester University Hospitals NHS Foundation, Manchester, United Kingdom.
Front Psychol. 2022 Jul 22;13:795951. doi: 10.3389/fpsyg.2022.795951. eCollection 2022.
To examine the trajectories of cognitive, motor and behavioural development in infants with NF1 compared to infants without a family history of neurodevelopmental difficulties.
Infants with NF1 and low-risk controls were recruited from 5 months of age and followed longitudinally. Data from standardised tests was gathered at 5, 10 and 14 months and developmental trajectories of motor, language, behaviour, sleep, social development and parent-infant interaction were examined. Linear mixed modelling was used to estimate group differences in cognitive and behavioural measures over time.
No group differences were observed on Mullen Scale of Early Learning, overall adaptive functioning, temperament or behavioural measures. There were no group differences observed on measures of social communication or parent-infant interaction. Over the course of development, the NF1 group slept less and took more time to settle to sleep as compared to the control group. Maternal education was significantly associated with cognitive and behavioural developmental outcomes in both groups.
Cognitive, social and behavioural impairments are a cause of significant functional morbidity in children with NF1. This report is the first study to investigate the trajectories of cognitive, motor and behavioural development in infancy in NF1. Our results demonstrate that overall cognitive and behavioural developmental trajectories of the NF1 group in the infancy period are similar to controls. Given previous reports of delayed development in the NF1 cohort by 40 months, early clinical interventions strategies to promote sleep hygiene may be beneficial to optimise developmental outcomes.
与无神经发育困难家族史的婴儿相比,研究1型神经纤维瘤病(NF1)婴儿的认知、运动和行为发育轨迹。
招募5个月大的NF1婴儿和低风险对照婴儿,并进行纵向随访。在5个月、10个月和14个月时收集标准化测试数据,研究运动、语言、行为、睡眠、社会发展和亲子互动的发育轨迹。使用线性混合模型估计随时间推移认知和行为指标的组间差异。
在早期学习的穆伦量表、总体适应功能、气质或行为指标上未观察到组间差异。在社会沟通或亲子互动指标上也未观察到组间差异。在发育过程中,与对照组相比,NF1组睡眠较少,入睡时间较长。母亲的教育程度与两组的认知和行为发育结果均显著相关。
认知、社会和行为障碍是NF1儿童严重功能发病的原因。本报告是第一项研究NF1婴儿期认知、运动和行为发育轨迹的研究。我们的结果表明,NF1组婴儿期的总体认知和行为发育轨迹与对照组相似。鉴于之前有报告称NF1队列发育延迟40个月,促进睡眠卫生的早期临床干预策略可能有助于优化发育结果。