Department of Psychology, University of Wisconsin-Milwaukee, USA.
Division of Neuropsychology, Department of Neurology, Medical College of Wisconsin, USA.
J Pediatr Psychol. 2022 Feb 14;47(2):180-188. doi: 10.1093/jpepsy/jsab090.
Research indicates that children with neurofibromatosis type 1 (NF1) have weaknesses in fine and gross motor development in early childhood; however, little is known about the stability and developmental trajectory of motor functioning. We investigated (1) whether motor difficulties are evident and stable in the preschool period in children with NF1 and (2) whether there are particular patterns of motor development in this population.
Participants with NF1 and a control group of unaffected siblings were enrolled at ages 3-8 years and were assessed yearly. Motor functioning was assessed longitudinally using the Scales of Independent Behavior-Revised Motor Scale and the Differential Ability Scales-II Copying subtest. Wilcoxon sign tests were used to compare motor functioning at 3 or 4 years to 5 or 6 years old for children with NF1 seen during both time periods (N = 27). Linear mixed model growth curve analyses were used to compare trajectories for both children with NF1 (N = 62) and unaffected siblings (N = 37).
Children with NF1 made relative gains in raw scores, but not standard scores, across measures. Growth curve analyses revealed a significant effect of NF1 status on gross motor, fine motor, and copying scores, as well as an age by NF1 status effect on fine and gross motor scores.
Motor difficulties are evident early in life in children with NF1. Though children with NF1 clearly acquire motor skills over time, they continue to fall behind unaffected siblings, with the gap potentially widening over time. Further implications are discussed.
研究表明，患有神经纤维瘤病 1 型（NF1）的儿童在幼儿期精细运动和粗大运动发育均存在缺陷；然而，对于运动功能的稳定性和发展轨迹知之甚少。我们调查了（1）NF1 患儿在学龄前时期是否存在明显且稳定的运动困难，以及（2）该人群是否存在特定的运动发展模式。
NF1 患儿及其无影响同胞的对照组在 3-8 岁时入组，并每年进行评估。使用独立行为修订量表-运动量表和差异能力量表-II 临摹分量表对运动功能进行纵向评估。Wilcoxon 符号检验用于比较两个时间点（共 27 名患儿）均接受评估的 NF1 患儿 3 或 4 岁时与 5 或 6 岁时的运动功能。线性混合模型生长曲线分析用于比较 NF1 患儿（N=62）和无影响同胞（N=37）的运动轨迹。
NF1 患儿在各测量指标上的原始分数均有提高，但标准分数无变化。生长曲线分析显示 NF1 状态对粗大运动、精细运动和临摹得分有显著影响，年龄与 NF1 状态对精细和粗大运动得分有交互作用。
NF1 患儿在生命早期就存在运动困难。尽管 NF1 患儿随着时间的推移确实获得了运动技能，但他们仍落后于无影响的同胞，且差距可能随时间推移而扩大。进一步的影响将在讨论中阐述。
