How do fine and gross motor skills develop in preschool boys with Duchenne Muscular Dystrophy?

IF 2.9 2区医学 Q1 EDUCATION, SPECIAL Research in Developmental Disabilities Pub Date : 2024-09-27 DOI:10.1016/j.ridd.2024.104845

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Abstract

Background

Boys with Duchenne Muscular Dystrophy (DMD) experience both fine and gross motor problems. Nowadays, early intervention focuses almost exclusively on gross motor skills.

Aims

We aimed to explore early motor development in preschool boys with DMD and investigate the influence of cognition.

Methods and procedures

Seventeen boys with DMD (11 months- 6 years) were compared to typically developing (TD) peers and followed-up with the Bayley Scales of Infant and Toddler Development (Bayley-III); Peabody developmental motor scales (PDMS-II) and Motor Function Measure (MFM-20). The longitudinal evolution of fine and gross motor skills was investigated using linear mixed effect models (LMM). Cognition was added to the LMM as a covariate.

Outcomes and results

Preschool boys with DMD scored lower compared to TD peers on both fine and gross motor skills (p<0.001). The evolution of motor development was subscale-dependent. A significant influence of cognition was found on different subscales (p= 0.002–0.04).

Conclusions and implications

Preschool boys with DMD do not achieve the same functioning level as TD boys. Cognition plays a crucial role in the evolution of motor skills. Our results suggest a shift to a broader psychomotor approach including both fine and gross motor skills, also considering the impact of cognition.

What this paper adds?

Our study provides a detailed mapping of early fine and gross motor development in preschool boys with Duchenne Muscular Dystrophy (DMD) and describes the influence of cognition on both fine and gross motor skills. Preschool boys with DMD do not achieve the same functioning level compared to typically developing boys. They score significantly lower on both fine and gross motor skills. The evolution of fine and gross motor development was subscale-dependent e.g. a negative-positive evolution was seen for grasping skills, with a tipping point around the age of four; stationary scaled scores decreased followed by a stabilization around the age four to five and locomotion scaled scores remained stable over time. Finally, we also found that cognition plays a crucial role in the evolution of both fine and gross motor skills. These new insights in the evolution of early motor development could be of added value for future clinical trials in young boys with DMD. Subsequently, increased alertness to early symptoms, e.g. developmental delay, may advance the age of diagnosis, as well as associated early intervention.

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患有杜兴氏肌肉萎缩症的学龄前男孩的精细和粗大运动技能是如何发展的？

背景患有杜兴氏肌肉萎缩症（DMD）的男孩会出现精细和粗大运动问题。方法和程序将 17 名患有 DMD 的男孩（11 个月至 6 岁）与发育正常的同龄人（TD）进行比较，并使用贝利婴幼儿发育量表（Bayley-III）、皮博迪发育运动量表（PDMS-II）和运动功能测量（MFM-20）进行随访。采用线性混合效应模型（LMM）对精细和粗大运动技能的纵向发展进行了研究。结果DMD学龄前男孩的精细和粗大运动技能得分均低于TD同龄人（p<0.001）。运动发展的演变与分量表有关。结论和启示患有 DMD 的学龄前男孩无法达到与 TD 男孩相同的功能水平。认知在运动技能的发展过程中起着至关重要的作用。我们的研究详细描绘了患有杜兴氏肌肉营养不良症（DMD）的学龄前男孩的早期精细和粗大运动发育情况，并描述了认知对精细和粗大运动技能的影响。与发育正常的男孩相比，患有 DMD 的学龄前男孩无法达到相同的功能水平。他们在精细和粗大运动技能方面的得分都明显较低。精细和粗大运动能力的发展与分量表有关，例如，抓握能力的发展由负转正，在四岁左右达到临界点；静止能力的分量表得分下降，在四至五岁左右趋于稳定，而运动能力的分量表得分则随着时间的推移保持稳定。最后，我们还发现认知在精细和粗大运动技能的进化过程中起着至关重要的作用。这些对早期运动发育演变的新认识可能对未来针对 DMD 男孩的临床试验具有重要价值。随后，提高对早期症状（如发育迟缓）的警觉性可能会提高诊断年龄，并促进相关的早期干预。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Research in Developmental Disabilities Multiple-

CiteScore

5.50

自引率

6.50%

发文量

178

期刊介绍： Research In Developmental Disabilities is aimed at publishing original research of an interdisciplinary nature that has a direct bearing on the remediation of problems associated with developmental disabilities. Manuscripts will be solicited throughout the world. Articles will be primarily empirical studies, although an occasional position paper or review will be accepted. The aim of the journal will be to publish articles on all aspects of research with the developmentally disabled, with any methodologically sound approach being acceptable.