KU Leuven, Department of Rehabilitation Sciences, Research Group for Neurorehabilitation (eNRGy), Herestraat 49, Leuven 3000, Belgium; UHasselt, Faculty of Rehabilitation Sciences, Rehabilitation Research Centre (REVAL), Campus Diepenbeek, Agoralaan, Diepenbeek, Hasselt 3590, Belgium.
KU Leuven, Department of Rehabilitation Sciences, Research Group for Neurorehabilitation (eNRGy), Herestraat 49, Leuven 3000, Belgium.
Res Dev Disabil. 2024 Nov;154:104845. doi: 10.1016/j.ridd.2024.104845. Epub 2024 Sep 27.
Boys with Duchenne Muscular Dystrophy (DMD) experience both fine and gross motor problems. Nowadays, early intervention focuses almost exclusively on gross motor skills.
We aimed to explore early motor development in preschool boys with DMD and investigate the influence of cognition.
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.
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).
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.
患有杜氏肌营养不良症（DMD）的男孩会出现精细运动和粗大运动问题。如今，早期干预几乎完全集中在粗大运动技能上。
我们旨在探索学龄前患有 DMD 的男孩的早期运动发育情况，并研究认知的影响。
将 17 名患有 DMD 的男孩（11 个月至 6 岁）与典型发育（TD）的同龄人进行比较，并通过贝利婴幼儿发育量表（Bayley-III）、皮博迪发育运动量表（PDMS-II）和运动功能测量量表（MFM-20）进行随访。使用线性混合效应模型（LMM）来研究精细和粗大运动技能的纵向演变。将认知作为协变量添加到 LMM 中。
与 TD 同龄人相比，患有 DMD 的学龄前男孩在精细和粗大运动技能方面的得分均较低（p<0.001）。运动发育的演变取决于子量表。发现认知对不同子量表有显著影响（p=0.002-0.04）。
患有 DMD 的学龄前男孩的功能水平不如 TD 男孩。认知在运动技能的发展中起着至关重要的作用。我们的研究结果表明，需要转向更广泛的精神运动方法，包括精细和粗大运动技能，同时考虑认知的影响。
本研究详细描绘了学龄前患有杜氏肌营养不良症（DMD）男孩的精细和粗大运动早期发育情况，并描述了认知对精细和粗大运动技能的影响。与 TD 男孩相比，患有 DMD 的学龄前男孩的功能水平不同。他们在精细和粗大运动技能方面的得分明显较低。精细和粗大运动发育的演变取决于子量表，例如抓握技能的演变是负正，转折点在 4 岁左右；静止量表评分下降，随后在 4 到 5 岁左右稳定，运动量表评分随时间保持稳定。最后，我们还发现认知在精细和粗大运动技能的发展中起着至关重要的作用。这些关于早期运动发育演变的新见解对于未来患有 DMD 的年轻男孩的临床试验可能具有附加价值。因此，对发育迟缓等早期症状的早期警觉性可能会提前诊断年龄，以及相关的早期干预。
