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

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.