Abstract

Duchenne muscular dystrophy (DMD) is an X-linked recessive disease caused by gene mutations, leading to an absence of dystrophin protein isoforms in various tissues including the brain. A lack of dystrophin isoforms in the brain is hold responsible for the cognitive abnormalities in DMD. Especially, working memory problems and learning difficulties are frequently described in DMD, and these are known to have a major influence on academic achievement throughout childhood and adolescence. Previous research in the general population suggests that working memory problems and learning difficulties are trainable by memory programs. Positive and long-lasting results have been described particularly for the computerized Jungle memory program in children and adolescents with ADHD or learning difficulties. The current study aimed to assess the trainability of the dystrophin-associated working memory problems and learning difficulties in five male patients (age range = 10 – 16 years) with DMD, using the computerized Jungle memory program. A single case experimental design (SCED) with an A (non-intervention) B (intervention) A (non-intervention) withdrawal method was used. The study consisted of distinct phases. At baseline (week 0 to 3), patients underwent a short neuropsychological assessment and received psycho-education on working memory problems. Hereafter, the intervention program commenced based on randomization outcomes. At post-test (1 to 3 weeks after the intervention), and at 3 and 8 months follow-up patients repeated the baseline neuropsychological battery. Additionally, a weekly questionnaire on working memory problems was completed by parents throughout the baseline, intervention, and post-test period. Weekly administration outcomes of parents will be assessed by Tau-U analyses for SCED and outcomes of the neuropsychological assessments by reliable change index analyses (RCI). Preliminary results will be presented.

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