Kenepp Amanda, Russell-Giller Shira, Seehra Sonia, Fee Robert, Hinton Veronica J
Queens College, City University of New York, Flushing, NY, USA.
The Graduate Center, City University of New York, New York, NY, USA.
Child Neuropsychol. 2025 Apr;31(3):467-486. doi: 10.1080/09297049.2024.2386078. Epub 2024 Aug 1.
Duchenne muscular dystrophy (DMD) is an X-linked progressive neuromuscular disorder with a distinct cognitive profile including decreased verbal span. Children with DMD are also at risk for lower scores on academic achievement tests and increased behavioral problems. Longitudinal analyses generally reveal a stable intellectual profile, although attention and behavioral problems may negatively impact longitudinal IQ scores. To date, no study has reported on reading over time in DMD. Reading performance was assessed longitudinally in children with DMD, examining for potential contributions to the trajectory. Retrospective data analysis on assessments completed at baseline, year 2, and year 4 on 26 boys with DMD and 27 unaffected sibling controls (age at baseline: DMD 8 ± 1.4, controls 9 ± 2.6) indicated that children with DMD performed slightly, yet significantly, worse than controls on reading skills, but the longitudinal trajectory of reading skills for children with DMD and controls was not significantly different. Verbal span at time 1 was uniquely associated with later reading skills in children with DMD. Behavior was not associated with declines. The results confirm that children with DMD underperform on reading tasks and align with previous research suggesting that cognitive skills in DMD are stable over time.
杜氏肌营养不良症（DMD）是一种X连锁的进行性神经肌肉疾病，具有独特的认知特征，包括言语跨度降低。患有DMD的儿童在学业成绩测试中得分较低以及行为问题增加的风险也更高。纵向分析通常显示出稳定的智力特征，尽管注意力和行为问题可能会对纵向智商分数产生负面影响。迄今为止，尚无关于DMD患者随时间推移阅读情况的研究报告。对患有DMD的儿童的阅读表现进行了纵向评估，以检查其对阅读轨迹的潜在影响。对26名患有DMD的男孩和27名未受影响的同胞对照（基线年龄：DMD组8±1.4岁，对照组9±2.6岁）在基线、第2年和第4年完成的评估进行回顾性数据分析表明，患有DMD的儿童在阅读技能方面的表现略逊于对照组，但差异显著，不过患有DMD的儿童和对照组的阅读技能纵向轨迹并无显著差异。时间1时的言语跨度与患有DMD的儿童后期的阅读技能存在独特关联。行为与阅读技能下降无关。结果证实，患有DMD的儿童在阅读任务上表现较差，这与之前的研究结果一致，即DMD患者的认知技能随时间推移是稳定的。
