Cavaloiu Bogdana, Simina Iulia-Elena, Vilciu Crisanda, Trăilă Iuliana-Anamaria, Puiu Maria
Faculty of Medicine, Department of Microscopic Morphology, Genetics Discipline, Center of Genomic Medicine, 'Victor Babes' University of Medicine and Pharmacy Timisoara, 2 E. Murgu, Sq., 300041 Timisoara, Romania.
Department of Radiology, 'Victor Gomoiu' Children's Clinical Hospital, 21 Basarabia Blvd., 022102 Bucharest, Romania.
Biomedicines. 2024 Aug 6;12(8):1782. doi: 10.3390/biomedicines12081782.
Spinal muscular atrophy (SMA) is a genetic disorder primarily caused by mutations in the SMN1 gene, leading to motor neuron degeneration and muscle atrophy, affecting multiple organ systems. Nusinersen treatment targets gene expression and is expected to enhance the motor function of voluntary muscles in the limbs and trunk. Motor skills can be assessed through specific scales like the Revised Upper Limb Module Scale (RULM) and Hammersmith Functional Motor Scale Expanded (HFMSE). This study aims to evaluate the influence of nusinersen on the motor skills of patients with SMA Type 2 and 3 using real-world data collected over 54 months. A prospective longitudinal study was conducted on 37 SMA patients treated with nusinersen, analyzing data with R statistical software. The outcomes revealed significant improvements in motor functions, particularly in SMA Type 3 patients with higher RULM and HFSME scores. Additionally, GEE analysis identified time, type, age, and exon deletions as essential predictors of motor score improvements. The extended observation period is both a major strength and a limitation of this research, as the dropout rates could present challenges in interpretation. Variability in responses, influenced by genetic background, SMA type, and onset age, highlights the need for personalized treatment approaches.
脊髓性肌萎缩症(SMA)是一种主要由SMN1基因突变引起的遗传性疾病,导致运动神经元退化和肌肉萎缩,影响多个器官系统。诺西那生治疗针对基因表达,有望增强四肢和躯干随意肌的运动功能。运动技能可通过特定量表进行评估,如修订上肢模块量表(RULM)和扩展哈默史密斯功能运动量表(HFMSE)。本研究旨在利用54个月收集的真实世界数据,评估诺西那生对2型和3型SMA患者运动技能的影响。对37例接受诺西那生治疗的SMA患者进行了一项前瞻性纵向研究,使用R统计软件分析数据。结果显示运动功能有显著改善,尤其是在RULM和HFSME评分较高的3型SMA患者中。此外,广义估计方程(GEE)分析确定时间、类型、年龄和外显子缺失是运动评分改善的重要预测因素。延长的观察期既是本研究的主要优势,也是局限性,因为失访率可能会给解释带来挑战。受遗传背景、SMA类型和发病年龄影响的反应变异性凸显了个性化治疗方法的必要性。