Institute of Biochemistry and Molecular Genetics, Faculty of Medicine, University of Ljubljana, Ljubljana, Slovenia.
Institute of Clinical Neurophysiology, University Medical Centre Ljubljana, Ljubljana, Slovenia.
Cell Mol Neurobiol. 2024 Oct 27;44(1):71. doi: 10.1007/s10571-024-01508-y.
The spinal muscular atrophy (SMA) phenotype strongly correlates with the SMN2 gene copy number. However, the severity and progression of the disease vary widely even among affected individuals with identical copy numbers. This study aimed to investigate the impact of genetic variability in oxidative stress, inflammatory, and neurodevelopmental pathways on SMA susceptibility and clinical progression. Genotyping for 31 genetic variants across 20 genes was conducted in 54 SMA patients and 163 healthy controls. Our results revealed associations between specific polymorphisms and SMA susceptibility, disease type, age at symptom onset, and motor and respiratory function. Notably, the TNF rs1800629 and BDNF rs6265 polymorphisms demonstrated a protective effect against SMA susceptibility, whereas the IL6 rs1800795 was associated with an increased risk. The polymorphisms CARD8 rs2043211 and BDNF rs6265 were associated with SMA type, while SOD2 rs4880, CAT rs1001179, and MIR146A rs2910164 were associated with age at onset of symptoms after adjustment for clinical parameters. In addition, GPX1 rs1050450 and HMOX1 rs2071747 were associated with motor function scores and lung function scores, while MIR146A rs2910164, NOTCH rs367398 SNPs, and GSTM1 deletion were associated with motor and upper limb function scores, and BDNF rs6265 was associated with lung function scores after adjustment. These findings emphasize the potential of genetic variability in oxidative stress, inflammatory processes, and neurodevelopmental pathways to elucidate the complex course of SMA. Further exploration of these pathways offers a promising avenue for developing personalized therapeutic strategies for SMA patients.
脊髓性肌萎缩症(SMA)表型与 SMN2 基因拷贝数密切相关。然而,即使在具有相同拷贝数的受影响个体中,疾病的严重程度和进展也存在很大差异。本研究旨在探讨氧化应激、炎症和神经发育途径中的遗传变异对 SMA 易感性和临床进展的影响。对 54 名 SMA 患者和 163 名健康对照者的 20 个基因中的 31 个基因变异进行了基因分型。我们的研究结果揭示了特定多态性与 SMA 易感性、疾病类型、症状出现年龄以及运动和呼吸功能之间的关联。值得注意的是,TNF rs1800629 和 BDNF rs6265 多态性对 SMA 易感性具有保护作用,而 IL6 rs1800795 与风险增加相关。CARD8 rs2043211 和 BDNF rs6265 多态性与 SMA 类型相关,而 SOD2 rs4880、CAT rs1001179 和 MIR146A rs2910164 与调整临床参数后的症状出现年龄相关。此外,GPX1 rs1050450 和 HMOX1 rs2071747 与运动功能评分和肺功能评分相关,而 MIR146A rs2910164、NOTCH rs367398 多态性、GSTM1 缺失与运动和上肢功能评分相关,BDNF rs6265 与肺功能评分相关。这些发现强调了氧化应激、炎症过程和神经发育途径中的遗传变异潜力,可以阐明 SMA 的复杂病程。进一步探索这些途径为 SMA 患者提供了开发个性化治疗策略的有希望的途径。