Department of Medical Genetics, Faculty of Medicine and Dentistry, University of Alberta, Edmonton, AB T6G 2R3, Canada.
Department of Biological Sciences, Faculty of Science, University of Alberta, Edmonton, AB T6G 2R3, Canada.
Genes (Basel). 2023 Aug 17;14(8):1634. doi: 10.3390/genes14081634.
Spinal and bulbar muscular atrophy (SBMA), also known as Kennedy's disease, is a debilitating neuromuscular disease characterized by progressive muscular weakness and neuronal degeneration, affecting 1-2 individuals per 100,000 globally. While SBMA is relatively rare, recent studies have shown a significantly higher prevalence of the disease among the indigenous population of Western Canada compared to the general population. The disease is caused by a pathogenic expansion of polyglutamine residues in the androgen receptor protein, which acts as a key transcriptional regulator for numerous genes. SBMA has no cure, and current treatments are primarily supportive and focused on symptom management. Recently, a form of precision medicine known as antisense therapy has gained traction as a promising therapeutic option for numerous neuromuscular diseases. Antisense therapy uses small synthetic oligonucleotides to confer therapeutic benefit by acting on pathogenic mRNA molecules, serving to either degrade pathogenic mRNA transcripts or helping to modulate splicing. Recent studies have explored the suitability of antisense therapy for the treatment of SBMA, primarily focused on gene therapy and antisense-mediated mRNA knockdown approaches. Advancements in understanding the pathogenesis of SBMA and the development of targeted therapies offer hope for improved quality of life for individuals affected by this debilitating condition. Continued research is essential to optimize these genetic approaches, ensuring their safety and efficacy.
脊髓延髓肌萎缩症(SBMA),又称肯尼迪病,是一种进行性肌肉无力和神经元退化的衰弱性神经肌肉疾病,全球每 10 万人中有 1-2 人受其影响。尽管 SBMA 相对罕见,但最近的研究表明,与一般人群相比,加拿大西部的原住民群体中该病的发病率明显更高。该病是由雄激素受体蛋白中多聚谷氨酰胺残基的病理性扩增引起的,该蛋白是许多基因的关键转录调节剂。SBMA 目前尚无治愈方法,现有的治疗方法主要是支持性的,侧重于症状管理。最近,一种称为反义疗法的精准医学形式作为许多神经肌肉疾病的有前途的治疗选择引起了关注。反义疗法使用小的合成寡核苷酸通过作用于致病的 mRNA 分子来发挥治疗作用,从而降解致病的 mRNA 转录物或有助于调节剪接。最近的研究探讨了反义疗法治疗 SBMA 的适用性,主要集中在基因治疗和反义介导的 mRNA 敲低方法上。对 SBMA 发病机制的深入了解和靶向治疗的发展为改善受这种衰弱性疾病影响的个体的生活质量带来了希望。持续的研究对于优化这些遗传方法至关重要,以确保其安全性和有效性。