Laboratory of Genomic Medicine, Department of Genetics, National Rehabilitation Institute Luis Guillermo Ibarra Ibarra (INRLGII), Mexico City 14389, Mexico.
Rehabilitation and Special Education Center of Veracruz (CRISVER), Xalapa de Enriquez 91097, Mexico.
Int J Mol Sci. 2024 Oct 6;25(19):10750. doi: 10.3390/ijms251910750.
Spinocerebellar ataxia type 7 (SCA7) is a rare genetic disease characterized by progressive cerebellar syndrome and macular degeneration. In a previous study, we clinically and genetically characterized a group of Mexican patients, which represented one of the largest cohorts of SCA7 patients worldwide and demonstrated that all patients had a unique genetic origin. Our laboratory developed a program for the diagnosis, medical care, and long-term follow-up of these patients living in Veracruz State, and in this report, we present an update to this research, covering 2013 to 2024. So far, we identified 172 SCA7 carriers, with a few cases outside Veracruz, and our data support that the length of the CAG repeat tract mainly determines disease severity and life expectancy, and accordingly, we define three different phenotypes, early-onset (EO), classical-onset (CO), and late-onset (LO), with EO patients showing the lowest life expectancy. Furthermore, we found that parental transmission of mutant alleles leads to increased CAG repeat instability, compared to maternal ones. Interestingly, a haplotype analysis revealed that patients outside Veracruz may have different genetic origins. In conclusion, longitudinal observations of SCA7 patients provide insight into the natural history of SCA7 and help to design strategies for diagnosis, genetic counseling, physical rehabilitation, and therapeutic alternatives.
脊髓小脑共济失调 7 型(SCA7)是一种罕见的遗传性疾病,其特征为进行性小脑综合征和黄斑变性。在之前的研究中,我们对一组墨西哥患者进行了临床和基因特征分析,这代表了全球最大的 SCA7 患者队列之一,并表明所有患者均具有独特的遗传起源。我们的实验室为居住在韦拉克鲁斯州的这些患者制定了诊断、医疗和长期随访计划,在此报告中,我们对该研究进行了更新,涵盖了 2013 年至 2024 年的内容。迄今为止,我们已经鉴定了 172 名 SCA7 携带者,其中有少数病例来自韦拉克鲁斯州以外,我们的数据表明 CAG 重复序列的长度主要决定疾病的严重程度和预期寿命,因此,我们定义了三种不同的表型,早发性(EO)、经典性(CO)和晚发性(LO),EO 患者的预期寿命最低。此外,我们发现与母系相比,突变等位基因的亲代传递会导致 CAG 重复序列不稳定增加。有趣的是,单体型分析表明,韦拉克鲁斯州以外的患者可能具有不同的遗传起源。总之,对 SCA7 患者的纵向观察提供了对 SCA7 自然史的深入了解,并有助于设计诊断、遗传咨询、身体康复和治疗选择的策略。