Centre for Neuroscience, Department of Biotechnology, Karpagam Academy of Higher Education (Deemed to be University), Coimbatore, Tamil Nadu, 641021, India.
Department of Human Genetics and Molecular Biology, Bharathiar University, Coimbatore, Tamil Nadu, 641 046, India.
Mol Neurobiol. 2024 Jul;61(7):4868-4878. doi: 10.1007/s12035-023-03882-y. Epub 2023 Dec 26.
Rett syndrome (RTT) is the rare neurodevelopmental disorder caused by mutations in methyl CpG binding protein 2 (MECP2) gene with a prevalence of 1:10,000 worldwide. The hallmark clinical features of RTT are developmental delay, microcephaly, repetitive behaviours, gait abnormalities, respiratory abnormalities and seizures. Still, the understanding on the diagnosis of RTT among clinicians are less. The aim of our work was to study various clinical manifestations and a spectrum of MECP2 genetic heterogeneity in RTT patients from South Indian population. We screened 208 autistic patients and diagnosed 20 RTT patients, who were further divided into classical RTT (group I; N = 11) and variant RTT (group II; N = 9). The clinical severity of RTT was measured using RSSS, RSBQ, SSI, SSS and RTT gross motor scale. The biochemical analysis showed that thyroid-stimulating hormone (TSH), plasma dopamine and cholesterol levels were higher in group I when compared to group II, whereas the level of blood pressure, calcium, ferritin and high-density lipoprotein levels were significantly decreased in both RTT groups, when compared to the control group. The genetic mutational spectrum of MECP2 mutations were found in 12/20 of RTT patients, which revealed the occurrence of 60% pathogenic mutation and 20% unknown mutation and it was correlated with the clinical finding of respiratory dysfunction, scoliosis and sleeping problems. The significant results of this study provided clinical and genetic aspects of RTT diagnosis and proposed the clinicians to screen abnormal cholesterol, calcium and TSH levels tailed with MECP2 gene mutations for early prognosis of disease severity.
雷特综合征(RTT)是一种罕见的神经发育障碍,由甲基 CpG 结合蛋白 2(MECP2)基因突变引起,全球患病率为 1:10000。RTT 的标志性临床特征是发育迟缓、小头畸形、重复行为、步态异常、呼吸异常和癫痫发作。然而,临床医生对 RTT 的诊断理解仍较少。我们的工作旨在研究来自印度南部人群的 RTT 患者的各种临床表现和 MECP2 基因异质性谱。我们筛选了 208 名自闭症患者,诊断出 20 名 RTT 患者,其中进一步分为经典 RTT(I 组;N = 11)和变异 RTT(II 组;N = 9)。使用 RSSS、RSBQ、SSI、SSS 和 RTT 粗大运动量表测量 RTT 的临床严重程度。生化分析显示,与 II 组相比,I 组的促甲状腺激素(TSH)、血浆多巴胺和胆固醇水平较高,而两组 RTT 的血压、钙、铁蛋白和高密度脂蛋白水平均显著降低,与对照组相比。在 20 名 RTT 患者中发现了 MECP2 突变的遗传突变谱,其中 60%的致病性突变和 20%的未知突变与呼吸功能障碍、脊柱侧凸和睡眠问题的临床发现相关。这项研究的重要结果提供了 RTT 诊断的临床和遗传方面,并提出临床医生筛选异常胆固醇、钙和 TSH 水平以及 MECP2 基因突变,以早期预测疾病严重程度。