Genetic Instability and Disease Progression of Indian Rett Syndrome Patients

被引:0
|
作者
Gomathi, Mohan [1 ,2 ]
Dhivya, Venkatesan [1 ,2 ]
Padmavathi, Vijayakumar [3 ]
Pradeepkumar, Murugasamy [4 ]
Wilson, S. Robert [5 ]
Kumar, Nachimuthu Senthil [6 ]
Balachandar, Vellingiri [2 ,7 ]
机构
[1] Deemed be Univ, Karpagam Acad Higher Educ, Ctr Neurosci, Coimbatore 641021, Tamil Nadu, India
[2] Bharathiar Univ, Dept Human Genet & Mol Biol, Coimbatore 641046, Tamil Nadu, India
[3] Sacred Heart Coll Autonomous, Dept Microbiol, Tirupattur 635601, Tamil Nadu, India
[4] KMCH Inst Hlth Sci & Res, Dept Med Genet, Civil Aerodrome Rd, Coimbatore 641014, Tamil Nadu, India
[5] SRM Med Coll Hosp & Res Ctr, Kattankulathur 603203, Tamil Nadu, India
[6] Mizoram Univ, Cent Univ, Aizawl 796004, Mizoram, India
[7] Cent Univ Punjab, Sch Basic Sci, Dept Zool, Human Cytogenet & Stem Cell Lab, Bathinda 151401, Punjab, India
关键词
Rett syndrome; Social interactions; Methyl CpG binding protein 2; Lipid metabolism; Scoliosis; MECP2; MUTATIONS; PHENOTYPE; CHILDREN;
D O I
10.1007/s12035-023-03882-y
中图分类号
Q189 [神经科学];
学科分类号
071006 ;
摘要
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.
引用
收藏
页码:4868 / 4878
页数:11
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