Key result
Pathogenic causative mutations were detected in 56.3% (9/16) of Chinese patients with nemaline myopathy, with NEB gene mutations being the most frequent and c.21522+3A>G identified as a hotspot.
Why the study?
What are the clinico-pathological features and mutational spectrum of nemaline myopathy in a Chinese cohort?
Observational (n=16)
No
What are the clinico-pathological features and mutational spectrum of nemaline myopathy in a Chinese cohort?
Answers are grounded in this paper and cite their sources.
NEB gene mutations are the most common cause of nemaline myopathy in this Chinese cohort, with c.21522 +3A > G identified as a hotspot mutation.
Nemaline myopathy (NM) is a congenital myopathy of great heterogeneity, characterized by the presence of rods in the cytoplasm of muscle fibers. The samples of 16 nemaline myopathy patients diagnosed by characteristically pathological features went through whole exon sequencing. Clinico-pathological and genetic features of the cases were systematically analyzed. According to the classification of nemaline myopathy by ENMC, 8 cases are typical congenital subtype, 6 cases are childhood/juvenile onset subtype and 2 case are adult onset subtype. In histological findings, characteristic purple-colored rods are discovered under modified gömöri trichrome staining (MGT). Electron microscopy revealed the presence of high electron-dense nemaline bodies around the submucosa and the nucleus nine patients (9/16 56.3%) were detected pathogenic causative mutations, among whom mutations in the NEB gene were the most frequent (6 patients, 66.7%). KBTBD13 gene mutation was discovered in two patients and ACTA1 gene mutation was discovered in 1 patient. Nemaline myopathy is a congenital myopathy with highly clinico-pathological and genetic heterogeneity. NEB gene mutation is the most common mutation, in which splicing change c.21522 +3A > G is hotspot mutation in Chinese NM patients.
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Yin et al. (2021) conducted an observational in Nemaline myopathy (n=16). Whole exome sequencing and clinico-pathological evaluation was evaluated on Detection of pathogenic causative mutations. Pathogenic causative mutations were detected in 56.3% (9/16) of Chinese patients with nemaline myopathy, with NEB gene mutations being the most frequent and c.21522+3A>G identified as a hotspot.
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