Novel variant of COL12A1 gene causing neonatal hypotonia and respiratory failure.

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Tác giả: Luyao Deng, Xiaofeng Hu, Huizhi Huang, Xianhong Lin, Yan Wang, Ying Wang, Yingyan Wang, Dongya Yan, Yu Zhang

Ngôn ngữ: eng

Ký hiệu phân loại:

Thông tin xuất bản: Italy : Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology , 2025

Mô tả vật lý:

Bộ sưu tập: NCBI

ID: 550229

 The COL12A1 gene encodes Collagen XII α 1 chain, forming a biologically functional Collagen XII through a trimeric structure. Collagen XII dysfunction can lead to hereditary neuromuscular diseases with phenotypic profiles ranging from rare Ullrich congenital muscular dystrophy 2 (biallelic variant) to Bethlem myopathy 2 (uniallelic variant). Currently, only 29 COL12A1 deficiency cases have been reported, mainly featuring hypotonia, progressive muscular dystrophy, and skeletal deformities. Newborns cases were relatively rare. Here, we describe a neonate with hypotonia, weak spontaneous movement, convulsions, and respiratory failure at birth. Identification of a novel variant of COL12A1 by whole exome sequencing and Sanger sequencing, NM_004370.6: c.7622 C >
  T, p.Ser2541Phe. Molecular dynamics simulation revealed its location in the thrombospondin N-terminal domain, potentially destabilizing the Collagen XII trimer's structure. Our case report expands the COL12A1 genotype and phenotype spectrum, suggesting the presence of neonatal hypotonia and highlighting the importance of vigilance for respiratory clinical manifestations.
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