ArticleNeurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology2025
Novel variant of COL12A1 gene causing neonatal hypotonia and respiratory failure.
Article in Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.
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2 citing papers in PubMed.
- Integrated Approach to Diagnosing Limb-Girdle Muscular Dystrophies in Resource-Limited Settings.Human mutation · 2026Article
- Myopathic Ehlers-Danlos Syndrome (mEDS) Related toInternational journal of molecular sciences · 2025Review
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9 authors.
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Abstract
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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