Evidence map›Paper›PMID 39985652›Full record

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.

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

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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.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

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Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Article
  2. Myopathic Ehlers-Danlos Syndrome (mEDS) Related toInternational journal of molecular sciences · 2025
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4 · The record

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PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors.

Huizhi HuangDepartment of Neonatology, Anhui Medical University Children's Medical Center/Anhui Provincial Children's Hospital, Hefei, 230022, Anhui, China.
Luyao DengDepartment of Neonatology, Anhui Medical University Children's Medical Center/Anhui Provincial Children's Hospital, Hefei, 230022, Anhui, China.
Yu ZhangDepartment of Neonatology, Anhui Medical University Children's Medical Center/Anhui Provincial Children's Hospital, Hefei, 230022, Anhui, China.
Ying WangDepartment of Neonatology, Anhui Medical University Children's Medical Center/Anhui Provincial Children's Hospital, Hefei, 230022, Anhui, China.
Dongya YanDepartment of Neonatology, Anhui Medical University Children's Medical Center/Anhui Provincial Children's Hospital, Hefei, 230022, Anhui, China.
Xianhong LinDepartment of Neonatology, Anhui Medical University Children's Medical Center/Anhui Provincial Children's Hospital, Hefei, 230022, Anhui, China.
Yan WangDepartment of Neonatology, Anhui Medical University Children's Medical Center/Anhui Provincial Children's Hospital, Hefei, 230022, Anhui, China.
Yingyan WangDepartment of Pediatrics, Shannan People's Hospital, 16th Zedang Avenue, Shannan, 856000, Tibet, China. 4919521@qq.com.
Xiaofeng HuDepartment of Radiology, First Affiliated Hospital of University of Science and Technology of China, 17th Lujiang Road, Hefei, 230001, Anhui, China. ustc_huxiaofeng@163.com.ORCID http://orcid.org/0009-0008-6802-4806

Funding

Project of Shannan Science and Technology Bureau SNSBJKJJHXM2023014
6 · The paper itself

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.

Indexed as

Collagen Type XIIMuscle HypotoniaRespiratory InsufficiencyHumansInfant, NewbornCOL12A1 protein, humanCollagen Type XIIBethlem myopathyCOL12A1Congenital myopathyRespiratory failure

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.