Literature DB >> 30733658

Identification of an Autosomal Dominant Mutation in the COL2A1 Gene Leading to Spondyloepiphyseal Dysplasia Congenita in a Greek Family.

Εirini Dikaiakou1, Εlpis A Vlachopapadopoulou1, Emanouil Manolakos2, Panagiotis Samelis3, Rodanthi Margariti3, Christos Zampakides3, Stefanos Michalacos1.   

Abstract

A boy and his father with severe short stature, progressively evolving body asymmetry, and skeletal abnormalities are presented. A next-generation sequencing exome study was performed, and the patient was found heterozygous for the c.1609G>A (p.Gly537Ser) mutation in the COL2A1 gene. This mutation is considered a pathogenic variant and has been previously registered in the Human Gene Mutation Database (HGMD) in association with spondyloepiphyseal dysplasia (accession: CM052184). It has been described in a patient as a sporadic case and resulted in a severe phenotype. Segregation studies, in order to determine the inheritance pattern, identified the same mutation in our patient's father. The variant was transmitted in an autosomal dominant pattern. In conclusion, we describe a patient with hereditary spondyloepiphyseal dysplasia congenita, caused by a c.1609G_A (p.Gly537Ser) mutation in the COL2A1 gene, which resulted in a milder phenotype.

Entities:  

Keywords:  Children; Short stature; Skeletal disorders; Spondyloepiphyseal dysplasia

Year:  2018        PMID: 30733658      PMCID: PMC6362925          DOI: 10.1159/000492190

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  18 in total

Review 1.  Collagens--structure, function, and biosynthesis.

Authors:  K Gelse; E Pöschl; T Aigner
Journal:  Adv Drug Deliv Rev       Date:  2003-11-28       Impact factor: 15.470

Review 2.  Clinical phenotypes associated with type II collagen mutations.

Authors:  Peter Kannu; John Bateman; Ravi Savarirayan
Journal:  J Paediatr Child Health       Date:  2011-02-18       Impact factor: 1.954

3.  Neurosurgical interventions for spondyloepiphyseal dysplasia congenita: clinical presentation and assessment of the literature.

Authors:  Anand Veeravagu; Shivanand P Lad; Joaquin Q Camara-Quintana; Bowen Jiang; Lawrence Shuer
Journal:  World Neurosurg       Date:  2012-01-25       Impact factor: 2.104

4.  Identification of three novel mutations in the COL2A1 gene in four unrelated Chinese families with spondyloepiphyseal dysplasia congenita.

Authors:  Zeng Zhang; Jin-Wei He; Wen-Zhen Fu; Chang-Qing Zhang; Zhen-Lin Zhang
Journal:  Biochem Biophys Res Commun       Date:  2011-09-06       Impact factor: 3.575

5.  The phenotypic spectrum of COL2A1 mutations.

Authors:  Gen Nishimura; Nobuhiko Haga; Hiroshi Kitoh; Yoko Tanaka; Toru Sonoda; Miho Kitamura; Shuya Shirahama; Taichi Itoh; Eiji Nakashima; Hirofumi Ohashi; Shiro Ikegawa
Journal:  Hum Mutat       Date:  2005-07       Impact factor: 4.878

6.  Spondyloepiphyseal dysplasia congenita with absent femoral head.

Authors:  Sung-Chul Jung; Santosh Mathew; Qi-Wei Li; Yong-Jun Lee; Kwang-Soo Lee; Hae-Ryong Song
Journal:  J Pediatr Orthop B       Date:  2004-03       Impact factor: 1.041

Review 7.  Skeletal dysplasia, growth hormone treatment and body proportion: comparison with other syndromic and non-syndromic short children.

Authors:  Lars Hagenäs; Thomas Hertel
Journal:  Horm Res       Date:  2003

8.  Efficacy of growth hormone therapy for patients with skeletal dysplasia.

Authors:  Hidemi Kanazawa; Hiroyuki Tanaka; Masaru Inoue; Yoshitaka Yamanaka; Noriyuki Namba; Yoshiki Seino
Journal:  J Bone Miner Metab       Date:  2003       Impact factor: 2.626

9.  A novel mutation in the COL2A1 gene in a Chinese family with Spondyloepiphyseal dysplasia congenita.

Authors:  Shiyin Li; Hao Zhou; Haitao Qin; Hong Guo; Yun Bai
Journal:  Joint Bone Spine       Date:  2013-08-09       Impact factor: 4.929

10.  Spondyloepiphyseal dysplasia congenita: genetic linkage to type II collagen (COL2AI).

Authors:  I J Anderson; R B Goldberg; R W Marion; W B Upholt; P Tsipouras
Journal:  Am J Hum Genet       Date:  1990-05       Impact factor: 11.025

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  2 in total

1.  Extreme Genu Recurvatum Deformity in a Pediatric Patient With Spondyloepiphyseal Dysplasia: Gradual Correction With Z-plates and Hexapod Frame.

Authors:  James Seymour; Neeraj Vij; Mohan Belthur
Journal:  Cureus       Date:  2022-05-23

2.  Familial Aggregation of a Novel Missense Variant of COL2A1 Gene Associated with Short Extremities: Case Report and Review of the Literature.

Authors:  Panagiotis Christopoulos; Anna Eleftheriades; George Paltoglou; Eleni Paschalidou; Emmanouil Kalampokas; Lina Florentin; Chrysanthi Billi; Makarios Eleftheriades
Journal:  Children (Basel)       Date:  2022-08-14
  2 in total

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