Literature DB >> 12205109

Vitreoretinopathy with phalangeal epiphyseal dysplasia, a type II collagenopathy resulting from a novel mutation in the C-propeptide region of the molecule.

A J Richards1, J Morgan, P W P Bearcroft, E Pickering, M J Owen, P Holmans, N Williams, C Tysoe, F M Pope, M P Snead, H Hughes.   

Abstract

A large family with dominantly inherited rhegmatogenous retinal detachment, premature arthropathy, and development of phalangeal epiphyseal dysplasia, resulting in brachydactyly was linked to COL2A1, the gene encoding proalpha1(II) collagen. Mutational analysis of the gene by exon sequencing identified a novel mutation in the C-propeptide region of the molecule. The glycine to aspartic acid change occurred in a region that is highly conserved in all fibrillar collagen molecules. The resulting phenotype does not fit easily into pre-existing subgroups of the type II collagenopathies, which includes spondyloepiphyseal dysplasia, and the Kniest, Strudwick, and Stickler dysplasias.

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Year:  2002        PMID: 12205109      PMCID: PMC1735224          DOI: 10.1136/jmg.39.9.661

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  8 in total

1.  The phenotypic spectrum in patients with arginine to cysteine mutations in the COL2A1 gene.

Authors:  K P Hoornaert; C Dewinter; I Vereecke; F A Beemer; W Courtens; A Fryer; H Fryssira; M Lees; A Müllner-Eidenböck; D L Rimoin; L Siderius; A Superti-Furga; K Temple; P J Willems; A Zankl; C Zweier; A De Paepe; P Coucke; G R Mortier
Journal:  J Med Genet       Date:  2005-09-09       Impact factor: 6.318

2.  Vitreous collagen metabolism before and after vitrectomy.

Authors:  Hirotaka Itakura; Shoji Kishi; Nobuo Kotajima; Masami Murakami
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2005-10-20       Impact factor: 3.117

3.  The heterozygous disproportionate micromelia (dmm) mouse: morphological changes in fetal cartilage precede postnatal dwarfism and compared with lethal homozygotes can explain the mild phenotype.

Authors:  Robert E Seegmiller; Brandon D Bomsta; Laura C Bridgewater; Cindy M Niederhauser; Carolina Montaño; Sterling Sudweeks; David R Eyre; Russell J Fernandes
Journal:  J Histochem Cytochem       Date:  2008-08-04       Impact factor: 2.479

4.  A novel mutation in the C-propeptide of COL2A1 causes atypical spondyloepiphyseal dysplasia congenita.

Authors:  Chieko Kusano; Masaki Takagi; Naoaki Hori; Jun Murotsuki; Gen Nishimura; Tomonobu Hasegawa
Journal:  Hum Genome Var       Date:  2017-03-02

5.  Expanding the clinical spectrum of COL2A1 related disorders by a mass like phenotype.

Authors:  Till Joscha Demal; Tasja Scholz; Maja Hempel; Georg Rosenberger; Helke Schüler; Jakob Olfe; Anja Fröhlich; Fabian Speth; Yskert von Kodolitsch; Thomas S Mir; Hermann Reichenspurner; Christian Kubisch
Journal:  Sci Rep       Date:  2022-03-16       Impact factor: 4.379

6.  Structural variations in articular cartilage matrix are associated with early-onset osteoarthritis in the spondyloepiphyseal dysplasia congenita (sedc) mouse.

Authors:  David W Macdonald; Ryan S Squires; Shaela A Avery; Jason Adams; Melissa Baker; Christopher R Cunningham; Nicholas B Heimann; David L Kooyman; Robert E Seegmiller
Journal:  Int J Mol Sci       Date:  2013-08-09       Impact factor: 5.923

7.  Short stature, platyspondyly, hip dysplasia, and retinal detachment: an atypical type II collagenopathy caused by a novel mutation in the C-propeptide region of COL2A1: a case report.

Authors:  Apiruk Sangsin; Chalurmpon Srichomthong; Monnat Pongpanich; Kanya Suphapeetiporn; Vorasuk Shotelersuk
Journal:  BMC Med Genet       Date:  2016-12-12       Impact factor: 2.103

8.  Phenotypic characterization of patients with early-onset high myopia due to mutations in COL2A1 or COL11A1: Why not Stickler syndrome?

Authors:  Lin Zhou; Xueshan Xiao; Shiqiang Li; Xiaoyun Jia; Panfeng Wang; Wenmin Sun; Fengsheng Zhang; Jiazhang Li; Tuo Li; Qingjiong Zhang
Journal:  Mol Vis       Date:  2018-08-10       Impact factor: 2.367

  8 in total

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