Literature DB >> 33633367

Heterozygous COL9A3 variants cause severe peripheral vitreoretinal degeneration and retinal detachment.

Benjamin M Nash1,2,3, Christopher J G Watson1,2, Edward Hughes4,5, Alec L Hou6, To Ha Loi1, Bruce Bennetts2,3, Diana Jelovic1,2, Philip J Polkinghorne6,7, Mark Gorbatov4, John R Grigg1,4,8, Andrea L Vincent6,7, Robyn V Jamieson9,10,11.   

Abstract

The COL9A3 gene encodes one of the three alpha chains of Type IX collagen, with heterozygous variants reported to cause multiple epiphyseal dysplasia, and suggested as contributory in some cases of sensorineural hearing loss. Patients with homozygous variants have midface hypoplasia, myopia, sensorineural hearing loss, epiphyseal changes and carry a diagnosis of Stickler syndrome. Variants in COL9A3 have not previously been reported to cause vitreoretinal degeneration and/or retinal detachments. This report describes two families with autosomal dominant inheritance and predominant features of peripheral vitreoretinal lattice degeneration and retinal detachment. Genomic sequencing revealed a heterozygous splice variant in COL9A3 [NG_016353.1(NM_001853.4):c.1107 + 1G>C, NC_000020.10(NM_001853.4):c.1107 + 1G>C, LRG1253t1] in Family 1, and a heterozygous missense variant [NG_016353.1(NM_001853.4):c.388G>A p.(Gly130Ser)] in Family 2, each segregating with disease. cDNA studies of the splice variant demonstrated an in-frame deletion in the COL2 domain, and the missense variant occurred in the COL3 domain, both indicating the critical role of Type IX collagen in the vitreous base of the eye.

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Year:  2021        PMID: 33633367      PMCID: PMC8110976          DOI: 10.1038/s41431-021-00820-1

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   5.351


  20 in total

Review 1.  Defining the extracellular matrix using proteomics.

Authors:  Adam Byron; Jonathan D Humphries; Martin J Humphries
Journal:  Int J Exp Pathol       Date:  2013-02-19       Impact factor: 1.925

2.  COL9A3: A third locus for multiple epiphyseal dysplasia.

Authors:  P Paassilta; J Lohiniva; S Annunen; J Bonaventure; M Le Merrer; L Pai; L Ala-Kokko
Journal:  Am J Hum Genet       Date:  1999-04       Impact factor: 11.025

3.  Variation in the vitreous phenotype of Stickler syndrome can be caused by different amino acid substitutions in the X position of the type II collagen Gly-X-Y triple helix.

Authors:  A J Richards; D M Baguley; J R Yates; C Lane; M Nicol; P S Harper; J D Scott; M P Snead
Journal:  Am J Hum Genet       Date:  2000-09-25       Impact factor: 11.025

4.  Homozygous Type IX collagen variants (COL9A1, COL9A2, and COL9A3) causing recessive Stickler syndrome-Expanding the phenotype.

Authors:  Thomas R W Nixon; Philip Alexander; Allan Richards; Annie McNinch; Philip W P Bearcroft; Jan Cobben; Martin P Snead
Journal:  Am J Med Genet A       Date:  2019-05-14       Impact factor: 2.802

5.  Mutation analysis of COL9A3, a gene highly expressed in the cochlea, in hearing loss patients.

Authors:  Kenji Asamura; Satoko Abe; Hisakuni Fukuoka; Yusuke Nakamura; Shin-ichi Usami
Journal:  Auris Nasus Larynx       Date:  2005-03-24       Impact factor: 1.863

Review 6.  Autosomal recessive Stickler syndrome resulting from a COL9A3 mutation.

Authors:  Andrea Hanson-Kahn; Bing Li; Daniel H Cohn; Deborah A Nickerson; Michael J Bamshad; Louanne Hudgins
Journal:  Am J Med Genet A       Date:  2018-11-18       Impact factor: 2.802

7.  A mutation in the gene encoding the alpha 2 chain of the fibril-associated collagen IX, COL9A2, causes multiple epiphyseal dysplasia (EDM2).

Authors:  Y Muragaki; E C Mariman; S E van Beersum; M Perälä; J B van Mourik; M L Warman; B R Olsen; B C Hamel
Journal:  Nat Genet       Date:  1996-01       Impact factor: 38.330

Review 8.  Mutation Update for COL2A1 Gene Variants Associated with Type II Collagenopathies.

Authors:  Mouna Barat-Houari; Guillaume Sarrabay; Vincent Gatinois; Aurélie Fabre; Bruno Dumont; David Genevieve; Isabelle Touitou
Journal:  Hum Mutat       Date:  2015-10-21       Impact factor: 4.878

9.  A subcellular map of the human proteome.

Authors:  Peter J Thul; Lovisa Åkesson; Mikaela Wiking; Diana Mahdessian; Aikaterini Geladaki; Hammou Ait Blal; Tove Alm; Anna Asplund; Lars Björk; Lisa M Breckels; Anna Bäckström; Frida Danielsson; Linn Fagerberg; Jenny Fall; Laurent Gatto; Christian Gnann; Sophia Hober; Martin Hjelmare; Fredric Johansson; Sunjae Lee; Cecilia Lindskog; Jan Mulder; Claire M Mulvey; Peter Nilsson; Per Oksvold; Johan Rockberg; Rutger Schutten; Jochen M Schwenk; Åsa Sivertsson; Evelina Sjöstedt; Marie Skogs; Charlotte Stadler; Devin P Sullivan; Hanna Tegel; Casper Winsnes; Cheng Zhang; Martin Zwahlen; Adil Mardinoglu; Fredrik Pontén; Kalle von Feilitzen; Kathryn S Lilley; Mathias Uhlén; Emma Lundberg
Journal:  Science       Date:  2017-05-11       Impact factor: 47.728

10.  Targeted exon sequencing successfully discovers rare causative genes and clarifies the molecular epidemiology of Japanese deafness patients.

Authors:  Maiko Miyagawa; Takehiko Naito; Shin-ya Nishio; Naoyuki Kamatani; Shin-ichi Usami
Journal:  PLoS One       Date:  2013-08-13       Impact factor: 3.240

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  4 in total

1.  Identification of three novel homozygous variants in COL9A3 causing autosomal recessive Stickler syndrome.

Authors:  Aboulfazl Rad; Maryam Najafi; Fatemeh Suri; Soheila Abedini; Stephen Loum; Ehsan Ghayoor Karimiani; Narsis Daftarian; David Murphy; Mohammad Doosti; Afrooz Moghaddasi; Hamid Ahmadieh; Hamideh Sabbaghi; Mohsen Rajati; Narges Hashemi; Barbara Vona; Miriam Schmidts
Journal:  Orphanet J Rare Dis       Date:  2022-03-03       Impact factor: 4.123

2.  Whole Genome Sequencing, Focused Assays and Functional Studies Increasing Understanding in Cryptic Inherited Retinal Dystrophies.

Authors:  Benjamin M Nash; Alan Ma; Gladys Ho; Elizabeth Farnsworth; Andre E Minoche; Mark J Cowley; Christopher Barnett; Janine M Smith; To Ha Loi; Karen Wong; Luke St Heaps; Dale Wright; Marcel E Dinger; Bruce Bennetts; John R Grigg; Robyn V Jamieson
Journal:  Int J Mol Sci       Date:  2022-03-31       Impact factor: 5.923

Review 3.  Autosomal Recessive Stickler Syndrome.

Authors:  Thomas R W Nixon; Allan J Richards; Howard Martin; Philip Alexander; Martin P Snead
Journal:  Genes (Basel)       Date:  2022-06-24       Impact factor: 4.141

4.  Human iPSC-Derived Retinal Organoids and Retinal Pigment Epithelium for Novel Intronic RPGR Variant Assessment for Therapy Suitability.

Authors:  Fidelle Chahine Karam; To Ha Loi; Alan Ma; Benjamin M Nash; John R Grigg; Darshan Parekh; Lisa G Riley; Elizabeth Farnsworth; Bruce Bennetts; Anai Gonzalez-Cordero; Robyn V Jamieson
Journal:  J Pers Med       Date:  2022-03-21
  4 in total

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