Literature DB >> 21332586

Clinical phenotypes associated with type II collagen mutations.

Peter Kannu1, John Bateman, Ravi Savarirayan.   

Abstract

COL2A1 mutations give rise to a spectrum of phenotypes predominantly affecting cartilage and bone from the severe disorders that are perinatally lethal to the milder conditions that are recognised in the post-natal period and childhood. The milder chondrodysplasias are characterised by disproportionate short stature, eye abnormalities, cleft palate and hearing loss. It remains poorly recognised that there is significant variability in the disease presentation, with early onset short stature conditions and later onset milder phenotypes. Similarly, it is under-acknowledged that COL2A1 mutations may solely cause joint disease in the absence of the other mentioned phenotypic clues. The underlying hypothesis is that there are novel phenotypes caused by mutations in type II collagen that extend from premature arthritis through to more severe bone dysplasias. The importance of finding a COL2A1 mutation lies in the subsequent ability to accurately assess recurrence risks and offer information regarding disease natural history. Most importantly, it enables at-risk individuals to be identified for implementation of preventative strategies and early ameliorative management of their condition. Such interventions potentially translate into a reduction in health costs associated with musculoskeletal disease.
© 2011 The Authors. Journal of Paediatrics and Child Health © 2011 Paediatrics and Child Health Division (Royal Australasian College of Physicians).

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Year:  2011        PMID: 21332586     DOI: 10.1111/j.1440-1754.2010.01979.x

Source DB:  PubMed          Journal:  J Paediatr Child Health        ISSN: 1034-4810            Impact factor:   1.954


  26 in total

Review 1.  The collagenopathies: review of clinical phenotypes and molecular correlations.

Authors:  Rebekah Jobling; Rohan D'Souza; Naomi Baker; Irene Lara-Corrales; Roberto Mendoza-Londono; Lucie Dupuis; Ravi Savarirayan; L Ala-Kokko; Peter Kannu
Journal:  Curr Rheumatol Rep       Date:  2014-01       Impact factor: 4.592

Review 2.  Novel COL2A1 mutations causing spondyloepiphyseal dysplasia congenita in three unrelated Chinese families.

Authors:  Limin Liu; QianQian Pang; Yan Jiang; Mei Li; Ou Wang; Weibo Xia
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Review 3.  Skeletal dysplasias.

Authors:  Deborah Krakow
Journal:  Clin Perinatol       Date:  2015-04-08       Impact factor: 3.430

4.  Types and effects of protein variations.

Authors:  Mauno Vihinen
Journal:  Hum Genet       Date:  2015-01-24       Impact factor: 4.132

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

Authors:  Εirini Dikaiakou; Εlpis A Vlachopapadopoulou; Emanouil Manolakos; Panagiotis Samelis; Rodanthi Margariti; Christos Zampakides; Stefanos Michalacos
Journal:  Mol Syndromol       Date:  2018-08-31

6.  Longitudinal Follow-Up of Two Patients with Dysspondyloenchondromatosis due to Novel Heterozygous Mutations in COL2A1.

Authors:  Nilay Güneş; Gözde Yeşil; Kubilay Beng; Sinan Kahraman; Beyhan Tüysüz
Journal:  Mol Syndromol       Date:  2018-04-24

7.  A pilot study of gene testing of genetic bone dysplasia using targeted next-generation sequencing.

Authors:  Huiwen Zhang; Rui Yang; Yu Wang; Jun Ye; Lianshu Han; Wenjuan Qiu; Xuefan Gu
Journal:  J Hum Genet       Date:  2015-09-17       Impact factor: 3.172

8.  Ophthalmic and molecular genetic findings in Kniest dysplasia.

Authors:  P I Sergouniotis; G S Fincham; A M McNinch; C Spickett; A V Poulson; A J Richards; M P Snead
Journal:  Eye (Lond)       Date:  2015-01-16       Impact factor: 3.775

9.  Stickler syndrome associated with epilepsy: report of three cases.

Authors:  Salvatore Savasta; Vincenzo Salpietro; Maria Valentina Spartà; Thomas Foiadelli; Daniela Laino; Lucio Lobefalo; Gian Luigi Marseglia; Alberto Verrotti
Journal:  Eur J Pediatr       Date:  2015-03-27       Impact factor: 3.183

10.  Identification of a Novel Mutation in the COL2A1 Gene in a Chinese Family with Spondyloepiphyseal Dysplasia Congenita.

Authors:  Xiangjun Huang; Xiong Deng; Hongbo Xu; Song Wu; Lamei Yuan; Zhijian Yang; Yan Yang; Hao Deng
Journal:  PLoS One       Date:  2015-06-01       Impact factor: 3.240

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