Literature DB >> 16458719

Evidence of genetic heterogeneity in MRCS (microcornea, rod-cone dystrophy, cataract, and posterior staphyloma) syndrome.

Michel Michaelides1, Jill Urquhart, Graham E Holder, Marie Restori, Nuha Kayali, Forbes D C Manson, Graeme C M Black.   

Abstract

PURPOSE: To present the detailed phenotype of a subject with MRCS (microcornea, retinal dystrophy, cataract, and posterior staphyloma) syndrome and to investigate the underlying molecular genetic basis.
DESIGN: Interventional case report.
METHODS: Clinical examination, electrophysiologic assessment, B-scan ultrasonography, and mutation screening of the gene VMD2. The protocol of the study was approved by the local ethics committee and informed consent was obtained.
RESULTS: A 12-year-old boy was identified with bilateral microcornea, rod-cone dystrophy, congenital cataracts, and posterior staphylomata associated with high myopia (MRCS). Mutation screening failed to identify disease-causing sequence variants in VMD2, the gene associated with MRCS syndrome. All previous subjects have had pathogenic VMD2 sequence alterations.
CONCLUSIONS: We present a further report of the MRCS syndrome and provide evidence in support of genetic heterogeneity in this phenotype.

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Year:  2006        PMID: 16458719     DOI: 10.1016/j.ajo.2005.09.018

Source DB:  PubMed          Journal:  Am J Ophthalmol        ISSN: 0002-9394            Impact factor:   5.258


  5 in total

1.  Best's macular dystrophy in Australia: phenotypic profile and identification of novel BEST1 mutations.

Authors:  A C Cohn; C Turnbull; J B Ruddle; R H Guymer; L S Kearns; S Staffieri; H T Daggett; A W Hewitt; D A Mackey
Journal:  Eye (Lond)       Date:  2010-11-26       Impact factor: 3.775

2.  Ocular phenotypes associated with biallelic mutations in BEST1 in Italian patients.

Authors:  Andrea Sodi; Francesca Menchini; Maria Pia Manitto; Ilaria Passerini; Vittoria Murro; Francesca Torricelli; Ugo Menchini
Journal:  Mol Vis       Date:  2011-11-24       Impact factor: 2.367

3.  Clinical Heterogeneity in Autosomal Recessive Bestrophinopathy with Biallelic Mutations in the BEST1 Gene.

Authors:  Karsten Hufendiek; Katerina Hufendiek; Herbert Jägle; Heidi Stöhr; Marius Book; Georg Spital; Günay Rustambayova; Carsten Framme; Bernhard H F Weber; Agnes B Renner; Ulrich Kellner
Journal:  Int J Mol Sci       Date:  2020-12-08       Impact factor: 5.923

4.  BEST1 sequence variants in Italian patients with vitelliform macular dystrophy.

Authors:  Andrea Sodi; Ilaria Passerini; Vittoria Murro; Roberto Caputo; Giacomo Maria Bacci; Mirela Bodoj; Francesca Torricelli; Ugo Menchini
Journal:  Mol Vis       Date:  2012-11-17       Impact factor: 2.367

5.  Detailed analysis of family with autosomal recessive bestrophinopathy associated with new BEST1 mutation.

Authors:  Daiki Kubota; Kiyoko Gocho; Keiichiro Akeo; Sachiko Kikuchi; Michitaka Sugahara; Celso Soiti Matsumoto; Kei Shinoda; Atsushi Mizota; Kunihiko Yamaki; Hiroshi Takahashi; Shuhei Kameya
Journal:  Doc Ophthalmol       Date:  2016-04-12       Impact factor: 2.379

  5 in total

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