Literature DB >> 16951682

Deficiency of hyccin, a newly identified membrane protein, causes hypomyelination and congenital cataract.

Federico Zara1, Roberta Biancheri, Claudio Bruno, Laura Bordo, Stefania Assereto, Elisabetta Gazzerro, Federica Sotgia, Xiao Bo Wang, Stefania Gianotti, Silvia Stringara, Marina Pedemonte, Graziella Uziel, Andrea Rossi, Angelo Schenone, Paolo Tortori-Donati, Marjo S van der Knaap, Michael P Lisanti, Carlo Minetti.   

Abstract

We describe a new autosomal recessive white matter disorder ('hypomyelination and congenital cataract') characterized by hypomyelination of the central and peripheral nervous system, progressive neurological impairment and congenital cataract. We identified mutations in five affected families, resulting in a deficiency of hyccin, a newly identified 521-amino acid membrane protein. Our study highlights the essential role of hyccin in central and peripheral myelination.

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Year:  2006        PMID: 16951682     DOI: 10.1038/ng1870

Source DB:  PubMed          Journal:  Nat Genet        ISSN: 1061-4036            Impact factor:   38.330


  27 in total

1.  Tremor-ataxia with central hypomyelination (TACH) leukodystrophy maps to chromosome 10q22.3-10q23.31.

Authors:  Geneviève Bernard; Isabelle Thiffault; Martine Tetreault; Maria Lisa Putorti; Isabelle Bouchard; Michel Sylvain; Serge Melançon; Rachel Laframboise; Pierre Langevin; Jean-Pierre Bouchard; Michel Vanasse; Adeline Vanderver; Guillaume Sébire; Bernard Brais
Journal:  Neurogenetics       Date:  2010-07-17       Impact factor: 2.660

2.  Novel infantile-onset leukoencephalopathy with high lactate level and slow improvement.

Authors:  Marjan E Steenweg; Adeline Vanderver; Berten Ceulemans; Prab Prabhakar; Luc Régal; Aviva Fattal-Valevski; Lawrence Richer; Barbara Goeggel Simonetti; Frederik Barkhof; Richard J T Rodenburg; Petra J W Pouwels; Marjo S van der Knaap
Journal:  Arch Neurol       Date:  2012-06

Review 3.  Systematic approaches to central nervous system myelin.

Authors:  Patricia de Monasterio-Schrader; Olaf Jahn; Stefan Tenzer; Sven P Wichert; Julia Patzig; Hauke B Werner
Journal:  Cell Mol Life Sci       Date:  2012-03-23       Impact factor: 9.261

Review 4.  A FOXG1 mutation in a boy with congenital variant of Rett syndrome.

Authors:  Tangui Le Guen; Nadia Bahi-Buisson; Juliette Nectoux; Nathalie Boddaert; Yann Fichou; Bertrand Diebold; Isabelle Desguerre; Florence Raqbi; Valérie Cormier Daire; Jamel Chelly; Thierry Bienvenu
Journal:  Neurogenetics       Date:  2010-08-24       Impact factor: 2.660

5.  Gene signatures of postoperative atrial fibrillation in atrial tissue after coronary artery bypass grafting surgery in patients receiving β-blockers.

Authors:  Miklos D Kertai; Wenjing Qi; Yi-Ju Li; Frederick W Lombard; Yutao Liu; Michael P Smith; Mark Stafford-Smith; Mark F Newman; Carmelo A Milano; Joseph P Mathew; Mihai V Podgoreanu
Journal:  J Mol Cell Cardiol       Date:  2016-02-06       Impact factor: 5.000

6.  Magnetic resonance imaging pattern recognition in hypomyelinating disorders.

Authors:  Marjan E Steenweg; Adeline Vanderver; Susan Blaser; Alberto Bizzi; Tom J de Koning; Grazia M S Mancini; Wessel N van Wieringen; Frederik Barkhof; Nicole I Wolf; Marjo S van der Knaap
Journal:  Brain       Date:  2010-10       Impact factor: 13.501

7.  Invited article: an MRI-based approach to the diagnosis of white matter disorders.

Authors:  Raphael Schiffmann; Marjo S van der Knaap
Journal:  Neurology       Date:  2009-02-24       Impact factor: 9.910

8.  Dysregulation of the Wnt pathway inhibits timely myelination and remyelination in the mammalian CNS.

Authors:  Stephen P J Fancy; Sergio E Baranzini; Chao Zhao; Dong-In Yuk; Karen-Amanda Irvine; Sovann Kaing; Nader Sanai; Robin J M Franklin; David H Rowitch
Journal:  Genes Dev       Date:  2009-06-10       Impact factor: 11.361

Review 9.  Genes involved in leukodystrophies: a glance at glial functions.

Authors:  Odile Boespflug-Tanguy; Pierre Labauge; Anne Fogli; Catherine Vaurs-Barriere
Journal:  Curr Neurol Neurosci Rep       Date:  2008-05       Impact factor: 5.081

10.  NUBPL mutations in patients with complex I deficiency and a distinct MRI pattern.

Authors:  Sietske H Kevelam; Richard J Rodenburg; Nicole I Wolf; Patrick Ferreira; Roelineke J Lunsing; Leo G Nijtmans; Anne Mitchell; Hugo A Arroyo; Dietz Rating; Adeline Vanderver; Carola G M van Berkel; Truus E M Abbink; Peter Heutink; Marjo S van der Knaap
Journal:  Neurology       Date:  2013-04-03       Impact factor: 9.910

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