Literature DB >> 26260654

Novel SIL1 mutations cause cerebellar ataxia and atrophy in a French-Canadian family.

Anne Noreau1,2, Roberta La Piana3,4, Camille Marcoux2, Patrick A Dion1,2,5, Bernard Brais1,3,5, Geneviève Bernard6, Guy A Rouleau7,8.   

Abstract

Two French-Canadian sibs with cerebellar ataxia and dysarthria were seen in our neurogenetics clinic. The older brother had global developmental delay and spastic paraplegia. Brain MRIs from these two affected individuals showed moderate to severe cerebellar atrophy. To identify the genetic basis for their disease, we conducted a whole exome sequencing (WES) investigation using genomic DNA prepared from the affected sibs and their healthy father. We identified two mutations in the SIL1 gene, which is reported to cause Marinesco-Sjögren syndrome. This study emphasizes how the diagnosis of patients with ataxic gait and cerebellar atrophy may benefit from WES to identify the genetic cause of their condition.

Entities:  

Keywords:  Ataxia; Cerebellar; Mutations; SIL1; Whole exome sequencing

Mesh:

Substances:

Year:  2015        PMID: 26260654     DOI: 10.1007/s10048-015-0455-z

Source DB:  PubMed          Journal:  Neurogenetics        ISSN: 1364-6745            Impact factor:   2.660


  11 in total

1.  Protein accumulation and neurodegeneration in the woozy mutant mouse is caused by disruption of SIL1, a cochaperone of BiP.

Authors:  Lihong Zhao; Chantal Longo-Guess; Belinda S Harris; Jeong-Woong Lee; Susan L Ackerman
Journal:  Nat Genet       Date:  2005-08-14       Impact factor: 38.330

2.  SIL1 mutations and clinical spectrum in patients with Marinesco-Sjogren syndrome.

Authors:  Michael Krieger; Andreas Roos; Claudia Stendel; Kristl G Claeys; Fatma Mujgan Sonmez; Michael Baudis; Peter Bauer; Antje Bornemann; Christian de Goede; Andreas Dufke; Richard S Finkel; Hans H Goebel; Martin Häussler; Helen Kingston; Janbernd Kirschner; Livija Medne; Petra Muschke; François Rivier; Sabine Rudnik-Schöneborn; Sabrina Spengler; Francesca Inzana; Franco Stanzial; Francesco Benedicenti; Matthis Synofzik; Ana Lia Taratuto; Laura Pirra; Stacey Kiat-Hong Tay; Haluk Topaloglu; Gökhan Uyanik; Dorothea Wand; Denise Williams; Klaus Zerres; Joachim Weis; Jan Senderek
Journal:  Brain       Date:  2013-10-30       Impact factor: 13.501

Review 3.  Diagnostic clinical genome and exome sequencing.

Authors:  Leslie G Biesecker; Robert C Green
Journal:  N Engl J Med       Date:  2014-06-19       Impact factor: 91.245

4.  EFNS/ENS Consensus on the diagnosis and management of chronic ataxias in adulthood.

Authors:  B P C van de Warrenburg; J van Gaalen; S Boesch; J-M Burgunder; A Dürr; P Giunti; T Klockgether; C Mariotti; M Pandolfo; O Riess
Journal:  Eur J Neurol       Date:  2014-01-13       Impact factor: 6.089

5.  The gene disrupted in Marinesco-Sjögren syndrome encodes SIL1, an HSPA5 cochaperone.

Authors:  Anna-Kaisa Anttonen; Ibrahim Mahjneh; Riikka H Hämäläinen; Clotilde Lagier-Tourenne; Outi Kopra; Laura Waris; Mikko Anttonen; Tarja Joensuu; Hannu Kalimo; Anders Paetau; Lisbeth Tranebjaerg; Denys Chaigne; Michel Koenig; Orvar Eeg-Olofsson; Bjarne Udd; Mirja Somer; Hannu Somer; Anna-Elina Lehesjoki
Journal:  Nat Genet       Date:  2005-11-13       Impact factor: 38.330

6.  Mutations in SIL1 cause Marinesco-Sjögren syndrome, a cerebellar ataxia with cataract and myopathy.

Authors:  Jan Senderek; Michael Krieger; Claudia Stendel; Carsten Bergmann; Markus Moser; Nico Breitbach-Faller; Sabine Rudnik-Schöneborn; Astrid Blaschek; Nicole I Wolf; Inga Harting; Kathryn North; Janine Smith; Francesco Muntoni; Martin Brockington; Susana Quijano-Roy; Francis Renault; Ralf Herrmann; Linda M Hendershot; J Michael Schröder; Hanns Lochmüller; Haluk Topaloglu; Thomas Voit; Joachim Weis; Friedrich Ebinger; Klaus Zerres
Journal:  Nat Genet       Date:  2005-11-13       Impact factor: 38.330

7.  Epidemiological, clinical, paraclinical and molecular study of a cohort of 102 patients affected with autosomal recessive progressive cerebellar ataxia from Alsace, Eastern France: implications for clinical management.

Authors:  M Anheim; M Fleury; B Monga; V Laugel; D Chaigne; G Rodier; E Ginglinger; C Boulay; S Courtois; N Drouot; M Fritsch; J P Delaunoy; D Stoppa-Lyonnet; C Tranchant; M Koenig
Journal:  Neurogenetics       Date:  2009-05-14       Impact factor: 2.660

8.  Marinesco-Sjögren syndrome due to SIL1 mutations with a comment on the clinical phenotype.

Authors:  M Horvers; A K Anttonen; A E Lehesjoki; E Morava; S Wortmann; S Vermeer; B P van de Warrenburg; M A Willemsen
Journal:  Eur J Paediatr Neurol       Date:  2012-10-11       Impact factor: 3.140

9.  Therapies for ataxias.

Authors:  Laurence Martineau; Anne Noreau; Nicolas Dupré
Journal:  Curr Treat Options Neurol       Date:  2014-07       Impact factor: 3.598

Review 10.  Hereditary ataxias: overview.

Authors:  Suman Jayadev; Thomas D Bird
Journal:  Genet Med       Date:  2013-03-28       Impact factor: 8.822

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  1 in total

1.  Sil1-Mutant Mice Elucidate Chaperone Function in Neurological Disorders.

Authors:  Stephan Buchkremer; José Andrés González Coraspe; Joachim Weis; Andreas Roos
Journal:  J Neuromuscul Dis       Date:  2016-05-27
  1 in total

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