Literature DB >> 23180398

CLN6 p.I154del mutation causing late infantile neuronal ceroid lipofuscinosis in a large consanguineous Moroccan family.

Ahmed Bouhouche1, Wafae Regragui, Elmostafa El Fahime, Naima Bouslam, Rachid Tazi-Ahnini, Marouane Melloul, Ali Benomar, Mohamed Yahyaoui.   

Abstract

The neuronal ceroid-lipofuscinosis (NCL) are a heterogeneous group of neurodegenerative diseases characterized by the lysosomal accumulation of ceroid and lipofuscin with mitochondrial ATP synthase subunit C in various tissues. Clinical features include progressive mental and motor deterioration, myoclonus, seizure, visual failure and premature death. Ten CLN genes have been identified, among them CLN6 genes for which 55 disease-causing mutations have already been reported. The authors describe here a large consanguineous Moroccan family with three affected patients due to the p.I154del mutation that has been exclusively reported in Portuguese patients. This is the first published report of a genetic study in a Moroccan family with NCL. A relatively inexpensive CLN6 mutation screening should be considered first in Morocco as an initial diagnosis step when the disease course is consistent with late infantile neuronal ceroid-lipofuscinosis.

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Year:  2012        PMID: 23180398     DOI: 10.1007/s12098-012-0889-3

Source DB:  PubMed          Journal:  Indian J Pediatr        ISSN: 0019-5456            Impact factor:   1.967


  9 in total

1.  Defective endoplasmic reticulum-resident membrane protein CLN6 affects lysosomal degradation of endocytosed arylsulfatase A.

Authors:  Claudia Heine; Bettina Koch; Stephan Storch; Alfried Kohlschütter; David N Palmer; Thomas Braulke
Journal:  J Biol Chem       Date:  2004-03-09       Impact factor: 5.157

2.  The gene mutated in variant late-infantile neuronal ceroid lipofuscinosis (CLN6) and in nclf mutant mice encodes a novel predicted transmembrane protein.

Authors:  Ruth B Wheeler; Julie D Sharp; Roger A Schultz; John M Joslin; Ruth E Williams; Sara E Mole
Journal:  Am J Hum Genet       Date:  2001-11-27       Impact factor: 11.025

Review 3.  The neuronal ceroid-lipofuscinoses: from past to present.

Authors:  Matti Haltia
Journal:  Biochim Biophys Acta       Date:  2006-07-08

4.  Kufs disease, the major adult form of neuronal ceroid lipofuscinosis, caused by mutations in CLN6.

Authors:  Todor Arsov; Katherine R Smith; John Damiano; Silvana Franceschetti; Laura Canafoglia; Catherine J Bromhead; Eva Andermann; Danya F Vears; Patrick Cossette; Sulekha Rajagopalan; Alan McDougall; Vito Sofia; Michael Farrell; Umberto Aguglia; Andrea Zini; Stefano Meletti; Michela Morbin; Saul Mullen; Frederick Andermann; Sara E Mole; Melanie Bahlo; Samuel F Berkovic
Journal:  Am J Hum Genet       Date:  2011-05-05       Impact factor: 11.025

5.  Autosomal dominant adult neuronal ceroid lipofuscinosis: a novel form of NCL with granular osmiophilic deposits without palmitoyl protein thioesterase 1 deficiency.

Authors:  Peter C G Nijssen; Chantal Ceuterick; Otto P van Diggelen; Milan Elleder; Jean-Jacques Martin; Johannes L J M Teepen; Jaana Tyynelä; Raymund A C Roos
Journal:  Brain Pathol       Date:  2003-10       Impact factor: 6.508

Review 6.  Interactions of the proteins of neuronal ceroid lipofuscinosis: clues to function.

Authors:  Amanda L Getty; David A Pearce
Journal:  Cell Mol Life Sci       Date:  2010-08-01       Impact factor: 9.207

Review 7.  Update of the mutation spectrum and clinical correlations of over 360 mutations in eight genes that underlie the neuronal ceroid lipofuscinoses.

Authors:  Maria Kousi; Anna-Elina Lehesjoki; Sara E Mole
Journal:  Hum Mutat       Date:  2011-11-16       Impact factor: 4.700

8.  Clinicopathological and molecular characterization of neuronal ceroid lipofuscinosis in the Portuguese population.

Authors:  Carla Teixeira; António Guimarães; Carlos Bessa; Maria José Ferreira; Lurdes Lopes; Eugénia Pinto; Rui Pinto; Rose-Mary Boustany; Maria Clara Sá Miranda; Maria Gil Ribeiro
Journal:  J Neurol       Date:  2003-06       Impact factor: 6.682

9.  Analysis of NCL Proteins from an Evolutionary Standpoint.

Authors:  Neda E Muzaffar; David A Pearce
Journal:  Curr Genomics       Date:  2008-04       Impact factor: 2.236

  9 in total
  3 in total

1.  A novel CLN2/TPP1 mutation in a patient with late infantile neuronal ceroid lipofuscinosis.

Authors:  Feng Yu; Xiao-Min Liu; Yin-He Chen; Sheng-Quan Zhang; Kai Wang
Journal:  Neurol Sci       Date:  2015-06-02       Impact factor: 3.307

2.  Electroclinical spectrum of the neuronal ceroid lipofuscinoses associated with CLN6 mutations.

Authors:  Laura Canafoglia; Isabella Gilioli; Federica Invernizzi; Vito Sofia; Valeria Fugnanesi; Michela Morbin; Luisa Chiapparini; Tiziana Granata; Simona Binelli; Vidmer Scaioli; Barbara Garavaglia; Nardo Nardocci; Samuel F Berkovic; Silvana Franceschetti
Journal:  Neurology       Date:  2015-06-26       Impact factor: 9.910

Review 3.  Myoclonus-Ataxia Syndromes: A Diagnostic Approach.

Authors:  Malco Rossi; Sterre van der Veen; Marcelo Merello; Marina A J Tijssen; Bart van de Warrenburg
Journal:  Mov Disord Clin Pract       Date:  2020-11-03
  3 in total

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