Literature DB >> 19277732

Neuronal ceroid lipofuscinosis caused by MFSD8 mutations: a common theme emerging.

M A Aldahmesh1, Z N Al-Hassnan, M Aldosari, F S Alkuraya.   

Abstract

Neuronal ceroid lipofuscinoses (NCLs) are a group of lysosomal neurodegenerative disorders that have in common the characteristic accumulation of abnormal storage material. Old clinical classification based on age of onset is now being revisited with the quickly accumulating knowledge of the various genetic defects that underlie this group of genetically heterogeneous disorders. We report our linkage data on a family with late-infantile NCL and show that the disease in this family is due to a homozygous novel mutation in the most recently described NCL gene (MFSD8). We use clinical data from our patients and the few others that have previously been reported to delineate the phenotype associated with mutations in this gene. We conclude that the phenotype is fairly consistent, which is a helpful guide to clinicians as they decide on the most cost-effective molecular testing strategies for NCLs.

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Year:  2009        PMID: 19277732     DOI: 10.1007/s10048-009-0185-1

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


  7 in total

1.  Transition-state structure as a unifying basis in protein-folding mechanisms: contact order, chain topology, stability, and the extended nucleus mechanism.

Authors:  A R Fersht
Journal:  Proc Natl Acad Sci U S A       Date:  2000-02-15       Impact factor: 11.205

2.  Neuronal ceroid lipofuscinoses (NCL).

Authors:  Sara E Mole
Journal:  Eur J Paediatr Neurol       Date:  2006-10-10       Impact factor: 3.140

Review 3.  Correlations between genotype, ultrastructural morphology and clinical phenotype in the neuronal ceroid lipofuscinoses.

Authors:  Sara E Mole; Ruth E Williams; Hans H Goebel
Journal:  Neurogenetics       Date:  2005-09-28       Impact factor: 2.660

Review 4.  The neuronal ceroid-lipofuscinoses: from past to present.

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

5.  A novel mutation in the MFSD8 gene in late infantile neuronal ceroid lipofuscinosis.

Authors:  E Stogmann; S El Tawil; J Wagenstaller; A Gaber; S Edris; A Abdelhady; E Assem-Hilger; F Leutmezer; S Bonelli; C Baumgartner; F Zimprich; T M Strom; A Zimprich
Journal:  Neurogenetics       Date:  2008-10-11       Impact factor: 2.660

6.  Homozygosity mapping: a way to map human recessive traits with the DNA of inbred children.

Authors:  E S Lander; D Botstein
Journal:  Science       Date:  1987-06-19       Impact factor: 47.728

7.  The novel neuronal ceroid lipofuscinosis gene MFSD8 encodes a putative lysosomal transporter.

Authors:  Eija Siintola; Meral Topcu; Nina Aula; Hannes Lohi; Berge A Minassian; Andrew D Paterson; Xiao-Qing Liu; Callum Wilson; Ulla Lahtinen; Anna-Kaisa Anttonen; Anna-Elina Lehesjoki
Journal:  Am J Hum Genet       Date:  2007-05-14       Impact factor: 11.025

  7 in total
  10 in total

Review 1.  Therapeutic approaches to the challenge of neuronal ceroid lipofuscinoses.

Authors:  R Kohan; I A Cismondi; A M Oller-Ramirez; N Guelbert; Tapia V Anzolini; G Alonso; S E Mole; Dodelson R de Kremer; Noher I de Halac
Journal:  Curr Pharm Biotechnol       Date:  2011-06       Impact factor: 2.837

2.  Expression and lysosomal targeting of CLN7, a major facilitator superfamily transporter associated with variant late-infantile neuronal ceroid lipofuscinosis.

Authors:  A Sharifi; M Kousi; C Sagné; G C Bellenchi; L Morel; M Darmon; H Hulková; R Ruivo; C Debacker; S El Mestikawy; M Elleder; A-E Lehesjoki; A Jalanko; B Gasnier; A Kyttälä
Journal:  Hum Mol Genet       Date:  2010-09-07       Impact factor: 6.150

3.  A rare homozygous MFSD8 single-base-pair deletion and frameshift in the whole genome sequence of a Chinese Crested dog with neuronal ceroid lipofuscinosis.

Authors:  Juyuan Guo; Dennis P O'Brien; Tendai Mhlanga-Mutangadura; Natasha J Olby; Jeremy F Taylor; Robert D Schnabel; Martin L Katz; Gary S Johnson
Journal:  BMC Vet Res       Date:  2015-01-03       Impact factor: 2.741

4.  Loss of CLN7 results in depletion of soluble lysosomal proteins and impaired mTOR reactivation.

Authors:  Tatyana Danyukova; Khandsuren Ariunbat; Melanie Thelen; Nahal Brocke-Ahmadinejad; Sara E Mole; Stephan Storch
Journal:  Hum Mol Genet       Date:  2018-05-15       Impact factor: 6.150

5.  Functional characterization of novel MFSD8 pathogenic variants anticipates neurological involvement in juvenile isolated maculopathy.

Authors:  Miriam Bauwens; Stephan Storch; Nicole Weisschuh; Chantal Ceuterick-de Groote; Riet De Rycke; Brecht Guillemyn; Sarah De Jaegere; Frauke Coppieters; Rudy Van Coster; Bart P Leroy; Elfride De Baere
Journal:  Clin Genet       Date:  2019-12-12       Impact factor: 4.438

6.  A Novel, Apparently Silent Variant in MFSD8 Causes Neuronal Ceroid Lipofuscinosis with Marked Intrafamilial Variability.

Authors:  Milda Reith; Lena Zeltner; Karin Schäferhoff; Dennis Witt; Theresia Zuleger; Tobias B Haack; Antje Bornemann; Michael Alber; Susanne Ruf; Ludger Schoels; Katarina Stingl; Nicole Weisschuh
Journal:  Int J Mol Sci       Date:  2022-02-18       Impact factor: 5.923

7.  AAV9/MFSD8 gene therapy is effective in preclinical models of neuronal ceroid lipofuscinosis type 7 disease.

Authors:  Xin Chen; Thomas Dong; Yuhui Hu; Frances C Shaffo; Nandkishore R Belur; Joseph R Mazzulli; Steven J Gray
Journal:  J Clin Invest       Date:  2022-03-01       Impact factor: 14.808

8.  Contribution of Whole-Genome Sequencing and Transcript Analysis to Decipher Retinal Diseases Associated with MFSD8 Variants.

Authors:  Anaïs F Poncet; Olivier Grunewald; Veronika Vaclavik; Isabelle Meunier; Isabelle Drumare; Valérie Pelletier; Béatrice Bocquet; Margarita G Todorova; Anne-Gaëlle Le Moing; Aurore Devos; Daniel F Schorderet; Florence Jobic; Sabine Defoort-Dhellemmes; Hélène Dollfus; Vasily M Smirnov; Claire-Marie Dhaenens
Journal:  Int J Mol Sci       Date:  2022-04-13       Impact factor: 6.208

9.  CLN7/MFSD8 may be an important factor for SARS-CoV-2 cell entry.

Authors:  Elena-Sofia Heinl; Sebastian Lorenz; Barbara Schmidt; Nouf Nasser M Laqtom; Joseph R Mazzulli; Laetitia Francelle; Timothy W Yu; Benjamin Greenberg; Stephan Storch; Ines Tegtmeier; Helga Othmen; Katja Maurer; Malin Steinfurth; Ralph Witzgall; Vladimir Milenkovic; Christian H Wetzel; Markus Reichold
Journal:  iScience       Date:  2022-09-06

10.  Pan-Cancer Analysis Reveals the Prognostic Potential of the THAP9/THAP9-AS1 Sense-Antisense Gene Pair in Human Cancers.

Authors:  Richa Rashmi; Sharmistha Majumdar
Journal:  Noncoding RNA       Date:  2022-07-08
  10 in total

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