Literature DB >> 19454373

Biochemical and molecular findings in a patient with myoclonic epilepsy due to a mistarget of the beta-glucosidase enzyme.

Andrea Dardis1, Mirella Filocamo, Serena Grossi, Giovanni Ciana, Silvana Franceschetti, Silvia Dominissini, Guido Rubboli, Maya Di Rocco, Bruno Bembi.   

Abstract

A deficiency of human LIMP-2, a receptor for lysosomal mannose 6-phosphate-independent targeting of the beta-glucosidase (betaGC), due to mutations in the SCARB2 gene was described only in six families presented with progressive myoclonic epilepsy and nephrotic syndrome. In one of them a mistarget of the betaGC was demonstrated. We report here the biochemical and molecular findings in a patient diagnosed with progressive myoclonic epilepsy due to a mistarget of the betaGC, probably caused by a LIMP-2 deficiency, providing valuable information for the diagnosis of this rare disorder.

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Year:  2009        PMID: 19454373     DOI: 10.1016/j.ymgme.2009.04.011

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  10 in total

Review 1.  Lysosomal integral membrane protein-2: a new player in lysosome-related pathology.

Authors:  Ashley Gonzalez; Mark Valeiras; Ellen Sidransky; Nahid Tayebi
Journal:  Mol Genet Metab       Date:  2013-12-11       Impact factor: 4.797

2.  A mutation in SCARB2 is a modifier in Gaucher disease.

Authors:  Arash Velayati; John DePaolo; Nidhi Gupta; Jae H Choi; Nima Moaven; Wendy Westbroek; Ozlem Goker-Alpan; Ehud Goldin; Barbara K Stubblefield; Edwin Kolodny; Nahid Tayebi; Ellen Sidransky
Journal:  Hum Mutat       Date:  2011-09-15       Impact factor: 4.878

Review 3.  Gaucher disease: insights from a rare Mendelian disorder.

Authors:  Ellen Sidransky
Journal:  Discov Med       Date:  2012-10       Impact factor: 2.970

Review 4.  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

5.  Novel SCARB2 mutation in action myoclonus-renal failure syndrome and evaluation of SCARB2 mutations in isolated AMRF features.

Authors:  Franziska Hopfner; Barbara Schormair; Franziska Knauf; Achim Berthele; Thomas R Tölle; Ralf Baron; Christoph Maier; Rolf-Detlef Treede; Andreas Binder; Claudia Sommer; Christian Maihöfner; Wolfram Kunz; Friedrich Zimprich; Uwe Heemann; Arne Pfeufer; Michael Näbauer; Stefan Kääb; Barbara Nowak; Christian Gieger; Peter Lichtner; Claudia Trenkwalder; Konrad Oexle; Juliane Winkelmann
Journal:  BMC Neurol       Date:  2011-10-27       Impact factor: 2.474

6.  Identification of a Novel Homozygous Splice-Site Mutation in SCARB2 that Causes Progressive Myoclonus Epilepsy with or without Renal Failure.

Authors:  Jin He; Han Lin; Jin-Jing Li; Hui-Zhen Su; Dan-Ni Wang; Yu Lin; Ning Wang; Wan-Jin Chen
Journal:  Chin Med J (Engl)       Date:  2018-07-05       Impact factor: 2.628

7.  Genotype-Phenotype correlations of SCARB2 associated clinical presentation: a case report and in-depth literature review.

Authors:  Burcu Atasu; Ayse Nur Ozdag Acarlı; Basar Bilgic; Betül Baykan; Erol Demir; Yasemin Ozluk; Aydin Turkmen; Ann-Kathrin Hauser; Gamze Guven; Hasmet Hanagasi; Hakan Gurvit; Murat Emre; Thomas Gasser; Ebba Lohmann
Journal:  BMC Neurol       Date:  2022-03-28       Impact factor: 2.474

8.  Action myoclonus-renal failure syndrome: diagnostic applications of activity-based probes and lipid analysis.

Authors:  Paulo Gaspar; Wouter W Kallemeijn; Anneke Strijland; Saskia Scheij; Marco Van Eijk; Jan Aten; Herman S Overkleeft; Andrea Balreira; Friederike Zunke; Michael Schwake; Clara Sá Miranda; Johannes M F G Aerts
Journal:  J Lipid Res       Date:  2013-11-08       Impact factor: 5.922

9.  SCARB2 variants and glucocerebrosidase activity in Parkinson's disease.

Authors:  Roy N Alcalay; Oren A Levy; Pavlina Wolf; Petra Oliva; Xiaokui Kate Zhang; Cheryl H Waters; Stanley Fahn; Un Kang; Christopher Liong; Blair Ford; Pietro Mazzoni; Sheng Kuo; Amelie Johnson; Lan Xiong; Guy A Rouleau; Wendy Chung; Karen S Marder; Ziv Gan-Or
Journal:  NPJ Parkinsons Dis       Date:  2016-03-10

Review 10.  Genetic perspective on the synergistic connection between vesicular transport, lysosomal and mitochondrial pathways associated with Parkinson's disease pathogenesis.

Authors:  Stefanie Smolders; Christine Van Broeckhoven
Journal:  Acta Neuropathol Commun       Date:  2020-05-06       Impact factor: 7.801

  10 in total

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