Literature DB >> 18524657

GM1 gangliosidosis: review of clinical, molecular, and therapeutic aspects.

Nicola Brunetti-Pierri1, Fernando Scaglia.   

Abstract

GM(1) gangliosidosis is a lysosomal storage disorder due to deficiency of the beta-galactosidase enzyme. This deficiency results in accumulation of GM(1) gangliosides and related glycoconjugates in the lysosomes leading to lysosomal swelling, cellular damage, and organ dysfunction. The disease is lethal in the infantile and juvenile forms. To date, up to 102 mutations distributed along the beta-galactosidase gene (GLB1) have been reported. This review gives an overview of the clinical and molecular findings in patients with GM(1) gangliosidosis. Furthermore, it describes therapeutic approaches which are currently under investigation in animal models of the disease.

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Year:  2008        PMID: 18524657     DOI: 10.1016/j.ymgme.2008.04.012

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


  67 in total

Review 1.  Beyond the cherry-red spot: Ocular manifestations of sphingolipid-mediated neurodegenerative and inflammatory disorders.

Authors:  Hui Chen; Annie Y Chan; Donald U Stone; Nawajes A Mandal
Journal:  Surv Ophthalmol       Date:  2013-09-05       Impact factor: 6.048

2.  Carrier rates of four single-gene disorders in Croatian Bayash Roma.

Authors:  Ana Barešić; Marijana Peričić Salihović
Journal:  Genet Test Mol Biomarkers       Date:  2013-11-04

Review 3.  Pathophysiological actions of neuropathy-related anti-ganglioside antibodies at the neuromuscular junction.

Authors:  Jaap J Plomp; Hugh J Willison
Journal:  J Physiol       Date:  2009-06-29       Impact factor: 5.182

4.  β-Galactosidosis in Patient with Intermediate GM1 and MBD Phenotype.

Authors:  Tereza Moore; Jonathan A Bernstein; Sylvie Casson-Parkin; Tina M Cowan
Journal:  JIMD Rep       Date:  2012-04-22

5.  Clinical Findings and Natural History in Ten Unrelated Families with Juvenile and Adult GM1 Gangliosidosis.

Authors:  João Stein Kannebley; Laura Silveira-Moriyama; Laís Orrico Donnabella Bastos; Carlos Eduardo Steiner
Journal:  JIMD Rep       Date:  2015-06-25

Review 6.  The GM1 and GM2 Gangliosidoses: Natural History and Progress toward Therapy.

Authors:  Debra S Regier; Richard L Proia; Alessandra D'Azzo; Cynthia J Tifft
Journal:  Pediatr Endocrinol Rev       Date:  2016-06

7.  Rapid Identification of New Biomarkers for the Classification of GM1 Type 2 Gangliosidosis Using an Unbiased 1H NMR-Linked Metabolomics Strategy.

Authors:  Benita C Percival; Yvonne L Latour; Cynthia J Tifft; Martin Grootveld
Journal:  Cells       Date:  2021-03-05       Impact factor: 6.600

Review 8.  In Vivo NMR Studies of the Brain with Hereditary or Acquired Metabolic Disorders.

Authors:  Erica B Sherry; Phil Lee; In-Young Choi
Journal:  Neurochem Res       Date:  2015-11-26       Impact factor: 3.996

9.  Three novel beta-galactosidase gene mutations in Han Chinese patients with GM1 gangliosidosis are correlated with disease severity.

Authors:  Chi-Fan Yang; Jer-Yuarn Wu; Fuu-Jen Tsai
Journal:  J Biomed Sci       Date:  2010-09-30       Impact factor: 8.410

10.  A bicyclic 1-deoxygalactonojirimycin derivative as a novel pharmacological chaperone for GM1 gangliosidosis.

Authors:  Tomoko Takai; Katsumi Higaki; Matilde Aguilar-Moncayo; Teresa Mena-Barragán; Yuki Hirano; Kei Yura; Liang Yu; Haruaki Ninomiya; M Isabel García-Moreno; Yasubumi Sakakibara; Kousaku Ohno; Eiji Nanba; Carmen Ortiz Mellet; José M García Fernández; Yoshiyuki Suzuki
Journal:  Mol Ther       Date:  2013-01-22       Impact factor: 11.454

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