Literature DB >> 23046582

Genotypic and phenotypic characterization of Brazilian patients with GM1 gangliosidosis.

Fernanda Sperb1, Filippo Vairo, Maira Burin, Fabiana Quoos Mayer, Ursula Matte, Roberto Giugliani.   

Abstract

GM1 gangliosidosis is a lysosomal disorder caused by β-galactosidase deficiency due to mutations in the GLB1 gene. It is a rare neurodegenerative disorder with an incidence of about 1:100,000-1:200,000 live births worldwide. Here we review GLB1 mutations and clinical features from 65 Brazilian GM1 gangliosidosis patients. Molecular analysis showed 17 different mutations and c.1622-1627insG was the most frequent, accounting for 50% of the alleles. Cognitive impairment was the main clinical sign, observed in 82% of patients, followed by hepatosplenomegaly observed in 56% of patients. It was possible to establish a significant correlation between age at onset of symptoms preceding the first year of life and the presence of the mutation c.1622-1627insG (p=0.03). Overall our findings differ from literature and represent the exclusive genotypic profile found in Brazilian GM1 gangliosidosis patients.
Copyright © 2012 Elsevier B.V. All rights reserved.

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Year:  2012        PMID: 23046582     DOI: 10.1016/j.gene.2012.09.106

Source DB:  PubMed          Journal:  Gene        ISSN: 0378-1119            Impact factor:   3.688


  7 in total

1.  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

2.  Clinical and molecular characteristics of 11 Chinese probands with GM1 gangliosidosis.

Authors:  Yuyu Feng; Yonglan Huang; Xiaoyuan Zhao; Huiying Sheng; Yi Feng; Wen Zhang; Li Liu
Journal:  Metab Brain Dis       Date:  2018-09-28       Impact factor: 3.584

Review 3.  The natural history of Type 1 infantile GM1 gangliosidosis: A literature-based meta-analysis.

Authors:  Frederick M Lang; Paul Korner; Mark Harnett; Ajith Karunakara; Cynthia J Tifft
Journal:  Mol Genet Metab       Date:  2019-12-30       Impact factor: 4.797

4.  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

5.  Intracerebroventricular enzyme replacement therapy with β-galactosidase reverses brain pathologies due to GM1 gangliosidosis in mice.

Authors:  Joseph C Chen; Amanda R Luu; Nathan Wise; Rolando De Angelis; Vishal Agrawal; Linley Mangini; Jon Vincelette; Britta Handyside; Harry Sterling; Melanie J Lo; Hio Wong; Nicole Galicia; Glenn Pacheco; Jeremy Van Vleet; Alexander Giaramita; Sylvia Fong; Sushmita M Roy; Chuck Hague; Roger Lawrence; Sherry Bullens; Terri M Christianson; Alessandra d'Azzo; Brett E Crawford; Stuart Bunting; Jonathan H LeBowitz; Gouri Yogalingam
Journal:  J Biol Chem       Date:  2019-09-03       Impact factor: 5.157

6.  Single Institutional Experience with GM1 Gangliosidosis: Clinical and Laboratory Results of 14 Patients

Authors:  Halil Tuna Akar; Yılmaz Yıldız; Gökhan Güvenkaya; Kısmet Çıkı; Ayça Burcu Kahraman; İzzet Erdal; Turgay Coşkun; Ali Dursun; Hatice Serap Sivri; Ayşegül Tokatlı
Journal:  Balkan Med J       Date:  2022-08-15       Impact factor: 3.570

7.  Clinical findings in Brazilian patients with adult GM1 gangliosidosis.

Authors:  Luciana Giugliani; Carlos Eduardo Steiner; Chong Ae Kim; Charles Marques Lourenço; Mara Lucia Schmitz Ferreira Santos; Carolina Fischinger Moura de Souza; Ana Carolina Brusius-Facchin; Guilherme Baldo; Mariluce Riegel; Roberto Giugliani
Journal:  JIMD Rep       Date:  2019-07-17
  7 in total

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