Literature DB >> 23622382

Lysosomal leukodystrophies: Krabbe disease and metachromatic leukodystrophy.

Alfried Kohlschütter1.   

Abstract

Genetic deficiencies of lysosomal catabolic pathways lead to storage disorders with multiple organ abnormalities or to degeneration of purely nervous structures. Krabbe disease and metachromatic leukodystrophy are caused by metabolic errors concerning lipids of neural membranes. They are characterized by demyelination of the central nervous system and, variably, the peripheral nerves. Their clinical presentation is a relentlessly progressive motor and mental deterioration starting at any age between infancy and adolescence. MRI demonstrates characteristic lesions of brain white matter. In Krabbe disease, deficient galactocerebroside β-galactosidase activity causes accumulation of lipids in "globoid" macrophages and of psychosine, which is toxic to oligodendrocytes. Diagnosis depends on demonstration of the enzyme deficiency. Experimental treatment is limited to hematopoietic stem cell transplantation, which can favorably alter the course of disease in certain situations. In metachromatic leukodystrophy, deficient activity of arylsulfatase A, or lack of a cofactor, causes accumulation of sulfatide in various tissues and diffuse demyelination. Symptoms are neurological, but gallbladder dysfunction may be present. Diagnosis depends on demonstrating the enzyme deficiency and elevated urinary sulfatide. In a rare variant, multiple sulfatases are deficient. Stem cell transplantation may prevent disease progression in selected cases. Enzyme replacement is being evaluated, and gene therapies are being developed.
Copyright © 2013 Elsevier B.V. All rights reserved.

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Year:  2013        PMID: 23622382     DOI: 10.1016/B978-0-444-59565-2.00029-0

Source DB:  PubMed          Journal:  Handb Clin Neurol        ISSN: 0072-9752


  15 in total

1.  Loss of the sphingolipid desaturase DEGS1 causes hypomyelinating leukodystrophy.

Authors:  Devesh C Pant; Imen Dorboz; Agatha Schluter; Stéphane Fourcade; Nathalie Launay; Javier Joya; Sergio Aguilera-Albesa; Maria Eugenia Yoldi; Carlos Casasnovas; Mary J Willis; Montserrat Ruiz; Dorothée Ville; Gaetan Lesca; Karine Siquier-Pernet; Isabelle Desguerre; Huifang Yan; Jingmin Wang; Margit Burmeister; Lauren Brady; Mark Tarnopolsky; Carles Cornet; Davide Rubbini; Javier Terriente; Kiely N James; Damir Musaev; Maha S Zaki; Marc C Patterson; Brendan C Lanpher; Eric W Klee; Filippo Pinto E Vairo; Elizabeth Wohler; Nara Lygia de M Sobreira; Julie S Cohen; Reza Maroofian; Hamid Galehdari; Neda Mazaheri; Gholamreza Shariati; Laurence Colleaux; Diana Rodriguez; Joseph G Gleeson; Cristina Pujades; Ali Fatemi; Odile Boespflug-Tanguy; Aurora Pujol
Journal:  J Clin Invest       Date:  2019-02-11       Impact factor: 14.808

Review 2.  Treatment for Krabbe's disease: Finding the combination.

Authors:  Christina R Mikulka; Mark S Sands
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

Review 3.  Astrocytes and lysosomal storage diseases.

Authors:  K V Rama Rao; T Kielian
Journal:  Neuroscience       Date:  2015-05-30       Impact factor: 3.590

4.  A HILIC-MS/MS method for simultaneous quantification of the lysosomal disease markers galactosylsphingosine and glucosylsphingosine in mouse serum.

Authors:  Rohini Sidhu; Christina R Mikulka; Hideji Fujiwara; Mark S Sands; Jean E Schaffer; Daniel S Ory; Xuntian Jiang
Journal:  Biomed Chromatogr       Date:  2018-04-26       Impact factor: 1.902

5.  Disease specific therapies in leukodystrophies and leukoencephalopathies.

Authors:  Guy Helman; Keith Van Haren; Joshua L Bonkowsky; Genevieve Bernard; Amy Pizzino; Nancy Braverman; Dean Suhr; Marc C Patterson; S Ali Fatemi; Jeff Leonard; Marjo S van der Knaap; Stephen A Back; Stephen Damiani; Steven A Goldman; Asako Takanohashi; Magdalena Petryniak; David Rowitch; Albee Messing; Lawrence Wrabetz; Raphael Schiffmann; Florian Eichler; Maria L Escolar; Adeline Vanderver
Journal:  Mol Genet Metab       Date:  2015-02-07       Impact factor: 4.797

Review 6.  Emerging treatments for pediatric leukodystrophies.

Authors:  Guy Helman; Keith Van Haren; Maria L Escolar; Adeline Vanderver
Journal:  Pediatr Clin North Am       Date:  2015-04-08       Impact factor: 3.278

7.  Metachromatic Leukodystrophy (MLD): a Pakistani Family with Novel ARSA Gene Mutation.

Authors:  Muhammad Aiman Shahzad; Saba Khaliq; Ali Amar; Saqib Mahmood
Journal:  J Mol Neurosci       Date:  2017-08-10       Impact factor: 3.444

8.  Two Cases of Female Chinese Adult-Onset Krabbe Disease with One Novel Mutation and a Review of Literature.

Authors:  Chengyi Zhang; Zheng Liu; Huiqing Dong
Journal:  J Mol Neurosci       Date:  2020-11-14       Impact factor: 3.444

9.  Lysosomal Re-acidification Prevents Lysosphingolipid-Induced Lysosomal Impairment and Cellular Toxicity.

Authors:  Christopher J Folts; Nicole Scott-Hewitt; Christoph Pröschel; Margot Mayer-Pröschel; Mark Noble
Journal:  PLoS Biol       Date:  2016-12-15       Impact factor: 8.029

10.  Pervasive supply of therapeutic lysosomal enzymes in the CNS of normal and Krabbe-affected non-human primates by intracerebral lentiviral gene therapy.

Authors:  Vasco Meneghini; Annalisa Lattanzi; Luigi Tiradani; Gabriele Bravo; Francesco Morena; Francesca Sanvito; Andrea Calabria; John Bringas; Jeanne M Fisher-Perkins; Jason P Dufour; Kate C Baker; Claudio Doglioni; Eugenio Montini; Bruce A Bunnell; Krystof Bankiewicz; Sabata Martino; Luigi Naldini; Angela Gritti
Journal:  EMBO Mol Med       Date:  2016-05-02       Impact factor: 12.137

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