Literature DB >> 33851745

Mechanisms of demyelination and neurodegeneration in globoid cell leukodystrophy.

M Laura Feltri1,2,3, Nadav I Weinstock1,2, Jacob Favret1,4, Narayan Dhimal1,2, Lawrence Wrabetz1,2,3, Daesung Shin1,4.   

Abstract

Globoid cell leukodystrophy (GLD), also known as Krabbe disease, is a lysosomal storage disorder causing extensive demyelination in the central and peripheral nervous systems. GLD is caused by loss-of-function mutations in the lysosomal hydrolase, galactosylceramidase (GALC), which catabolizes the myelin sphingolipid galactosylceramide. The pathophysiology of GLD is complex and reflects the expression of GALC in a number of glial and neural cell types in both the central and peripheral nervous systems (CNS and PNS), as well as leukocytes and kidney in the periphery. Over the years, GLD has garnered a wide range of scientific and medical interests, especially as a model system to study gene therapy and novel preclinical therapeutic approaches to treat the spontaneous murine model for GLD. Here, we review recent findings in the field of Krabbe disease, with particular emphasis on novel aspects of GALC physiology, GLD pathophysiology, and therapeutic strategies.
© 2021 Wiley Periodicals LLC.

Entities:  

Keywords:  Krabbe disease; demyelination; leukodystrophy; neurodegeneration

Mesh:

Substances:

Year:  2021        PMID: 33851745      PMCID: PMC8502241          DOI: 10.1002/glia.24008

Source DB:  PubMed          Journal:  Glia        ISSN: 0894-1491            Impact factor:   7.452


  242 in total

Review 1.  Mannose-6-phosphate pathway: a review on its role in lysosomal function and dysfunction.

Authors:  Maria Francisca Coutinho; Maria João Prata; Sandra Alves
Journal:  Mol Genet Metab       Date:  2011-12-23       Impact factor: 4.797

2.  Bone marrow transplantation increases efficacy of central nervous system-directed enzyme replacement therapy in the murine model of globoid cell leukodystrophy.

Authors:  Elizabeth Y Qin; Jacqueline A Hawkins-Salsbury; Xuntian Jiang; Adarsh S Reddy; Nuri B Farber; Daniel S Ory; Mark S Sands
Journal:  Mol Genet Metab       Date:  2012-06-01       Impact factor: 4.797

3.  Expression of immune-related molecules is downregulated in twitcher mice following bone marrow transplantation.

Authors:  Y P Wu; E J McMahon; J Matsuda; K Suzuki; G K Matsushima; K Suzuki
Journal:  J Neuropathol Exp Neurol       Date:  2001-11       Impact factor: 3.685

Review 4.  Lipid metabolism in myelinating glial cells: lessons from human inherited disorders and mouse models.

Authors:  Roman Chrast; Gesine Saher; Klaus-Armin Nave; Mark H G Verheijen
Journal:  J Lipid Res       Date:  2010-11-09       Impact factor: 5.922

5.  Neuropathology of twitcher mice: examination by histochemistry, immunohistochemistry, lectin histochemistry and Fourier transform infrared microspectroscopy.

Authors:  S M LeVine; D L Wetzel; A J Eilert
Journal:  Int J Dev Neurosci       Date:  1994-06       Impact factor: 2.457

6.  Retrovirus-mediated gene transfer and galactocerebrosidase uptake into twitcher glial cells results in appropriate localization and phenotype correction.

Authors:  A Luddi; M Volterrani; M Strazza; A Smorlesi; M A Rafi; J Datto; D A Wenger; E Costantino-Ceccarini
Journal:  Neurobiol Dis       Date:  2001-08       Impact factor: 5.996

7.  Role of extracellular calcium and mitochondrial oxygen species in psychosine-induced oligodendrocyte cell death.

Authors:  V Voccoli; I Tonazzini; G Signore; M Caleo; M Cecchini
Journal:  Cell Death Dis       Date:  2014-11-20       Impact factor: 8.469

8.  Fingolimod Rescues Demyelination in a Mouse Model of Krabbe's Disease.

Authors:  Sibylle Béchet; Sinead A O'Sullivan; Justin Yssel; Steven G Fagan; Kumlesh K Dev
Journal:  J Neurosci       Date:  2020-03-03       Impact factor: 6.167

9.  Can early treatment of twitcher mice with high dose AAVrh10-GALC eliminate the need for BMT?

Authors:  Mohammad A Rafi; Paola Luzi; David A Wenger
Journal:  Bioimpacts       Date:  2021-02-26

10.  Brainstem development requires galactosylceramidase and is critical for pathogenesis in a model of Krabbe disease.

Authors:  Nadav I Weinstock; Conlan Kreher; Jacob Favret; Duc Nguyen; Ernesto R Bongarzone; Lawrence Wrabetz; M Laura Feltri; Daesung Shin
Journal:  Nat Commun       Date:  2020-10-23       Impact factor: 14.919

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  2 in total

1.  Neurons contribute to pathology in a mouse model of Krabbe disease in a cell-autonomous manner.

Authors:  Pedro Brites; Monica M Sousa
Journal:  PLoS Biol       Date:  2022-07-06       Impact factor: 9.593

2.  Neuron-specific ablation of the Krabbe disease gene galactosylceramidase in mice results in neurodegeneration.

Authors:  Conlan Kreher; Jacob Favret; Nadav I Weinstock; Malabika Maulik; Xinying Hong; Michael H Gelb; Lawrence Wrabetz; M Laura Feltri; Daesung Shin
Journal:  PLoS Biol       Date:  2022-07-05       Impact factor: 9.593

  2 in total

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