Literature DB >> 35066608

Gaucher disease - more than just a rare lipid storage disease.

Jaehyeok Roh1, Subbaya Subramanian2, Neal J Weinreb3, Reena V Kartha4.   

Abstract

Gaucher disease (GD), one of the most common lysosomal storage diseases, is caused by mutations in the gene, GBA1, that leads to defective glucocerebrosidase activity resulting in the accumulation and storage of glycosphingolipids. However, the pathophysiology of GD is more complicated leading to various associated conditions such as skeletal manifestations and Parkinson's disease (PD). These may result from oxidative stress and inflammatory responses due to complex interconnection of downstream factors such as substrate accumulation, endoplasmic reticulum (ER) stress, unfolded protein response (UPR), calcium dysregulation, mitochondrial dysfunction, defective autophagy, accumulation of α-synuclein aggregates, altered secretion and function of extracellular vesicles (EVs), and immunologic hyperactivity. Here we provide an overview of lysosomal storage diseases followed by a comprehensive review of the factors contributing to oxidative stress and inflammation in GD pathophysiology, mechanisms underlying the possible associated complications, current established treatments for GD, their limitations, and potential primary and adjunctive treatment options targeting these factors.
© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

Entities:  

Keywords:  Extracellular vesicles; Gaucher disease; Glycosphingolipids; Inflammation; Lysosomal storage disease; Oxidative stress

Mesh:

Substances:

Year:  2022        PMID: 35066608     DOI: 10.1007/s00109-021-02174-z

Source DB:  PubMed          Journal:  J Mol Med (Berl)        ISSN: 0946-2716            Impact factor:   4.599


  199 in total

1.  The Gaucher registry: demographics and disease characteristics of 1698 patients with Gaucher disease.

Authors:  J Charrow; H C Andersson; P Kaplan; E H Kolodny; P Mistry; G Pastores; B E Rosenbloom; C R Scott; R S Wappner; N J Weinreb; A Zimran
Journal:  Arch Intern Med       Date:  2000-10-09

Review 2.  The emergence of Parkinson disease among patients with Gaucher disease.

Authors:  Deborah Elstein; Roy Alcalay; Ari Zimran
Journal:  Best Pract Res Clin Endocrinol Metab       Date:  2014-08-23       Impact factor: 4.690

Review 3.  Gaucher disease paradigm: from ERAD to comorbidity.

Authors:  Inna Bendikov-Bar; Mia Horowitz
Journal:  Hum Mutat       Date:  2012-06-11       Impact factor: 4.878

4.  The lysosomal localization of sphingolipid hydrolases.

Authors:  N J Weinreb; R O Brady; A L Tappel
Journal:  Biochim Biophys Acta       Date:  1968-04-24

Review 5.  Gaucher disease and its treatment options.

Authors:  Lunawati L Bennett; Devipriya Mohan
Journal:  Ann Pharmacother       Date:  2013-09       Impact factor: 3.154

Review 6.  Lysosomal membrane proteins and their central role in physiology.

Authors:  Michael Schwake; Bernd Schröder; Paul Saftig
Journal:  Traffic       Date:  2013-03-06       Impact factor: 6.215

7.  Neuropathology provides clues to the pathophysiology of Gaucher disease.

Authors:  Kondi Wong; Ellen Sidransky; Ajay Verma; Tonghui Mixon; Glenn D Sandberg; Laura K Wakefield; Alan Morrison; Alicia Lwin; Carlos Colegial; John M Allman; Raphael Schiffmann
Journal:  Mol Genet Metab       Date:  2004-07       Impact factor: 4.797

8.  CNS, lung, and lymph node involvement in Gaucher disease type 3 after 11 years of therapy: clinical, histopathologic, and biochemical findings.

Authors:  Thomas A Burrow; Ying Sun; Carlos E Prada; Laurie Bailey; Wujuan Zhang; Amanda Brewer; Steve W Wu; Kenneth D R Setchell; David Witte; Mitchell B Cohen; Gregory A Grabowski
Journal:  Mol Genet Metab       Date:  2014-09-02       Impact factor: 4.797

Review 9.  The lysosome as a command-and-control center for cellular metabolism.

Authors:  Chun-Yan Lim; Roberto Zoncu
Journal:  J Cell Biol       Date:  2016-09-12       Impact factor: 10.539

10.  Identification of risk features for complication in Gaucher's disease patients: a machine learning analysis of the Spanish registry of Gaucher disease.

Authors:  Marcio M Andrade-Campos; Laura López de Frutos; Jorge J Cebolla; Irene Serrano-Gonzalo; Blanca Medrano-Engay; Mercedes Roca-Espiau; Beatriz Gomez-Barrera; Jorge Pérez-Heredia; David Iniguez; Pilar Giraldo
Journal:  Orphanet J Rare Dis       Date:  2020-09-22       Impact factor: 4.123

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