The Role of Sphingomyelin and Ceramide in Motor Neuron Diseases.
Amyotrophic Lateral Sclerosis
ceramide
motor neuron disease
sphingolipid
sphingomyelin
Journal
Journal of personalized medicine
ISSN: 2075-4426
Titre abrégé: J Pers Med
Pays: Switzerland
ID NLM: 101602269
Informations de publication
Date de publication:
30 Aug 2022
30 Aug 2022
Historique:
received:
08
08
2022
revised:
26
08
2022
accepted:
27
08
2022
entrez:
23
9
2022
pubmed:
24
9
2022
medline:
24
9
2022
Statut:
epublish
Résumé
Amyotrophic Lateral Sclerosis (ALS), Spinal Bulbar Muscular Atrophy (SBMA), and Spinal Muscular Atrophy (SMA) are motor neuron diseases (MNDs) characterised by progressive motor neuron degeneration, weakness and muscular atrophy. Lipid dysregulation is well recognised in each of these conditions and occurs prior to neurodegeneration. Several lipid markers have been shown to predict prognosis in ALS. Sphingolipids are complex lipids enriched in the central nervous system and are integral to key cellular functions including membrane stability and signalling pathways, as well as being mediators of neuroinflammation and neurodegeneration. This review highlights the metabolism of sphingomyelin (SM), the most abundant sphingolipid, and of its metabolite ceramide, and its role in the pathophysiology of neurodegeneration, focusing on MNDs. We also review published lipidomic studies in MNDs. In the 13 studies of patients with ALS, 12 demonstrated upregulation of multiple SM species and 6 demonstrated upregulation of ceramides. SM species also correlated with markers of clinical progression in five of six studies. These data highlight the potential use of SM and ceramide as biomarkers in ALS. Finally, we review potential therapeutic strategies for targeting sphingolipid metabolism in neurodegeneration.
Identifiants
pubmed: 36143200
pii: jpm12091418
doi: 10.3390/jpm12091418
pmc: PMC9501626
pii:
doi:
Types de publication
Journal Article
Review
Langues
eng
Subventions
Organisme : Association of British Neurologists
ID : Clinical Research Fellowship
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