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Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature

Multiple system atrophy (MSA) and progressive supranuclear palsy (PSP) are uncommon multifactorial atypical Parkinsonian syndromes, expressed by various clinical features. MSA and PSP are commonly considered sporadic neurodegenerative disorders; however, our understanding is improving of their genet...

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Autor principal: Bougea, Anastasia
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10048872/
https://www.ncbi.nlm.nih.gov/pubmed/36982356
http://dx.doi.org/10.3390/ijms24065281
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author Bougea, Anastasia
author_facet Bougea, Anastasia
author_sort Bougea, Anastasia
collection PubMed
description Multiple system atrophy (MSA) and progressive supranuclear palsy (PSP) are uncommon multifactorial atypical Parkinsonian syndromes, expressed by various clinical features. MSA and PSP are commonly considered sporadic neurodegenerative disorders; however, our understanding is improving of their genetic framework. The purpose of this study was to critically review the genetics of MSA and PSP and their involvement in the pathogenesis. A systemized literature search of PubMed and MEDLINE was performed up to 1 January 2023. Narrative synthesis of the results was undertaken. In total, 43 studies were analyzed. Although familial MSA cases have been reported, the hereditary nature could not be demonstrated. COQ2 mutations were involved in familial and sporadic MSA, without being reproduced in various clinical populations. In terms of the genetics of the cohort, synuclein alpha (SNCA) polymorphisms were correlated with an elevated likelihood of manifesting MSA in Caucasians, but a causal effect relationship could not be demonstrated. Fifteen MAPT mutations were linked with PSP. Leucine-rich repeat kinase 2 (LRRK2) is an infrequent monogenic mutation of PSP. Dynactin subunit 1 (DCTN1) mutations may imitate the PSP phenotype. GWAS have noted many risk loci of PSP (STX6 and EIF2AK3), suggesting pathogenetic mechanisms related to PSP. Despite the limited evidence, it seems that genetics influence the susceptibility to MSA and PSP. MAPT mutations result in the MSA and PSP pathologies. Further studies are crucial to elucidate the pathogeneses of MSA and PSP, which will support efforts to develop novel drug options.
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spelling pubmed-100488722023-03-29 Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature Bougea, Anastasia Int J Mol Sci Review Multiple system atrophy (MSA) and progressive supranuclear palsy (PSP) are uncommon multifactorial atypical Parkinsonian syndromes, expressed by various clinical features. MSA and PSP are commonly considered sporadic neurodegenerative disorders; however, our understanding is improving of their genetic framework. The purpose of this study was to critically review the genetics of MSA and PSP and their involvement in the pathogenesis. A systemized literature search of PubMed and MEDLINE was performed up to 1 January 2023. Narrative synthesis of the results was undertaken. In total, 43 studies were analyzed. Although familial MSA cases have been reported, the hereditary nature could not be demonstrated. COQ2 mutations were involved in familial and sporadic MSA, without being reproduced in various clinical populations. In terms of the genetics of the cohort, synuclein alpha (SNCA) polymorphisms were correlated with an elevated likelihood of manifesting MSA in Caucasians, but a causal effect relationship could not be demonstrated. Fifteen MAPT mutations were linked with PSP. Leucine-rich repeat kinase 2 (LRRK2) is an infrequent monogenic mutation of PSP. Dynactin subunit 1 (DCTN1) mutations may imitate the PSP phenotype. GWAS have noted many risk loci of PSP (STX6 and EIF2AK3), suggesting pathogenetic mechanisms related to PSP. Despite the limited evidence, it seems that genetics influence the susceptibility to MSA and PSP. MAPT mutations result in the MSA and PSP pathologies. Further studies are crucial to elucidate the pathogeneses of MSA and PSP, which will support efforts to develop novel drug options. MDPI 2023-03-09 /pmc/articles/PMC10048872/ /pubmed/36982356 http://dx.doi.org/10.3390/ijms24065281 Text en © 2023 by the author. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Review
Bougea, Anastasia
Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature
title Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature
title_full Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature
title_fullStr Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature
title_full_unstemmed Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature
title_short Genetics of Multiple System Atrophy and Progressive Supranuclear Palsy: A Systemized Review of the Literature
title_sort genetics of multiple system atrophy and progressive supranuclear palsy: a systemized review of the literature
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10048872/
https://www.ncbi.nlm.nih.gov/pubmed/36982356
http://dx.doi.org/10.3390/ijms24065281
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