A Comprehensive Assessment of Qualitative and Quantitative Prodromal Parkinsonian Features in Carriers of Gaucher Disease-Identifying Those at the Greatest Risk.
Gaucher disease carriers
Parkinson’s disease
carriers of GBA1 variants
prodromal Parkinson disease
Journal
International journal of molecular sciences
ISSN: 1422-0067
Titre abrégé: Int J Mol Sci
Pays: Switzerland
ID NLM: 101092791
Informations de publication
Date de publication:
13 Oct 2022
13 Oct 2022
Historique:
received:
01
08
2022
revised:
12
09
2022
accepted:
14
09
2022
entrez:
27
10
2022
pubmed:
28
10
2022
medline:
29
10
2022
Statut:
epublish
Résumé
Carriers of GBA1 gene variants have a significant risk of developing Parkinson’s disease (PD). A cohort study of GBA carriers between 40−75 years of age was initiated to study the presence of prodromal PD features. Participants underwent non-invasive tests to assess different domains of PD. Ninety-eight unrelated GBA carriers were enrolled (43 males) at a median age (range) of 51 (40−74) years; 71 carried the N370S variant (c.1226A > G) and 25 had a positive family history of PD. The Montreal Cognitive Assessment (MoCA) was the most frequently abnormal (23.7%, 95% CI 15.7−33.4%), followed by the ultrasound hyperechogenicity (22%, 95% CI 14−32%), Unified Parkinson’s Disease Rating Scale part III (UPDRS-III) (17.2%, 95% CI 10.2−26.4%), smell assessment (12.4%, 95% CI 6.6−20.6%) and abnormalities in sleep questionnaires (11%, 95% CI 5.7−19.4%). Significant correlations were found between tests from different domains. To define the risk for PD, we assessed the bottom 10th percentile of each prodromal test, defining this level as “abnormal”. Then we calculated the percentage of “abnormal” tests for each subject; the median (range) was 4.55 (0−43.5%). Twenty-two subjects had more than 15% “abnormal” tests. The limitations of the study included ascertainment bias of individuals with GBA-related PD in relatives, some incomplete data due to technical issues, and a lack of well-characterized normal value ranges in some tests. We plan to enroll additional participants and conduct longitudinal follow-up assessments to build a model for identifying individuals at risk for PD and investigate interventions aiming to delay the onset or perhaps to prevent full-blown PD.
Identifiants
pubmed: 36293067
pii: ijms232012211
doi: 10.3390/ijms232012211
pmc: PMC9603254
pii:
doi:
Substances chimiques
Glucosylceramidase
EC 3.2.1.45
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Subventions
Organisme : Takeda Israel
ID : None
Références
Neurology. 2015 Mar 3;84(9):866-7
pubmed: 25653294
Metabolism. 2015 Mar;64(3 Suppl 1):S40-6
pubmed: 25510818
Lancet Neurol. 2008 May;7(5):417-24
pubmed: 18394965
Ultrasound Med Biol. 2020 May;46(5):1208-1215
pubmed: 32102740
Brain. 2009 May;132(Pt 5):1128-45
pubmed: 19336464
Int Rev Neurobiol. 2017;132:197-231
pubmed: 28554408
Blood Cells Mol Dis. 2018 Feb;68:115-116
pubmed: 27864021
J Clin Neurosci. 2020 Jun;76:31-35
pubmed: 32331947
Brain. 2015 Sep;138(Pt 9):2648-58
pubmed: 26117366
Brain. 2012 Jun;135(Pt 6):1860-70
pubmed: 22561644
CNS Neurol Disord Drug Targets. 2012 Nov 1;11(7):836-43
pubmed: 23198691
Neurosci Bull. 2014 Feb;30(1):134-40
pubmed: 24136244
Mov Disord. 2010 Jul 15;25(9):1150-6
pubmed: 20629141
Mov Disord. 2015 Jun;30(7):919-27
pubmed: 25737166
Neurology. 2008 Jun 10;70(24):2277-83
pubmed: 18434642
J Affect Disord. 2015 Sep 15;184:216-24
pubmed: 26114228
Neurosci Biobehav Rev. 2008;32(4):611-56
pubmed: 18061262
J Parkinsons Dis. 2015;5(4):681-97
pubmed: 26485429
Nature. 2021 Nov;599(7886):650-656
pubmed: 34732887
N Engl J Med. 2009 Oct 22;361(17):1651-61
pubmed: 19846850
JAMA Neurol. 2015 Feb;72(2):201-8
pubmed: 25506732
Neurol Sci. 2020 Jun;41(6):1419-1426
pubmed: 32030568
Parkinsonism Relat Disord. 2018 Jul;52:83-89
pubmed: 29625875
Eur Neurol. 2017;78(5-6):330-337
pubmed: 29084403
Neurotox Res. 2019 Oct;36(3):452-462
pubmed: 31016688
Neurology. 2020 Aug 25;95(8):e1008-e1016
pubmed: 32591474
J Clin Neurosci. 2019 Feb;60:68-72
pubmed: 30340975
Parkinsonism Relat Disord. 2016 Mar;24:28-33
pubmed: 26842545
Isr Med Assoc J. 2020 Jan;22(1):37-42
pubmed: 31927804
Eur J Neurol. 2019 Jul;26(7):1013-1018
pubmed: 30714262
Brain. 2009 Dec;132(Pt 12):3298-307
pubmed: 19843648
Int J Geriatr Psychiatry. 2018 Feb;33(2):237-251
pubmed: 28627719
Mov Disord. 2015 Oct;30(12):1600-11
pubmed: 26474317
Ultrasound Med Biol. 2019 Jan;45(1):122-128
pubmed: 30482710
Psychol Bull. 2017 Aug;143(8):783-822
pubmed: 28447828
Parkinsonism Relat Disord. 2017 Mar;36:93-97
pubmed: 28089265
Neurol Genet. 2016 Mar 04;2(2):e57
pubmed: 27123476
Eur J Neurol. 2019 Apr;26(4):694-698
pubmed: 30107068
JAMA. 2020 Feb 11;323(6):548-560
pubmed: 32044947
Br J Ophthalmol. 2002 Dec;86(12):1408-11
pubmed: 12446376
Neurology. 2015 Mar 3;84(9):880-7
pubmed: 25653295
Sci Rep. 2020 May 26;10(1):8660
pubmed: 32457446
Mov Disord. 2006 May;21(5):668-72
pubmed: 16450355
Int Rev Psychiatry. 2010;22(5):429-36
pubmed: 21047157
QJM. 1996 Sep;89(9):691-4
pubmed: 8917744
Blood Cells Mol Dis. 2006 May-Jun;36(3):426-8
pubmed: 16651014
Int J Neurosci. 2010 Aug;120(8):538-43
pubmed: 20615057
Iran J Neurol. 2018 Jul 6;17(3):145-148
pubmed: 30886682
Sensors (Basel). 2019 Nov 26;19(23):
pubmed: 31779224
Int J Mol Sci. 2017 Mar 04;18(3):
pubmed: 28273839
Mov Disord. 2007 Dec;22(16):2386-93
pubmed: 17894337
Transl Neurodegener. 2022 Feb 21;11(1):11
pubmed: 35184752
Biomed Pharmacother. 2020 Dec;132:110890
pubmed: 33080465
Mov Disord. 2018 Mar;33(3):405-413
pubmed: 29436728
Parkinsonism Relat Disord. 2019 Sep;66:138-142
pubmed: 31371182
Mov Disord. 2022 Mar;37(3):629-634
pubmed: 34762337
Lancet Neurol. 2020 Jan;19(1):71-80
pubmed: 31678032
J Neurosurg Sci. 2019 Aug;63(4):441-449
pubmed: 31210040
Ultraschall Med. 2014 Aug;35(4):322-31
pubmed: 24764215
Mov Disord. 2019 Oct;34(10):1464-1470
pubmed: 31412427