Predictive modeling of spread in adult-onset isolated dystonia: Key properties and effect of tremor inclusion.
isolated dystonia
neurological diseases
predictive models
spread
tremor
Journal
European journal of neurology
ISSN: 1468-1331
Titre abrégé: Eur J Neurol
Pays: England
ID NLM: 9506311
Informations de publication
Date de publication:
12 2021
12 2021
Historique:
revised:
12
07
2021
received:
18
05
2021
accepted:
16
07
2021
pubmed:
24
7
2021
medline:
2
4
2022
entrez:
23
7
2021
Statut:
ppublish
Résumé
Several clinical and demographic factors relate to anatomic spread of adult-onset isolated dystonia, but a predictive model is still lacking. The aims of this study were: (i) to develop and validate a predictive model of anatomic spread of adult-onset isolated dystonia; and (ii) to evaluate whether presence of tremor associated with dystonia influences model predictions of spread. Adult-onset isolated dystonia participants with focal onset from the Dystonia Coalition Natural History Project database were included. We developed two prediction models, one with dystonia as sole disease manifestation ("dystonia-only") and one accepting dystonia OR tremor in any body part as disease manifestations ("dystonia OR tremor"). Demographic and clinical predictors were selected based on previous evidence, clinical plausibility of association with spread, or both. We used logistic regressions and evaluated model discrimination and calibration. Internal validation was carried out based on bootstrapping. Both predictive models showed an area under the curve of 0.65 (95% confidence intervals 0.62-0.70 and 0.62-0.69, respectively) and good calibration after internal validation. In both models, onset of dystonia in body regions other than the neck, older age, depression and history of neck trauma were predictors of spread. This predictive modeling of spread in adult-onset isolated dystonia based on accessible predictors (demographic and clinical) can be easily implemented to inform individuals' risk of spread. Because tremor did not influence prediction of spread, our results support the argument that tremor is a part of the dystonia syndrome, and not an independent or coincidental disorder.
Sections du résumé
BACKGROUND AND PURPOSE
Several clinical and demographic factors relate to anatomic spread of adult-onset isolated dystonia, but a predictive model is still lacking. The aims of this study were: (i) to develop and validate a predictive model of anatomic spread of adult-onset isolated dystonia; and (ii) to evaluate whether presence of tremor associated with dystonia influences model predictions of spread.
METHODS
Adult-onset isolated dystonia participants with focal onset from the Dystonia Coalition Natural History Project database were included. We developed two prediction models, one with dystonia as sole disease manifestation ("dystonia-only") and one accepting dystonia OR tremor in any body part as disease manifestations ("dystonia OR tremor"). Demographic and clinical predictors were selected based on previous evidence, clinical plausibility of association with spread, or both. We used logistic regressions and evaluated model discrimination and calibration. Internal validation was carried out based on bootstrapping.
RESULTS
Both predictive models showed an area under the curve of 0.65 (95% confidence intervals 0.62-0.70 and 0.62-0.69, respectively) and good calibration after internal validation. In both models, onset of dystonia in body regions other than the neck, older age, depression and history of neck trauma were predictors of spread.
CONCLUSIONS
This predictive modeling of spread in adult-onset isolated dystonia based on accessible predictors (demographic and clinical) can be easily implemented to inform individuals' risk of spread. Because tremor did not influence prediction of spread, our results support the argument that tremor is a part of the dystonia syndrome, and not an independent or coincidental disorder.
Identifiants
pubmed: 34296504
doi: 10.1111/ene.15031
pmc: PMC9100858
mid: NIHMS1797827
doi:
Types de publication
Journal Article
Research Support, N.I.H., Extramural
Langues
eng
Sous-ensembles de citation
IM
Pagination
3999-4009Subventions
Organisme : NINDS NIH HHS
ID : U54 NS065701
Pays : United States
Organisme : NINDS NIH HHS
ID : U54 NS116025
Pays : United States
Organisme : NCATS NIH HHS
ID : U54 TR001456
Pays : United States
Informations de copyright
© 2021 European Academy of Neurology.
Références
Neurol Genet. 2016 Apr 11;2(3):e69
pubmed: 27123488
Mov Disord. 2020 Nov;35(11):2086-2090
pubmed: 32845549
Mov Disord. 2012 Sep 15;27(11):1447-50
pubmed: 22890501
Neurosci Biobehav Rev. 2021 Jun;125:221-230
pubmed: 33662441
Mov Disord. 2020 Mar;35(3):450-456
pubmed: 31774238
Mov Disord. 2016 Dec;31(12):1874-1882
pubmed: 27753188
Neurology. 2021 Jan 26;96(4):e563-e574
pubmed: 33046615
Eur J Neurol. 2006 Oct;13(10):1083-8
pubmed: 16987160
J Neurosci. 2020 Nov 25;40(48):9317-9326
pubmed: 33097635
Arq Neuropsiquiatr. 2009 Jun;67(2B):402-6
pubmed: 19623434
Neurol Neurochir Pol. 2013 May-Jun;47(3):223-31
pubmed: 23821419
J Neurol. 2007 Jul;254(7):879-83
pubmed: 17401742
J Neurol Neurosurg Psychiatry. 2017 Jul;88(7):595-602
pubmed: 28438790
Mov Disord. 2011 Aug 15;26(10):1789-92
pubmed: 21735481
Mov Disord. 2020 Nov;35(11):2038-2045
pubmed: 32662572
J Neurol Neurosurg Psychiatry. 2008 Apr;79(4):392-6
pubmed: 17635969
Mov Disord. 2020 Dec;35(12):2270-2278
pubmed: 32940390
Mov Disord. 2006 Aug;21(8):1175-81
pubmed: 16673404
Parkinsonism Relat Disord. 2016 Sep;30:7-12
pubmed: 27321988
J Biomed Inform. 2014 Apr;48:193-204
pubmed: 24582925
Cerebellum. 2021 Oct;20(5):678-686
pubmed: 31965455
J Neurol. 2004 Feb;251(2):150-5
pubmed: 14991348
Mov Disord. 2010 Mar 15;25(4):459-65
pubmed: 20108377
Eur J Neurol. 2018 Nov;25(11):1341-1344
pubmed: 29935029
Neurology. 2018 Nov 20;91(21):e2020-e2026
pubmed: 30341158
Mov Disord. 2015 May;30(6):834-42
pubmed: 25879911
Psychometrika. 2010 Dec;75(4):649-674
pubmed: 21720450
Parkinsonism Relat Disord. 2020 Feb;71:40-43
pubmed: 32007783
J Neurol Neurosurg Psychiatry. 2020 Mar;91(3):314-320
pubmed: 31848221
Front Neurol. 2015 Dec 14;6:258
pubmed: 26696957
Mov Disord. 1998;13 Suppl 3:2-23
pubmed: 9827589
Mov Disord. 2018 Jan;33(1):75-87
pubmed: 29193359
Clin Neurol Neurosurg. 2015 May;132:41-3
pubmed: 25764999
J Neurol Neurosurg Psychiatry. 2013 Apr;84(4):404-8
pubmed: 23142961
Neurology. 2020 Feb 11;94(6):e639-e650
pubmed: 31937622
Circulation. 2015 Jan 13;131(2):211-9
pubmed: 25561516
J Neurol Neurosurg Psychiatry. 2014 Sep;85(9):965-8
pubmed: 24249781
Mov Disord. 2020 Jun;35(6):1067-1071
pubmed: 32199036
J Neurol Neurosurg Psychiatry. 1999 Nov;67(5):613-9
pubmed: 10519867
Mov Disord. 2013 Jun 15;28(7):863-73
pubmed: 23649720
Neurology. 2015 Mar 10;84(10):1053-9
pubmed: 25663232
Epidemiology. 2010 Jan;21(1):128-38
pubmed: 20010215