Multiple N-of-1 trials to investigate hypoxia therapy in Parkinson's disease: study rationale and protocol.


Journal

BMC neurology
ISSN: 1471-2377
Titre abrégé: BMC Neurol
Pays: England
ID NLM: 100968555

Informations de publication

Date de publication:
14 Jul 2022
Historique:
received: 23 05 2022
accepted: 24 06 2022
entrez: 14 7 2022
pubmed: 15 7 2022
medline: 19 7 2022
Statut: epublish

Résumé

Parkinson's disease (PD) is a neurodegenerative disease, for which no disease-modifying therapies exist. Preclinical and clinical evidence suggest that hypoxia-based therapy might have short- and long-term benefits in PD. We present the contours of the first study to assess the safety, feasibility and physiological and symptomatic impact of hypoxia-based therapy in individuals with PD. In 20 individuals with PD, we will investigate the safety, tolerability and short-term symptomatic efficacy of continuous and intermittent hypoxia using individual, double-blind, randomized placebo-controlled N-of-1 trials. This design allows for dose finding and for including more individualized outcomes, as each individual serves as its own control. A wide range of exploratory outcomes is deployed, including the Movement Disorders Society Unified Parkinson's Disease Rating scale (MDS-UPDRS) part III, Timed Up & Go Test, Mini Balance Evaluation Systems (MiniBES) test and wrist accelerometry. Also, self-reported impression of overall symptoms, motor and non-motor symptoms and urge to take dopaminergic medication will be assessed on a 10-point Likert scale. As part of a hypothesis-generating part of the study, we also deploy several exploratory outcomes to probe possible underlying mechanisms of action, including cortisol, erythropoietin and platelet-derived growth factor β. Efficacy will be assessed primarily by a Bayesian analysis. This evaluation of hypoxia therapy could provide insight in novel pathways that may be pursued for PD treatment. This trial also serves as a proof of concept for deploying an N-of-1 design and for including individualized outcomes in PD research, as a basis for personalized treatment approaches. ClinicalTrials.gov Identifier: NCT05214287 (registered January 28, 2022).

Sections du résumé

BACKGROUND BACKGROUND
Parkinson's disease (PD) is a neurodegenerative disease, for which no disease-modifying therapies exist. Preclinical and clinical evidence suggest that hypoxia-based therapy might have short- and long-term benefits in PD. We present the contours of the first study to assess the safety, feasibility and physiological and symptomatic impact of hypoxia-based therapy in individuals with PD.
METHODS/DESIGN METHODS
In 20 individuals with PD, we will investigate the safety, tolerability and short-term symptomatic efficacy of continuous and intermittent hypoxia using individual, double-blind, randomized placebo-controlled N-of-1 trials. This design allows for dose finding and for including more individualized outcomes, as each individual serves as its own control. A wide range of exploratory outcomes is deployed, including the Movement Disorders Society Unified Parkinson's Disease Rating scale (MDS-UPDRS) part III, Timed Up & Go Test, Mini Balance Evaluation Systems (MiniBES) test and wrist accelerometry. Also, self-reported impression of overall symptoms, motor and non-motor symptoms and urge to take dopaminergic medication will be assessed on a 10-point Likert scale. As part of a hypothesis-generating part of the study, we also deploy several exploratory outcomes to probe possible underlying mechanisms of action, including cortisol, erythropoietin and platelet-derived growth factor β. Efficacy will be assessed primarily by a Bayesian analysis.
DISCUSSION CONCLUSIONS
This evaluation of hypoxia therapy could provide insight in novel pathways that may be pursued for PD treatment. This trial also serves as a proof of concept for deploying an N-of-1 design and for including individualized outcomes in PD research, as a basis for personalized treatment approaches.
TRIAL REGISTRATION BACKGROUND
ClinicalTrials.gov Identifier: NCT05214287 (registered January 28, 2022).

Identifiants

pubmed: 35836147
doi: 10.1186/s12883-022-02770-7
pii: 10.1186/s12883-022-02770-7
pmc: PMC9281145
doi:

Banques de données

ClinicalTrials.gov
['NCT05214287']

Types de publication

Clinical Trial Protocol Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

262

Subventions

Organisme : Michael J. Fox Foundation for Parkinson's Research
ID : 019201
Organisme : Michael J. Fox Foundation for Parkinson's Research
ID : 019201
Organisme : Michael J. Fox Foundation for Parkinson's Research
ID : 019201

Commentaires et corrections

Type : CommentIn

Informations de copyright

© 2022. The Author(s).

Références

J Neurochem. 2003 Apr;85(2):483-91
pubmed: 12675925
J Cereb Blood Flow Metab. 2018 Jan;38(1):45-57
pubmed: 28741407
BMJ. 2015 May 14;350:h1738
pubmed: 25976398
Exp Neurol. 2021 Aug;342:113722
pubmed: 33932397
BMC Geriatr. 2019 Jun 14;19(1):167
pubmed: 31200649
J Pharmacol Sci. 2009 Jul;110(3):270-5
pubmed: 19542682
Int J Technol Assess Health Care. 2014 Oct;30(4):416-22
pubmed: 25407328
Int J Cardiol. 2004 Aug;96(2):247-54
pubmed: 15262041
J Appl Physiol (1985). 2015 Mar 1;118(5):520-32
pubmed: 25549763
Front Neurol. 2016 Mar 08;7:27
pubmed: 27014181
Mol Neurobiol. 2016 Aug;53(6):3812-3821
pubmed: 26156288
Acta Neuropathol Commun. 2020 Oct 28;8(1):175
pubmed: 33115539
N Engl J Med. 1986 Apr 3;314(14):889-92
pubmed: 2936958
Neurology. 2014 Jan 14;82(2):104-13
pubmed: 24285617
Scand J Med Sci Sports. 2012 Oct;22(5):e79-85
pubmed: 22853822
Elife. 2018 Apr 24;7:
pubmed: 29688171
J Neurochem. 2010 Oct;115(1):209-19
pubmed: 20649842
BMJ. 2020 Feb 27;368:m122
pubmed: 32107202
J Neurosci Res. 2009 May 15;87(7):1686-94
pubmed: 19156859
Metab Brain Dis. 1989 Jun;4(2):143-53
pubmed: 2755415
Int J Environ Res Public Health. 2021 Apr 02;18(7):
pubmed: 33918381
J Am Geriatr Soc. 1991 Feb;39(2):142-8
pubmed: 1991946
J Biol Chem. 2005 Feb 11;280(6):4321-8
pubmed: 15569687
J Appl Physiol (1985). 1994 Dec;77(6):2537-42
pubmed: 7896588
Eur Heart J. 2001 May;22(10):879-86
pubmed: 11350098
JAMA. 2018 Dec 11;320(22):2365-2366
pubmed: 30535206
Adv Gerontol. 2017;30(2):255-261
pubmed: 28575566
FASEB J. 2003 Sep;17(12):1709-11
pubmed: 12958184
J Parkinsons Dis. 2020;10(4):1315-1333
pubmed: 32986684
Respir Physiol. 1997 Mar;107(3):231-9
pubmed: 9128904
Am J Respir Crit Care Med. 2020 Sep 15;202(6):887-890
pubmed: 32369393
Fiziol Zh (1994). 2016;62(3):78-91
pubmed: 29569889
J Appl Psychol. 1948 Jun;32(3):234-47
pubmed: 18867059
Cardiovasc Drugs Ther. 2010 Jun;24(3):235-54
pubmed: 20496105
Front Physiol. 2020 Nov 20;11:570472
pubmed: 33329021
Neurobiol Dis. 2016 Oct;94:95-105
pubmed: 27288154
Neurorehabil Neural Repair. 2012 Feb;26(2):163-72
pubmed: 21821826
Per Med. 2011 Mar;8(2):161-173
pubmed: 21695041
Exp Neurol. 2022 Jan;347:113891
pubmed: 34637802
J Neurochem. 2006 Jan;96(2):428-43
pubmed: 16336625
J Appl Physiol (1985). 1989 Apr;66(4):1736-43
pubmed: 2732164
Physiol Rev. 2012 Jul;92(3):967-1003
pubmed: 22811423
J Card Fail. 2012 May;18(5):387-91
pubmed: 22555269
Neurosci Lett. 2015 Oct 21;607:97-101
pubmed: 26407747
Neurochem Int. 2005 Jun;46(8):623-33
pubmed: 15863240
Obes Facts. 2015;8(3):200-9
pubmed: 26008855
Mov Disord. 2021 Apr;36(4):857-861
pubmed: 33638916
Am J Physiol Regul Integr Comp Physiol. 2014 Nov 15;307(10):R1181-97
pubmed: 25231353
Sports Med. 2009;39(2):107-27
pubmed: 19203133
Steroids. 2005 Nov;70(12):803-10
pubmed: 16019044
JAMA Neurol. 2018 Jan 1;75(1):9-10
pubmed: 29131880
J Physiol. 2000 Nov 15;529 Pt 1:215-9
pubmed: 11080263
J Physiol. 2003 Feb 1;546(Pt 3):921-9
pubmed: 12563015
Mov Disord. 2014 Dec;29(14):1710-9
pubmed: 25297066
Fundam Clin Pharmacol. 2012 Aug;26(4):557-64
pubmed: 21585523
Am J Prev Med. 2009 May;36(5):452-7
pubmed: 19362699
J Neurochem. 1991 Sep;57(3):997-1002
pubmed: 1861163
Annu Rev Physiol. 2014;76:39-56
pubmed: 23988176
Curr Med Chem. 2011;18(28):4335-43
pubmed: 21861815
BMC Med Res Methodol. 2013 Aug 20;13:104
pubmed: 23961782
BMC Neurol. 2015 Mar 25;15:43
pubmed: 25880166
Int J Behav Nutr Phys Act. 2011 Oct 21;8:115
pubmed: 22018588
Diabetes. 2013 Dec;62(12):4220-7
pubmed: 23733200
Wilderness Environ Med. 2000 Fall;11(3):172-9
pubmed: 11055563
J Sports Sci. 2010 Mar;28(5):513-9
pubmed: 20419592
Acta Physiol Scand. 1998 Mar;162(3):389-99
pubmed: 9578385
Diabetes Care. 2013 Aug;36(8):2183-9
pubmed: 23536585
Respir Physiol Neurobiol. 2009 Jan 1;165(1):97-103
pubmed: 19013544
Eur Respir J. 2011 Apr;37(4):880-7
pubmed: 20947680
Alzheimers Dement (N Y). 2017 Feb 08;3(1):114-122
pubmed: 29067323
Microvasc Res. 2012 Sep;84(2):222-5
pubmed: 22668821
J Appl Physiol (1985). 2017 Dec 1;123(6):1689-1697
pubmed: 29074711
ASN Neuro. 2019 Jan-Dec;11:1759091419871420
pubmed: 31450955
J Rehabil Med. 2010 Apr;42(4):323-31
pubmed: 20461334
Healthcare (Basel). 2019 Nov 06;7(4):
pubmed: 31698799
Exp Neurol. 2021 Jan;335:113483
pubmed: 32987000
JAMA. 2018 Dec 11;320(22):2344-2353
pubmed: 30535218
Neurology. 2017 Oct 31;89(18):1904-1907
pubmed: 28972191
Respiration. 2000;67(3):253-60
pubmed: 10867592
Age Ageing. 1997 Sep;26(5):353-7
pubmed: 9351479
Science. 2016 Apr 1;352(6281):54-61
pubmed: 26917594
Front Neurosci. 2020 Aug 18;14:869
pubmed: 32973432
J Cereb Blood Flow Metab. 2019 Aug;39(8):1501-1515
pubmed: 29629621
Int J Mol Sci. 2019 Oct 30;20(21):
pubmed: 31671598

Auteurs

Jules M Janssen Daalen (JM)

Center of Expertise for Parkinson & Movement Disorders; Nijmegen, the Netherlands, Department of Neurology, Donders Institute for Brain, Cognition and Behavior, Radboud University Medical Center, Nijmegen, The Netherlands.

Marjan J Meinders (MJ)

Center of Expertise for Parkinson & Movement Disorders; Nijmegen, the Netherlands, Department of Neurology, Donders Institute for Brain, Cognition and Behavior, Radboud University Medical Center, Nijmegen, The Netherlands.
IQ Healthcare, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, The Netherlands.

Federica Giardina (F)

Department of Health Evidence, Radboud Institute for Health Sciences, Radboud University Medical Center, Section Biostatistics, Nijmegen, The Netherlands.

Kit C B Roes (KCB)

Department of Health Evidence, Radboud Institute for Health Sciences, Radboud University Medical Center, Section Biostatistics, Nijmegen, The Netherlands.

Bas C Stunnenberg (BC)

Department of Neurology, Donders Institute for Brain, Cognition and Behavior, Radboud University Medical Center, Nijmegen, The Netherlands.
Department of Neurology, Rijnstate Hospital, Arnhem, Netherlands.

Soania Mathur (S)

UnshakeableMD, Oshawa, ON, Canada.

Philip N Ainslie (PN)

Center for Heart, Lung and Vascular Health, School of Health and Exercise Sciences, University of British Columbia, Kelowna, Canada.

Dick H J Thijssen (DHJ)

Department of Physiology, Radboud University Medical Center, Nijmegen, The Netherlands.

Bastiaan R Bloem (BR)

Center of Expertise for Parkinson & Movement Disorders; Nijmegen, the Netherlands, Department of Neurology, Donders Institute for Brain, Cognition and Behavior, Radboud University Medical Center, Nijmegen, The Netherlands. Bas.Bloem@radboudumc.nl.

Articles similaires

[Redispensing of expensive oral anticancer medicines: a practical application].

Lisanne N van Merendonk, Kübra Akgöl, Bastiaan Nuijen
1.00
Humans Antineoplastic Agents Administration, Oral Drug Costs Counterfeit Drugs

Smoking Cessation and Incident Cardiovascular Disease.

Jun Hwan Cho, Seung Yong Shin, Hoseob Kim et al.
1.00
Humans Male Smoking Cessation Cardiovascular Diseases Female
Humans United States Aged Cross-Sectional Studies Medicare Part C
1.00
Humans Yoga Low Back Pain Female Male

Classifications MeSH