Accelerometer; Healthy volunteers; Home monitoring; Stride length; Stride velocity
Abstract :
[en] Background: Normative data are necessary for validation of new outcome measures. Recently, the 95th centile of stride speed was qualified by the European Medicines Agency as a valid secondary outcome for clinical trials in subjects with Duchenne muscular dystrophy. This study aims to obtain normative data on spontaneous stride velocity and length in a non-controlled environment and their evolution after 12 months.
Method: Ninety-one healthy volunteers (50 females, 41 males), with a mean age of 16 years and 2 months, were recruited and assessed at baseline and 12 months later. The 4-stair climb, 6-min walk test, 10-m walk test and rise from floor assessments were performed. Stride length, stride velocity, and the distance walked per hour were studied in an everyday setting for one month after each evaluation.
Results: Of the 91 subjects assessed, 82 provided more than 50 h of recordings at baseline; and 73 subjects provided the same at the end of the year. We observed significant positive correlations of the stride length with age and height of participants, and a significant increase of the median stride length in children after the period. In this group, the 95th centile stride velocity was not correlated with age and was stable after one year. All measures but the 10MWT were stable in adults after a one-year period.
Conclusion: This study provides with data on the influence of age, height, and gender on stride velocity and length as well as accounting for natural changes after one year in controls.
Disciplines :
Neurology
Author, co-author :
Poleur, Margaux ; Centre Hospitalier Universitaire de Liège - CHU > Département de médecine interne > Service de neurologie (CHR)
Ulinici, Ana
DARON, Aurore ; Centre Hospitalier Universitaire de Liège - CHU > Département de Pédiatrie > Service de pédiatrie
Schneider, Olivier
Dal Farra, Fabian
Demonceau, Marie
Annoussamy, Mélanie
Vissière, David
Eggenspieler, Damien
Servais, Laurent ; Centre Hospitalier Universitaire de Liège - CHU > Département de Pédiatrie > Service de pédiatrie
Language :
English
Title :
Normative data on spontaneous stride velocity, stride length, and walking activity in a non-controlled environment.
Ryder S, Leadley RM, Armstrong N, Westwood M, de Kock S, Butt T, et al. The burden, epidemiology, costs and treatment for Duchenne muscular dystrophy: an evidence review. Orphanet J Rare Dis. 2017;12(1):79. 10.1186/s13023-017-0631-3. DOI: 10.1186/s13023-017-0631-3
Mah JK, Korngut L, Dykeman J, Day L, Pringsheim T, Jette N. A systematic review and meta-analysis on the epidemiology of Duchenne and Becker muscular dystrophy. Neuromuscul Disord. 2014;24(6):482–91. 10.1016/j.nmd.2014.03.008. DOI: 10.1016/j.nmd.2014.03.008
Clemens PR, Rao VK, Connolly AM, Harper AD, Mah JK, Smith EC, et al. Safety, tolerability, and efficacy of viltolarsen in boys with duchenne muscular dystrophy amenable to exon 53 skipping: a phase 2 randomized clinical trial. JAMA Neurol. 2020;77(8):982–91. 10.1001/jamaneurol.2020.1264. DOI: 10.1001/jamaneurol.2020.1264
Frank DE, Schnell FJ, Akana C, El-Husayni SH, Desjardins CA, Morgan J, et al. Increased dystrophin production with golodirsen in patients with Duchenne muscular dystrophy. Neurology. 2020;94(21):e2270–82. 10.1212/wnl.0000000000009233. DOI: 10.1212/wnl.0000000000009233
McDonald CM, Campbell C, Torricelli RE, Finkel RS, Flanigan KM, Goemans N, et al. Ataluren in patients with nonsense mutation Duchenne muscular dystrophy (ACT DMD): a multicentre, randomised, double-blind, placebo-controlled, phase 3 trial. Lancet. 2017;390(10101):1489–98. 10.1016/s0140-6736(17)31611-2. DOI: 10.1016/s0140-6736(17)31611-2
Mendell JR, Rodino-Klapac LR, Sahenk Z, Roush K, Bird L, Lowes LP, et al. Eteplirsen for the treatment of Duchenne muscular dystrophy. Ann Neurol. 2013;74(5):637–47. 10.1002/ana.23982. DOI: 10.1002/ana.23982
Bushby K, Finkel R, Wong B, Barohn R, Campbell C, Comi GP, et al. Ataluren treatment of patients with nonsense mutation dystrophinopathy. Muscle Nerve. 2014;50(4):477–87. 10.1002/mus.24332. DOI: 10.1002/mus.24332
Goemans N, Mercuri E, Belousova E, Komaki H, Dubrovsky A, McDonald CM, et al. A randomized placebo-controlled phase 3 trial of an antisense oligonucleotide, drisapersen Duchenne muscular dystrophy. Neuromuscul Disord. 2018;28(1):4–15. 10.1016/j.nmd.2017.10.004. DOI: 10.1016/j.nmd.2017.10.004
Voit T, Topaloglu H, Straub V, Muntoni F, Deconinck N, Campion G, et al. Safety and efficacy of drisapersen for the treatment of Duchenne muscular dystrophy (DEMAND II): an exploratory, randomised, placebo-controlled phase 2 study. Lancet Neurol. 2014;13(10):987–96. 10.1016/s1474-4422(14)70195-4. DOI: 10.1016/s1474-4422(14)70195-4
Assessment report for Kyndrisa. European Medicines Agency. https://www.ema.europa.eu/en/documents/withdrawal-report/withdrawal-assessment-report-kyndrisa_en.pdf. Accessed 1 Dec 2020.
Piwek L, Ellis DA, Andrews S, Joinson A. The Rise of Consumer Health Wearables: Promises and Barriers. PLoS Med. 2016;13(2): e1001953. 10.1371/journal.pmed.1001953. DOI: 10.1371/journal.pmed.1001953
Caulfield B, Reginatto B, Slevin P. Not all sensors are created equal: a framework for evaluating human performance measurement technologies. NPJ Digit Med. 2019;2:7. 10.1038/s41746-019-0082-4. DOI: 10.1038/s41746-019-0082-4
Le Moing AG, Seferian AM, Moraux A, Annoussamy M, Dorveaux E, Gasnier E, et al. A movement monitor based on magneto-inertial sensors for non-ambulant patients with duchenne muscular dystrophy: a pilot study in controlled environment. PLoS ONE. 2016;11(6): e0156696. 10.1371/journal.pone.0156696. DOI: 10.1371/journal.pone.0156696
Lilien C, Gasnier E, Gidaro T, Seferian A, Grelet M, Vissière D, et al. Home-based monitor for gait and activity analysis. J Vis Exp. 2019. 10.3791/59668. DOI: 10.3791/59668
Qualification opinion on stride velocity 95th centile as a secondary endpoint in Duchenne muscular dystrophy measured by a valid and suitable wearable device*European Medicines Agency. https://www.ema.europa.eu/en/documents/scientific-guideline/qualification-opinion-stride-velocity-95th-centile-secondary-endpoint-duchenne-muscular-dystrophy_en.pdf. Accessed 15 July 2021.
Haberkamp M, Moseley J, Athanasiou D, de Andres-Trelles F, Elferink A, Rosa MM, et al. European regulators’ views on a wearable-derived performance measurement of ambulation for Duchenne muscular dystrophy regulatory trials. Neuromuscul Disord. 2019;29(7):514–6. 10.1016/j.nmd.2019.06.003. DOI: 10.1016/j.nmd.2019.06.003
Chabanon A, Seferian AM, Daron A, Péréon Y, Cances C, Vuillerot C, et al. Prospective and longitudinal natural history study of patients with Type 2 and 3 spinal muscular atrophy: baseline data NatHis-SMA study. PLoS ONE. 2018;13(7): e0201004. 10.1371/journal.pone.0201004. DOI: 10.1371/journal.pone.0201004
Annoussamy M, Seferian AM, Daron A, Péréon Y, Cances C, Vuillerot C, et al. Natural history of Type 2 and 3 spinal muscular atrophy: 2-year NatHis-SMA study. Ann Clin Transl Neurol. 2020. 10.1002/acn3.51281. DOI: 10.1002/acn3.51281
Goemans N, Klingels K, van den Hauwe M, Boons S, Verstraete L, Peeters C, et al. Six-minute walk test: reference values and prediction equation in healthy boys aged 5 to 12 years. PLoS ONE. 2013;8(12): e84120. 10.1371/journal.pone.0084120. DOI: 10.1371/journal.pone.0084120
Hogrel JY, Decostre V, Ledoux I, de Antonio M, Niks EH, de Groot I, et al. Normalized grip strength is a sensitive outcome measure through all stages of Duchenne muscular dystrophy. J Neurol. 2020;267(7):2022–8. 10.1007/s00415-020-09800-9. DOI: 10.1007/s00415-020-09800-9
Seferian AM, Moraux A, Canal A, Decostre V, Diebate O, Le Moing AG, et al. Upper limb evaluation and one-year follow up of non-ambulant patients with spinal muscular atrophy: an observational multicenter trial. PLoS ONE. 2015;10(4): e0121799. 10.1371/journal.pone.0121799. DOI: 10.1371/journal.pone.0121799
McDonald CM, Henricson EK, Han JJ, Abresch RT, Nicorici A, Elfring GL, et al. The 6-minute walk test as a new outcome measure in Duchenne muscular dystrophy. Muscle Nerve. 2010;41(4):500–10. 10.1002/mus.21544. DOI: 10.1002/mus.21544
Perry B, Herrington W, Goldsack JC, Grandinetti CA, Vasisht KP, Landray MJ, et al. Use of mobile devices to measure outcomes in clinical research, 2010–2016: a systematic literature review. Digit Biomark. 2018;2(1):11–30. 10.1159/000486347. DOI: 10.1159/000486347
Fang X, Liu C, Jiang Z. Reference values of gait using APDM movement monitoring inertial sensor system. R Soc Open Sci. 2018;5(1): 170818. 10.1098/rsos.170818. DOI: 10.1098/rsos.170818
Voss S, Joyce J, Biskis A, Parulekar M, Armijo N, Zampieri C, et al. Normative database of spatiotemporal gait parameters using inertial sensors in typically developing children and young adults. Gait Posture. 2020;80:206–13. 10.1016/j.gaitpost.2020.05.010. DOI: 10.1016/j.gaitpost.2020.05.010
Shah VV, McNames J, Mancini M, Carlson-Kuhta P, Spain RI, Nutt JG, et al. Quantity and quality of gait and turning in people with multiple sclerosis, Parkinson’s disease and matched controls during daily living. J Neurol. 2020;267(4):1188–96. 10.1007/s00415-020-09696-5. DOI: 10.1007/s00415-020-09696-5
Wiedmann I, Grassi M, Duran I, Lavrador R, Alberg E, Daumer M, et al. Accelerometric Gait analysis devices in children-will they accept them? results from the AVAPed study. Front Pediatr. 2020;8: 574443. 10.3389/fped.2020.574443. DOI: 10.3389/fped.2020.574443
Dusing SC, Thorpe DE. A normative sample of temporal and spatial gait parameters in children using the GAITRite electronic walkway. Gait Posture. 2007;25(1):135–9. 10.1016/j.gaitpost.2006.06.003. DOI: 10.1016/j.gaitpost.2006.06.003
Pierrynowski MR, Galea V. Enhancing the ability of gait analyses to differentiate between groups: scaling gait data to body size. Gait Posture. 2001;13(3):193–201. 10.1016/s0966-6362(01)00097-2. DOI: 10.1016/s0966-6362(01)00097-2
Clinical Review Report: Nusinersen (Spinraza): (Biogen Canada Inc.): Indication: Treatment of patients with 5q SMA [Internet]. Ottawa (ON): Canadian Agency for Drugs and Technologies in Health; 2018 Jan. https://www.ncbi.nlm.nih.gov/books/NBK533989/. Accessed 14 July 2021.
de Lattre C, Payan C, Vuillerot C, Rippert P, de Castro D, Bérard C, et al. Motor function measure: validation of a short form for young children with neuromuscular diseases. Arch Phys Med Rehabil. 2013;94(11):2218–26. 10.1016/j.apmr.2013.04.001. DOI: 10.1016/j.apmr.2013.04.001
Klingels K, Van Verdegem L, van den Hauwe M, Buyse G, Goemans N. Reference values for the three-minute walk test, North Star ambulatory assessment and timed tests in typically developing boys aged 2.5–5 years. Neuromuscul Disord. 2015;25:S228. 10.1016/j.nmd.2015.06.159. DOI: 10.1016/j.nmd.2015.06.159
Kolb SJ, Coffey CS, Yankey JW, Krosschell K, Arnold WD, Rutkove SB, et al. Baseline results of the NeuroNEXT spinal muscular atrophy infant biomarker study. Ann Clin Transl Neurol. 2016;3(2):132–45. 10.1002/acn3.283. DOI: 10.1002/acn3.283
Servais L, Deconinck N, Moraux A, Benali M, Canal A, Van Parys F, et al. Innovative methods to assess upper limb strength and function in non-ambulant Duchenne patients. Neuromuscul Disord. 2013;23(2):139–48. 10.1016/j.nmd.2012.10.022. DOI: 10.1016/j.nmd.2012.10.022
Lamb MM, West NA, Ouyang L, Yang M, Weitzenkamp D, James K, et al. Corticosteroid treatment and growth patterns in ambulatory males with Duchenne muscular dystrophy. J Pediatr. 2016;173:207-13.e3. 10.1016/j.jpeds.2016.02.067. DOI: 10.1016/j.jpeds.2016.02.067
