Longitudinal Alterations in Gait Features in Growing Children With Duchenne Muscular Dystrophy

Ines Vandekerckhove, Marleen Van den Hauwe, Nathalie De Beukelaer, Elze Stoop, Marije Goudriaan, Margaux Delporte, Geert Molenberghs, Anja Van Campenhout, Liesbeth De Waele, Nathalie Goemans, Friedl De Groote, Kaat Desloovere*

*Corresponding author for this work

Research output: Contribution to JournalArticleAcademicpeer-review

Abstract

Prolonging ambulation is an important treatment goal in children with Duchenne muscular dystrophy (DMD). Three-dimensional gait analysis (3DGA) could provide sensitive parameters to study the efficacy of clinical trials aiming to preserve ambulation. However, quantitative descriptions of the natural history of gait features in DMD are first required. The overall goal was to provide a full delineation of the progressive gait pathology in children with DMD, covering the entire period of ambulation, by performing a so-called mixed cross-sectional longitudinal study. Firstly, to make our results comparable with previous literature, we aimed to cross-sectionally compare 31 predefined gait features between children with DMD and a typically developing (TD) database (1). Secondly, we aimed to explore the longitudinal changes in the 31 predefined gait features in growing boys with DMD using follow-up 3DGA sessions (2). 3DGA-sessions (n = 124) at self-selected speed were collected in 27 boys with DMD (baseline age: 4.6–15 years). They were repeatedly measured over a varying follow-up period (range: 6 months–5 years). The TD group consisted of 27 children (age: 5.4–15.6 years). Per measurement session, the spatiotemporal parameters, and the kinematic and kinetic waveforms were averaged over the selected gait cycles. From the averaged waveforms, discrete gait features (e.g., maxima and minima) were extracted. Mann-Whitney U tests were performed to cross-sectionally analyze the differences between DMD at baseline and TD (1). Linear mixed effect models were performed to assess the changes in gait features in the same group of children with DMD from both a longitudinal (i.e., increasing time) as well as a cross-sectional perspective (i.e., increasing baseline age) (2). At baseline, the boys with DMD differed from the TD children in 17 gait features. Additionally, 21 gait features evolved longitudinally when following-up the same boys with DMD and 25 gait features presented a significant cross-sectional baseline age-effect. The current study quantitatively described the longitudinal alterations in gait features in boys with DMD, thereby providing detailed insight into how DMD gait deteriorates. Additionally, our results highlight that gait features extracted from 3DGA are promising outcome measures for future clinical trials to quantify the efficacy of novel therapeutic strategies.

Original languageEnglish
Article number861136
Pages (from-to)1-20
Number of pages20
JournalFrontiers in Human Neuroscience
Volume16
Issue numberJune
Early online date2 Jun 2022
DOIs
Publication statusPublished - Jun 2022

Bibliographical note

Funding Information:
This work was supported by the Duchenne Parent Project NL (17.011) and by the Research Foundation - Flanders (FWO-Vlaanderen) through a research fellowship to IV (1188921N). MG was funded by the Dutch Organization for Scientific Research (NWO) VIDI grant (no. 016.156.346 FirSTeps), the European Research Council (ERC) under the European Union’s Horizon 2020 Research and Innovation Program (no. 715945 Learn2Walk), and the Johanna Kinderfonds and Kinderrevalidatie Fonds Adriaanstichting (no. 20200028).

Publisher Copyright:
Copyright © 2022 Vandekerckhove, Van den Hauwe, De Beukelaer, Stoop, Goudriaan, Delporte, Molenberghs, Van Campenhout, De Waele, Goemans, De Groote and Desloovere.

Funding

This work was supported by the Duchenne Parent Project NL (17.011) and by the Research Foundation - Flanders (FWO-Vlaanderen) through a research fellowship to IV (1188921N). MG was funded by the Dutch Organization for Scientific Research (NWO) VIDI grant (no. 016.156.346 FirSTeps), the European Research Council (ERC) under the European Union’s Horizon 2020 Research and Innovation Program (no. 715945 Learn2Walk), and the Johanna Kinderfonds and Kinderrevalidatie Fonds Adriaanstichting (no. 20200028).

FundersFunder number
Johanna Kinderfonds and Kinderrevalidatie Fonds Adriaanstichting20200028
Horizon 2020 Framework Programme715945
European Research Council
Fonds Wetenschappelijk Onderzoek
Nederlandse Organisatie voor Wetenschappelijk Onderzoek016.156.346 FirSTeps
Duchenne Parent Project17.011
Vlaamse regering1188921N

    Keywords

    • Duchenne muscular dystrophy
    • gait pattern
    • longitudinal study
    • mixed models for repeated measures
    • three-dimensional gait analysis
    • typically developing children

    Fingerprint

    Dive into the research topics of 'Longitudinal Alterations in Gait Features in Growing Children With Duchenne Muscular Dystrophy'. Together they form a unique fingerprint.

    Cite this