Outcome measures of instrumented gait analysis in hereditary spastic paraplegia: a systematic review

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Abstract

Background

Hereditary spastic paraplegias (HSPs) comprise a group of genetic movement disorders characterized by progressive spasticity and weakness of the lower limbs leading to gait deficits. Instrumented gait measures are applied to quantify gait patterns in HSP objectively. However, there is no consensus on the most relevant HSP-specific digital outcome measures for future clinical studies.

Aim

This systematic review aims to summarize outcome measures of instrumented gait analysis in HSP patients, focusing on both traditional motion capture (MOCAP) and inertial sensor systems.

Methods

Following PRISMA-2020 guidelines, a comprehensive literature search was conducted in PubMed, Scopus, and Web of Science to identify studies using instrumented gait analysis in HSP. Data on participant characteristics, measurement systems, outcome measures, results, and risk of bias were systematically extracted.

Results

In total, 38 studies published between 2004 and 2024, including 29 observational studies and 9 interventional studies, met the inclusion criteria. Various gait parameters were used, including spatio-temporal, kinematic, kinetic, and electromyography (EMG) parameters. Walking speed and range-of-motion (ROM) knee were identified as important parameters for differentiating HSP patients from healthy controls, but these parameters are more general rather than disease-specific. Foot lift, ROM foot, and gait variability are promising, more disease-specific parameters, as they reflect disease severity and increased balance deficits. However, a deeper understanding of all gait parameter categories is necessary, particularly for the upper body. Few studies explored sub-cohorts that exhibit different HSP gait characteristics.

Conclusion

While MOCAP provides valuable data in controlled hospital environments, there is a need for validated mobile sensor systems capturing the gait patterns of HSP patients in real-life without supervision. Future research must focus on better longitudinal multicenter studies with larger sample sizes to establish robust digital outcomes and monitor disease progression and therapeutic response in HSP.

Supplementary Information

The online version contains supplementary material available at 10.1186/s12984-025-01646-4.

Related collections

Most cited references 61

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QUADAS-2: a revised tool for the quality assessment of diagnostic accuracy studies.

Penny F Whiting, Anne W S Rutjes, Marie E Westwood … (2011)

In 2003, the QUADAS tool for systematic reviews of diagnostic accuracy studies was developed. Experience, anecdotal reports, and feedback suggested areas for improvement; therefore, QUADAS-2 was developed. This tool comprises 4 domains: patient selection, index test, reference standard, and flow and timing. Each domain is assessed in terms of risk of bias, and the first 3 domains are also assessed in terms of concerns regarding applicability. Signalling questions are included to help judge risk of bias. The QUADAS-2 tool is applied in 4 phases: summarize the review question, tailor the tool and produce review-specific guidance, construct a flow diagram for the primary study, and judge bias and applicability. This tool will allow for more transparent rating of bias and applicability of primary diagnostic accuracy studies.

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PRISMA 2020 explanation and elaboration: updated guidance and exemplars for reporting systematic reviews

Matthew J Page (2021)

The methods and results of systematic reviews should be reported in sufficient detail to allow users to assess the trustworthiness and applicability of the review findings. The Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) statement was developed to facilitate transparent and complete reporting of systematic reviews and has been updated (to PRISMA 2020) to reflect recent advances in systematic review methodology and terminology. Here, we present the explanation and elaboration paper for PRISMA 2020, where we explain why reporting of each item is recommended, present bullet points that detail the reporting recommendations, and present examples from published reviews. We hope that changes to the content and structure of PRISMA 2020 will facilitate uptake of the guideline and lead to more transparent, complete, and accurate reporting of systematic reviews.

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Effects of walking speed on gait biomechanics in healthy participants: a systematic review and meta-analysis

Claudiane Arakaki Fukuchi, Reginaldo Kisho Fukuchi, Marcos Duarte (2019)

Background Understanding the effects of gait speed on biomechanical variables is fundamental for a proper evaluation of alterations in gait, since pathological individuals tend to walk slower than healthy controls. Therefore, the aim of the study was to perform a systematic review of the effects of gait speed on spatiotemporal parameters, joint kinematics, joint kinetics, and ground reaction forces in healthy children, young adults, and older adults. Methods A systematic electronic search was performed on PubMed, Embase, and Web of Science databases to identify studies published between 1980 and 2019. A modified Quality Index was applied to assess methodological quality, and effect sizes with 95% confidence intervals were calculated as the standardized mean differences. For the meta-analyses, a fixed or random effect model and the statistical heterogeneity were calculated using the I 2 index. Results Twenty original full-length studies were included in the final analyses with a total of 587 healthy individuals evaluated, of which four studies analyzed the gait pattern of 227 children, 16 studies of 310 young adults, and three studies of 59 older adults. In general, gait speed affected the amplitude of spatiotemporal gait parameters, joint kinematics, joint kinetics, and ground reaction forces with a decrease at slow speeds and increase at fast speeds in relation to the comfortable speed. Specifically, moderate-to-large effect sizes were found for each age group and speed: children (slow, − 3.61 to 0.59; fast, − 1.05 to 2.97), young adults (slow, − 3.56 to 4.06; fast, − 4.28 to 4.38), and older adults (slow, − 1.76 to 0.52; fast, − 0.29 to 1.43). Conclusions This review identified that speed affected the gait patterns of different populations with respect to the amplitude of spatiotemporal parameters, joint kinematics, joint kinetics, and ground reaction forces. Specifically, most of the values analyzed decreased at slower speeds and increased at faster speeds. Therefore, the effects of speed on gait patterns should also be considered when comparing the gait analysis of pathological individuals with normal or control ones. Electronic supplementary material The online version of this article (10.1186/s13643-019-1063-z) contains supplementary material, which is available to authorized users.

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Author and article information

Contributors

Heiko Gassner:

ORCID: http://orcid.org/0000-0003-2037-9460

heiko.gassner@uk-erlangen.de

Journal

Journal ID (nlm-ta): J Neuroeng Rehabil

Journal ID (iso-abbrev): J Neuroeng Rehabil

Title: Journal of NeuroEngineering and Rehabilitation

Publisher: BioMed Central (London )

ISSN (Electronic): 1743-0003

Publication date (Electronic): 5 June 2025

Publication date PMC-release: 5 June 2025

Publication date Collection: 2025

Volume: 22

Electronic Location Identifier: 129

Affiliations

[1 ]Fraunhofer Institute for Integrated Circuits IIS, ( https://ror.org/024ape423) Erlangen, Germany

[2 ]Machine Learning and Data Analytics Lab, Department Artificial Intelligence in Biomedical, Engineering (AIBE), Friedrich-Alexander-Universität Erlangen-Nürnberg (FAU), ( https://ror.org/00f7hpc57) Erlangen, Germany

[3 ]Department of Molecular Neurology, University Hospital Erlangen, Friedrich-Alexander-Universität, Erlangen-Nürnberg (FAU), ( https://ror.org/00f7hpc57) Erlangen, Germany

[4 ]Center for Rare Diseases Erlangen (ZSEER), University Hospital Erlangen, ( https://ror.org/0030f2a11) Erlangen, Germany

[5 ]Translational Digital Health Group, Institute of AI for Health, German Research Center for Environmental Health, Helmholtz Zentrum München, ( https://ror.org/00cfam450) Neuherberg, Germany

Author information

Veronika Koch http://orcid.org/0000-0002-2246-7686

Juergen Winkler http://orcid.org/0000-0003-0630-9204

Bjoern Eskofier http://orcid.org/0000-0002-0417-0336

Martin Regensburger http://orcid.org/0000-0002-2172-7386

Heiko Gassner http://orcid.org/0000-0003-2037-9460

Article

Publisher ID: 1646

DOI: 10.1186/s12984-025-01646-4

PMC ID: 12139076

PubMed ID: 40474269

SO-VID: 3413ce89-50c9-43ca-83f0-c86ae993bcf7

License:

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.

History

Date received : 23 January 2025

Date accepted : 12 May 2025

Funding

Funded by: TreatHSP consortium

Award ID: 01GM2209B

Funded by: Deutsche Forschungsgemeinschaft (DFG) collaborative research center EmpkinS

Award ID: CRC 1483

Funded by: EU-wide ERAPerMed project DIGIPD

Award ID: 01KU2110

Award Recipient : Juergen Winkler

Funded by: Fraunhofer Internal Programs

Award ID: Attract 044-602140

Award Recipient : Heiko Gassner

Funded by: Fraunhofer-Institut für Integrierte Schaltungen IIS (1050)

Open Access :

Open Access funding enabled and organized by Projekt DEAL.

Custom metadata

ScienceOpen disciplines: Neurosciences

Keywords: hereditary spastic paraplegias,motion capture,gait analysis,sensors,mobile sensor systems,inertial measurement unit

Data availability: