Evidence map›Paper›PMID 36920465›Full record

ArticleJMIR formative research2023

Smartphone and Wearable Sensors for the Estimation of Facioscapulohumeral Muscular Dystrophy Disease Severity: Cross-sectional Study.

Ahnjili Zhuparris, Ghobad Maleki, Ingrid Koopmans, Robert J Doll, Nicoline Voet, Wessel Kraaij, Adam Cohen, Emilie van Brummelen, Joris H De Maeyer, Geert Jan Groeneveld

Registry-linked trialAbstract read
In one paragraph

Article in JMIR formative research, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT04999735 (An Exploratory, Non-interventional Study to Biotype Patients With Facioscapulohumeral Muscular Dystrophy), which is not on this map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT04999735 completednot on this map

An Exploratory, Non-interventional Study to Biotype Patients With Facioscapulohumeral Muscular Dystrophy (FSHD) and Controls Using Digital Technologies

Typeobservational_patient_registrySponsorCentre for Human Drug Research, NetherlandsRan2019 to 2019Enrolled58ConditionsMuscular Dystrophy, FacioscapulohumeralArmsCHDR Monitoring Remotely (MORE), Withings Steel HR, Withings Body+ scale, Withings Blood Pressure Monitor
3 · Its place in the literature

Who cites it

4 citing papers in PubMed.

  1. Article
  2. Article
  3. Review
  4. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors.

Ahnjili ZhuparrisCentre for Human Drug Research (CHDR), Leiden, Netherlands.ORCID https://orcid.org/0000-0002-1413-1648
Ghobad MalekiCentre for Human Drug Research (CHDR), Leiden, Netherlands.ORCID https://orcid.org/0000-0003-2096-7000
Ingrid KoopmansCentre for Human Drug Research (CHDR), Leiden, Netherlands.ORCID https://orcid.org/0000-0003-4199-9781
Robert J DollCentre for Human Drug Research (CHDR), Leiden, Netherlands.ORCID https://orcid.org/0000-0002-7551-9072
Nicoline VoetDepartment of Rehabilitation, Rehabilitation Center Klimmendaal, Nijmegen, Netherlands.ORCID https://orcid.org/0000-0001-7381-2817
Wessel KraaijLeiden Institute of Advanced Computer Science, Leiden University, Leiden, Netherlands.ORCID https://orcid.org/0000-0001-7797-619X
Adam CohenCentre for Human Drug Research (CHDR), Leiden, Netherlands.ORCID https://orcid.org/0000-0001-7452-6222
Emilie van BrummelenCentre for Human Drug Research (CHDR), Leiden, Netherlands.ORCID https://orcid.org/0000-0002-6526-4790
Joris H De MaeyerFacio Therapies, Leiden, Netherlands.ORCID https://orcid.org/0000-0002-3574-6158
Geert Jan GroeneveldCentre for Human Drug Research (CHDR), Leiden, Netherlands.ORCID https://orcid.org/0000-0002-4655-6667

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundFacioscapulohumeral muscular dystrophy (FSHD) is a progressive neuromuscular disease. Its slow and variable progression makes the development of new treatments highly dependent on validated biomarkers that can quantify disease progression and response to drug interventions.

objectiveWe aimed to build a tool that estimates FSHD clinical severity based on behavioral features captured using smartphone and remote sensor data. The adoption of remote monitoring tools, such as smartphones and wearables, would provide a novel opportunity for continuous, passive, and objective monitoring of FSHD symptom severity outside the clinic.

methodsIn total, 38 genetically confirmed patients with FSHD were enrolled. The FSHD Clinical Score and the Timed Up and Go (TUG) test were used to assess FSHD symptom severity at days 0 and 42. Remote sensor data were collected using an Android smartphone, Withings Steel HR+, Body+, and BPM Connect+ for 6 continuous weeks. We created 2 single-task regression models that estimated the FSHD Clinical Score and TUG separately. Further, we built 1 multitask regression model that estimated the 2 clinical assessments simultaneously. Further, we assessed how an increasingly incremental time window affected the model performance. To do so, we trained the models on an incrementally increasing time window (from day 1 until day 14) and evaluated the predictions of the clinical severity on the remaining 4 weeks of data.

resultsThe single-task regression models achieved an R

conclusionsWe demonstrated that smartphone and remote sensor data could be used to estimate FSHD clinical severity and therefore complement the assessment of FSHD outside the clinic. In addition, our results illustrated that training the models on the first week of data allows for consistent and stable prediction of FSHD symptom severity. Longitudinal follow-up studies should be conducted to further validate the reliability and validity of the multitask model as a tool to monitor disease progression over a longer period.

trial registrationClinicalTrials.gov NCT04999735; https://www.clinicaltrials.gov/ct2/show/NCT04999735.

Indexed as

facioscapulohumeral muscular dystrophyFSHDmachine learningmHealthmobile healthmobile phoneneuromuscular diseaseregressionsmartphoneTime Up and Gowearables

Identifiers

PMID36920465
PMCPMC10131943

What OpenQuestion holds

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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.