Evidence map›Paper›PMID 37821981›Full record

ArticleOrphanet journal of rare diseases2023

Long-term benefits of physical activity in adult patients with late onset Pompe disease: a retrospective cohort study with 10 years of follow-up.

Gamida Ismailova, Margreet A E M Wagenmakers, Esther Brusse, Ans T van der Ploeg, Marein M Favejee, Nadine A M E van der Beek, Linda E M van den Berg

Open access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Orphanet journal of rare diseases, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.7field-weighted citation impact, top 29% of its field
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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed, 4 citations in OpenAlex.

  1. Article
  2. Article
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

7 authors at 2 institutions in 1 country.

Gamida IsmailovaDepartment of Pediatrics, Center for Lysosomal and Metabolic Diseases, Erasmus Medical Center, Sophia Children's Hospital, Mailbox 2060, 3000 CB, Rotterdam, The Netherlands.
Margreet A E M WagenmakersDepartment of Internal Medicine, Center for Lysosomal and Metabolic Diseases, Erasmus Medical Center, Mailbox 2040, 3000 CA, Rotterdam, The Netherlands.
Esther BrusseDepartment of Neurology, Center for Lysosomal and Metabolic Diseases, Erasmus Medical Center, Mailbox 2040, 3000 CA, Rotterdam, The Netherlands.
Ans T van der PloegDepartment of Pediatrics, Center for Lysosomal and Metabolic Diseases, Erasmus Medical Center, Sophia Children's Hospital, Mailbox 2060, 3000 CB, Rotterdam, The Netherlands.
Marein M FavejeeDepartment of Physical Therapy, Erasmus Medical Center, Mailbox 2040, 3000 CA, Rotterdam, The Netherlands.
Nadine A M E van der BeekDepartment of Neurology, Center for Lysosomal and Metabolic Diseases, Erasmus Medical Center, Mailbox 2040, 3000 CA, Rotterdam, The Netherlands.
Linda E M van den BergDepartment of Orthopedics and Sports Medicine, Center for Lysosomal and Metabolic Diseases, Erasmus Medical Center, Sophia Children's Hospital, Mailbox 2060, 3000 CB, Rotterdam, The Netherlands. l.e.m.vandenberg@erasmusmc.nl.ORCID http://orcid.org/0000-0002-8726-1304
Erasmus MC · NLErasmus MC - Sophia Children’s Hospital · NL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundIn 2011 a 12 weeks personalized exercise training program in 23 mildly affected adult late onset Pompe patients (age 19.6-70.5 years) improved endurance, muscle strength and function. Data on long-term effects of this program or of other physical activity in Pompe disease are absent. This retrospective cohort study aimed to explore effects of long-term healthy physical activity according to the WHO norm and the former exercise training program on the disease course.

resultsA total of 29 adult late onset Pompe patients were included: 19 former exercise training program participants and 10 comparable control patients. Patients, who based on interviews, met the 2010 WHO healthy physical activity norm (active, n = 16) performed better on endurance (maximal cardiopulmonary exercise test), muscle strength and function compared to patients not meeting this norm (inactive, n = 13) (p < 0.05). Majority of the outcomes, including endurance and manually tested muscle strength, tended to be higher in the active patients of the 2011 training cohort who continued the program compared to active control patients (p > 0.05).

conclusionIn Pompe disease long-term healthy physical activity according to the 2010 WHO norm leads to physical benefits and a personalized exercise training program may have additional favorable effects and both should be recommended as standard of care.

Indexed as

Exercise TherapyGlycogen Storage Disease Type IIAdultAgedExerciseFollow-Up StudiesHumansMiddle AgedMuscle StrengthPhysical EnduranceRetrospective StudiesYoung AdultEnduranceExercise training programLate onset Pompe diseaseMuscle functionMuscle strengthWHO physical activity norm

Identifiers

PMID37821981
PMCPMC10566098
OpenAlexW4387517365

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

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.