Evidence map›Paper›PMID 39407277›Full record

ArticleOrphanet journal of rare diseases2024

Oral functions in adult persons with spinal muscular atrophy compared to a healthy control group: a prospective cross-sectional study with a multimodal approach.

Teresa Kruse, Diana Leflerovà, Annette Cap, Sara Portegys, Brunhilde Wirth, Raoul Heller, Svenja Brakemeier, Tim Hagenacker, Bert Braumann, Gilbert Wunderlich

Abstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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.

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

1 citing paper in PubMed.

  1. 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.

Teresa KruseDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany. teresa.kruse@uk-koeln.de.ORCID 0000-0002-3431-7749
Diana LeflerovàDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
Annette CapDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
Sara PortegysDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
Brunhilde WirthDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
Raoul HellerDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
Svenja BrakemeierDepartment of Neurology and Center for Translational Neuro- and Behavioral Sciences (C-TNBS), University Hospital Essen, Essen, Germany.
Tim HagenackerDepartment of Neurology and Center for Translational Neuro- and Behavioral Sciences (C-TNBS), University Hospital Essen, Essen, Germany.
Bert BraumannDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
Gilbert WunderlichDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundOral function tests have been shown to reliably detect impaired bulbar function in adults with spinal muscular atrophy (SMA). Although not routinely recorded, it is known that persons with SMA are affected to varying degrees. Detecting differences in bite and tongue force, endurance, and maximum mouth opening has become particularly promising since the introduction of causal therapy for SMA. This study aimed to compare oral function among adult persons with SMA with different SMA types, walking abilities, and treatment status to a healthy control group.

methodsData from oral function tests conducted on 58 persons with SMA and 45 healthy individuals were analyzed. Differences in oral function between SMA subgroups were pairwise tested and compared to the healthy control group using Wilcoxon rank sum tests.

resultsIn an overall comparison, three out of five oral function tests revealed lower values for the SMA group compared to the control group. Subgroup analyses indicated lower scores for most oral function tests in non-ambulatory, untreated patients with SMA type 2 compared to controls. Ambulatory, treated patients with SMA type 3 achieved strength and endurance values comparable to those of healthy individuals.

conclusionsThe impairment of oral function varies across persons with SMA. Routine measurement of oral function is warranted to determine individual bulbar involvement stages. Further evaluation should be scheduled if indicators such as restricted maximum mouth opening arise. Trial registration DRKS, DRKS00015842. Registered 30 July 2019, https://drks.de/register/de/trial/DRKS00015842/preview .

Indexed as

Muscular Atrophy, SpinalAdolescentAdultCross-Sectional StudiesFemaleHumansMaleMiddle AgedProspective StudiesYoung AdultBite forceBulbar neuromuscular functionEnduranceOutcome measuresSpinal muscular atrophyTongue pressure

Identifiers

PMID39407277
PMCPMC11481369

What OpenQuestion holds

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LicenceCC BY
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Registered trials

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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.