Evidence map›Paper›PMID 40553144›Full record

Observational studyJournal of orofacial orthopedics = Fortschritte der Kieferorthopadie : Organ/official journal Deutsche Gesellschaft fur Kieferorthopadie2026

Oral function tests in spinal muscular atrophy: closing the diagnostic gap in severely affected adult patients : A prospective observational study.

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

Abstract readObservational Study
PubMed Full text at PMC Publisher
In one paragraph

Observational study in Journal of orofacial orthopedics = Fortschritte der Kieferorthopadie : Organ/official journal Deutsche Gesellschaft fur Kieferorthopadie, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
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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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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 http://orcid.org/0000-0002-3431-7749
Sara PortegysDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
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.
Brunhilde WirthCenter for Rare Diseases Cologne, University of Cologne, Kerpener Str. 62, 50937, Cologne, Germany.
Raoul HellerCenter for Rare Diseases Cologne, University of Cologne, Kerpener Str. 62, 50937, Cologne, Germany.
Svenja NeuhoffDepartment of Neurology and Center for Translational Neuro- and Behavioral Sciences (C-TNBS), University Hospital Essen, Hufelandstr. 55, 45147, Essen, Germany.
Tim HagenackerDepartment of Neurology and Center for Translational Neuro- and Behavioral Sciences (C-TNBS), University Hospital Essen, Hufelandstr. 55, 45147, Essen, Germany.
Bert BraumannDepartment of Orthodontics, Faculty of Medicine and University Hospital Cologne, University of Cologne, Kerpener Str. 32, 50931, Cologne, Germany.
Gilbert WunderlichCenter for Rare Diseases Cologne, University of Cologne, Kerpener Str. 62, 50937, Cologne, Germany.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeIn advanced stages of spinal muscular atrophy (SMA), established motor scores are unable to distinguish between the different degrees of remaining motor function. Bulbar muscles are affected at a later stage. The aim of the present study was to test whether oral function tests are able to better discriminate motor function than established scores and to replicate known associations between disease-related altered craniofacial anatomy and oral dysfunction in SMA.

methodsA total of 43 adult individuals with SMA (mean age 39.7 ± 12; 25 men, 18 women) were included in this prospective, cross-sectional study. Oral function was measured using a piezoelectric sensor system and an Iowa Oral Performance Instrument (IOPI) device. Data from oral function tests and established motor scores were analyzed with regard to a possible floor or ceiling effect. It was tested to what extent SMA patients with different malocclusions presented with variable scores.

resultsPatients differed in ambulatory and treatment status (15 ambulatory vs. 28 nonambulatory; 35 treated vs. 8 nontreated) and orthodontic findings (22 with a class II molar relationship and increased overjet, 35 with posterior crossbite). In contrast to the oral function tests, some of the established motor scores showed a clear floor effect. Statistically significant associations were identified between reduced oral function values and an enlarged overjet, a class II molar relationship, and a posterior crossbite. This should be taken into account in neuromuscular evaluations.

conclusionIn severely affected patients, oral function tests appear to be superior to established motor scores and fill a diagnostic gap in research and clinical practice.

Indexed as

MalocclusionMuscular Atrophy, SpinalAdultCross-Sectional StudiesFemaleHumansMaleMiddle AgedProspective StudiesReproducibility of ResultsSensitivity and SpecificityBite forceBulbar neuromuscular functionCeiling effectMalocclusionTongue pressure

Identifiers

PMID40553144
PMCPMC13275608

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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.