Evidence map›Paper›PMID 39237554›Full record

ArticleScientific reports2024

Language use predicts symptoms of fragile X-associated tremor/ataxia syndrome in men and women with the FMR1 premutation.

Nell Maltman, Audra Sterling, Ellery Santos, Randi Hagerman

Abstract read
In one paragraph

Article in Scientific reports, 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. Cognitive-behavioral phenotypes inFrontiers in molecular neuroscience · 2026
    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

4 authors.

Nell MaltmanWaisman Center, University of Wisconsin-Madison, 1500 Highland Ave, Madison, WI, 53705, USA. nmaltman@arizona.edu.
Audra SterlingWaisman Center, University of Wisconsin-Madison, 1500 Highland Ave, Madison, WI, 53705, USA.
Ellery SantosMIND Institute, University of California-Davis, 2825 50th St., Sacramento, CA, 95817, USA.
Randi HagermanMIND Institute, University of California-Davis, 2825 50th St., Sacramento, CA, 95817, USA.

Funding

GENOTYPE/PHENOTYPE RELATIONSHIPS IN FRAGILE X FAMILIESR01HD036071 · NICHD · UNIVERSITY OF CALIFORNIA DAVIS · PI PAUL J HAGERMAN, RANDI J. HAGERMAN · 1998 to 2026
$13.8M
POSTDOCTORAL TRAINING IN MENTAL RETARDATION RESEARCHT32HD007489 · NICHD · UNIVERSITY OF WISCONSIN-MADISON · PI Sigan L Hartley · 1995 to 2026
$6.0M
Waisman Intellectual and Developmental Disabilities Research Center Down Syndrome RegistryU54HD090256 · NICHD · UNIVERSITY OF WISCONSIN-MADISON · PI CHANG, QIANG · 2016 to 2020
$6.0M
The relationship between language and executive function in DLD and FXS over timeR01DC019092 · NIDCD · UNIVERSITY OF WISCONSIN-MADISON · PI HOOVER, JILL, STERLING, AUDRA MARIE · 2021 to 2025
$2.7M
Eunice Kennedy Shriver National Institute of Child Health and Human Development R01HD036071-23Eunice Kennedy Shriver National Institute of Child Health and Human Development T32HD007489NICHD NIH HHS R01 HD036071NICHD NIH HHS T32 HD007489NICHD NIH HHS U54 HD090256NIDCD NIH HHS R01 DC019092
6 · The paper itself

Abstract

Fragile X-associated tremor/ataxia syndrome (FXTAS) is an age-related neurodegenerative disorder caused by a premutation of the FMR1 gene on the X chromosome. Despite the pervasive physical and cognitive effects of FXTAS, no studies have examined language in symptomatic males and females, limiting utility as an outcome measure in clinical trials of FXTAS. The goal of this work is to determine (a) the extent to which male and female FMR1 premutation carriers with FXTAS symptoms differ in their language use and (b) whether language production predicts FXTAS symptoms. Thirty-one individuals with the FMR1 premutation (21M, 10F), ages 58-85 years with some symptoms of FXTAS, were recruited from a larger cross-sectional study. Participants completed a five-minute monologic language sample. Language transcripts were assessed for rate of dysfluencies, lexical-semantics, syntax, and speech rate. Multivariable linear and ordinal regressions were used to predict FXTAS-associated symptoms, cognitive functioning, and executive functioning. Males and females did not differ in their language use. Language production predicted FXTAS symptom severity, cognitive functioning, and executive functioning. Language production difficulties may co-occur with FXTAS-associated symptoms and may be a viable outcome measure in future clinical trials, with future research needed.

Indexed as

AtaxiaFragile X Messenger Ribonucleoprotein 1Fragile X SyndromeLanguageTremorAgedAged, 80 and overCognitionCross-Sectional StudiesFemaleHumansMaleMiddle AgedFMR1 protein, humanFragile X Messenger Ribonucleoprotein 1CognitionExecutive functionFMR1Fragile X-associated tremor/ataxia syndromeLanguage

Identifiers

PMID39237554
PMCPMC11377817

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LicenceCC BY-NC-ND
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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.