Evidence map›Paper›PMID 39885828›Full record

ArticleDisease models & mechanisms2025

The behavioural consequences of dystrophinopathy.

Minou A T Verhaeg, Elizabeth M van der Pijl, Davy van de Vijver, Christa L Tanganyika-de Winter, Tiberiu L Stan, Angel van Uffelen, Luciano Censoni, Maaike van Putten

Abstract read
In one paragraph

Article in Disease models & mechanisms, 2025. 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
–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

2 citing papers in PubMed.

  1. Article
  2. 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

8 authors.

Minou A T VerhaegDepartment of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.ORCID 0000-0001-9142-0972
Elizabeth M van der PijlDepartment of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.
Davy van de VijverDepartment of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.
Christa L Tanganyika-de WinterDepartment of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.
Tiberiu L StanDepartment of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.
Angel van UffelenDepartment of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.
Luciano CensoniThe Group for Integrative Neurophysiology, Department of Medical and Translational Biology, Umeå University, 901 87 Umeå, Sweden.
Maaike van PuttenDepartment of Human Genetics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands.ORCID 0000-0002-0683-8897

Funding

Horizon 2020 Framework Programme 847826Leids Universitair Medisch Centrum
6 · The paper itself

Abstract

Duchenne muscular dystrophy is a severe neuromuscular disorder, caused by mutations in the DMD gene. Normally, the DMD gene gives rise to many dystrophin isoforms, of which multiple are expressed in the brain. The location of the mutation determines the number of dystrophin isoforms affected, and the absence thereof leads to behavioral and cognitive impairments. Even though behavioral studies have thoroughly investigated the effects of the loss of Dp427, and to a lesser extent of Dp140, in mice, direct comparisons between models lacking multiple dystrophin isoforms are sparse. Furthermore, a behavioral characterization of the DMD-null mouse, which lacks all dystrophin isoforms, has never been undertaken. Using a wide variety of behavioral tests, we directly compared impairments between mdx5cv, mdx52 and DMD-null mice. We confirmed the role of Dp427 in emotional reactivity. We did not find any added effects of loss of Dp140 on fear, but showed the involvement of Dp140 in spontaneous behavior, specifically in habituation and activity changes due to light/dark switches. Lastly, our results indicate that Dp71/Dp40 play an important role in many behavioral domains, including anxiety and spontaneous behavior.

Indexed as

Behavior, AnimalDystrophinMuscular Dystrophy, DuchenneAnimalsAnxietyFearHabituation, PsychophysiologicMaleMiceMice, Inbred C57BLMice, Inbred mdxMice, KnockoutMotor ActivityDystrophinAnxietyCognitionDystrophinLearningMdx52 and DMD-nullMdx5cvSocial interactionSpontaneous behavior

Identifiers

PMID39885828
PMCPMC11911635

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

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 Socratic graph.