Evidence map›Paper›PMID 24760771›Full record

ArticleHuman molecular genetics2014

Fluoxetine prevents dystrophic changes in a zebrafish model of Duchenne muscular dystrophy.

Trent A Waugh, Eric Horstick, Junguk Hur, Samuel W Jackson, Ann E Davidson, Xingli Li, James J Dowling

Open access · bronzeAbstract read
In one paragraph

Article in Human molecular genetics, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 46 papers.

0numbers the graph read from it
0cells of the map it votes in
46citing papers in PubMed
3.4field-weighted citation impact, top 7% 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

46 citing papers in PubMed, 67 citations in OpenAlex.

  1. Review
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  13. Modeling neuromuscular diseases in zebrafish.Frontiers in molecular neuroscience · 2022
    Review
  14. Review
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  17. International journal of molecular sciences · 2021
    Review
  18. Article
  19. PDE10A Inhibition Reduces the Manifestation of Pathology in DMD Zebrafish and Represses the Genetic Modifier PITPNA.Molecular therapy : the journal of the American Society of Gene Therapy · 2021
    Article
  20. 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 3 institutions in 2 countries.

Trent A WaughDepartment of Pediatrics.
Eric HorstickDepartment of Pediatrics.
Junguk HurDepartment of Neurology, and.
Samuel W JacksonDepartment of Pediatrics.
Ann E DavidsonDivision of Neurology and Program in Genetics and Genome Biology, Hospital for Sick Children, Toronto, ON, Canada.
Xingli LiDepartment of Pediatrics.
James J DowlingDepartment of Pediatrics, Department of Neurology, and Department of Neuroscience, University of Michigan, Ann Arbor, MI 48109-2200, USA Division of Neurology and Program in Genetics and Genome Biology, Hospital for Sick Children, Toronto, ON, Canada Department of Paediatrics, and Department of Molecular Genetics, University of Toronto, Toronto, ON, Canada james.dowling@sickkids.ca.
Pediatrics and Genetics · USHospital for Sick Children · CAUniversity of Toronto · CA

Funding

Myotubularin and the Pathogenesis of Myotubular MyopathyK08AR054835 · NIAMS · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI DOWLING, JAMES J · 2009 to 2013
$640k
NIAMS NIH HHS K08AR054835
6 · The paper itself

Abstract

Duchenne muscular dystrophy (DMD) is a common and relentlessly progressive muscle disease. Some interventions have been identified that modestly slow progression and prolong survival, but more meaningful therapies are lacking. The goal of this study is to identify new therapeutic pathways for DMD using a zebrafish model of the disease. To accomplish this, we performed a non-biased drug screen in sapje, a zebrafish line with a recessive nonsense mutation in dystrophin. We identified 6 positive hits (out of 640 total drugs tested) by their ability to prevent abnormal birefringence in sapje. Follow-up analyses demonstrated that fluoxetine, a selective serotonin reuptake inhibitor (SSRI), provided the most substantial benefit. Morpholino-based experimentation confirmed that modulation of the serotonin pathway alone can prevent the dystrophic phenotype, and transcriptomic analysis revealed changes in calcium homeostasis as a potential mechanism. In all, we demonstrate that monoamine agonists can prevent disease in a vertebrate model of DMD. Given the safe and widespread use of SSRIs in clinical practice, our study identifies an attractive target pathway for therapy development.

Indexed as

AnimalsBase SequenceBirefringenceCalciumDisease Models, AnimalDrug Evaluation, PreclinicalDystrophinEmbryo, NonmammalianEvans BlueFluoxetineGene Expression ProfilingGene Knockdown TechniquesHomeostasisMolecular Sequence DataMorpholinosMuscular Dystrophy, AnimalCalciumDystrophinEvans BlueFluoxetineMorpholinosSerotonin Plasma Membrane Transport Proteins

Identifiers

PMID24760771
PMCPMC4119416
OpenAlexW2110237175

What OpenQuestion holds

Textmetadata
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 OpenQuestion graph.