Evidence map›Paper›PMID 34948153›Full record

ReviewInternational journal of molecular sciences2021

Use of Zebrafish Models to Boost Research in Rare Genetic Diseases.

Lucie Crouzier, Elodie M Richard, Jo Sourbron, Lieven Lagae, Tangui Maurice, Benjamin Delprat

Open access · goldAbstract readReview
In one paragraph

Review in International journal of molecular sciences, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 27 papers.

0numbers the graph read from it
0cells of the map it votes in
27citing papers in PubMed
2.8field-weighted citation impact, top 9% 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

27 citing papers in PubMed, 40 citations in OpenAlex.

  1. Article
  2. Aquatic Advancements: Refining and Validating a Zebrafish Survival Blood Collection Technique.Journal of the American Association for Laboratory Animal Science : JAALAS · 2026
    Article
  3. Article
  4. Review
  5. Review
  6. Article
  7. Vertebrate and Invertebrate Animal Models for the Study of Down Syndrome.International journal of molecular sciences · 2025
    Review
  8. Article
  9. Article
  10. Review
  11. Review
  12. Review
  13. Review
  14. Review
  15. Modelling phenotypes, variants and pathomechanisms of syndromic diseases in different systems.Medizinische Genetik : Mitteilungsblatt des Berufsverbandes Medizinische Genetik e.V · 2024
    Article
  16. Article
  17. Review
  18. Review
  19. Complexity in Genetic Epilepsies: A Comprehensive Review.International journal of molecular sciences · 2023
    Review
  20. 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

6 authors at 2 institutions in 2 countries.

Lucie CrouzierMMDN, University of Montpellier, EPHE, INSERM, 34095 Montpellier, France.
Elodie M RichardMMDN, University of Montpellier, EPHE, INSERM, 34095 Montpellier, France.ORCID 0000-0002-4255-4699
Jo SourbronDepartment of Development and Regeneration, Section Pediatric Neurology, University Hospital KU Leuven, 3000 Leuven, Belgium.ORCID 0000-0002-5319-6495
Lieven LagaeDepartment of Development and Regeneration, Section Pediatric Neurology, University Hospital KU Leuven, 3000 Leuven, Belgium.
Tangui MauriceMMDN, University of Montpellier, EPHE, INSERM, 34095 Montpellier, France.ORCID 0000-0002-4074-6793
Benjamin DelpratMMDN, University of Montpellier, EPHE, INSERM, 34095 Montpellier, France.ORCID 0000-0002-2457-6923
Inserm · FRKU Leuven · BE

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Rare genetic diseases are a group of pathologies with often unmet clinical needs. Even if rare by a single genetic disease (from 1/2000 to 1/more than 1,000,000), the total number of patients concerned account for approximatively 400 million peoples worldwide. Finding treatments remains challenging due to the complexity of these diseases, the small number of patients and the challenge in conducting clinical trials. Therefore, innovative preclinical research strategies are required. The zebrafish has emerged as a powerful animal model for investigating rare diseases. Zebrafish combines conserved vertebrate characteristics with high rate of breeding, limited housing requirements and low costs. More than 84% of human genes responsible for diseases present an orthologue, suggesting that the majority of genetic diseases could be modelized in zebrafish. In this review, we emphasize the unique advantages of zebrafish models over other in vivo models, particularly underlining the high throughput phenotypic capacity for therapeutic screening. We briefly introduce how the generation of zebrafish transgenic lines by gene-modulating technologies can be used to model rare genetic diseases. Then, we describe how zebrafish could be phenotyped using state-of-the-art technologies. Two prototypic examples of rare diseases illustrate how zebrafish models could play a critical role in deciphering the underlying mechanisms of rare genetic diseases and their use to identify innovative therapeutic solutions.

Indexed as

Genetic Diseases, InbornModels, GeneticRare DiseasesZebrafishAnimalsBiomedical ResearchDisease Models, AnimalHumansanimal modelDravet syndromephenotypingrare diseasesWolfram syndromezebrafish

Identifiers

PMID34948153
PMCPMC8706563
OpenAlexW4200142723

What OpenQuestion 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 OpenQuestion graph.