Evidence map›Paper›PMID 27879346›Full record

ArticleGenetics2017

A Zebrafish Model for a Human Myopathy Associated with Mutation of the Unconventional Myosin MYO18B.

Ritika Gurung, Yosuke Ono, Sarah Baxendale, Samantha Lin Chiou Lee, Steven Moore, Meredith Calvert, Philip W Ingham

Open access · bronzeAbstract read
In one paragraph

Article in Genetics, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.

0numbers the graph read from it
0cells of the map it votes in
16citing papers in PubMed
2.7field-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

16 citing papers in PubMed, 33 citations in OpenAlex.

  1. Review
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  7. Article
  8. Zebrafish models of sarcopenia.Disease models & mechanisms · 2020
    Review
  9. Defective sarcomere organization and reduced larval locomotion and fish survival in slow muscle heavy chain 1 (smyhc1) mutants.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2020
    Article
  10. Article
  11. Nemaline myopathies: a current view.Journal of muscle research and cell motility · 2019
    Review
  12. Article
  13. Article
  14. Article
  15. Review
  16. 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 5 institutions in 2 countries.

Ritika GurungA*STAR Institute of Molecular and Cell Biology, Singapore 138673, Singapore.
Yosuke OnoLee Kong Chian School of Medicine, Nanyang Technological University, Singapore 639798, Singapore.
Sarah BaxendaleDepartment of Biomedical Science, University of Sheffield, S10 2TN, UK.
Samantha Lin Chiou LeeA*STAR Institute of Molecular and Cell Biology, Singapore 138673, Singapore.
Steven MooreDepartment of Biomedical Science, University of Sheffield, S10 2TN, UK.
Meredith CalvertTemasek Lifesciences Laboratory, Singapore 117604, Singapore.
Philip W InghamA*STAR Institute of Molecular and Cell Biology, Singapore 138673, Singapore pingham@ntu.edu.sg.ORCID 0000-0001-8224-9958
Agency for Science, Technology and Research · SGInstitute of Molecular and Cell Biology · SGTemasek Life Sciences Laboratory · SGUniversity of Exeter · GBUniversity of Sheffield · GB

Funding

Medical Research Council G0100151
6 · The paper itself

Abstract

Myosin 18B is an unconventional myosin that has been implicated in tumor progression in humans. In addition, loss-of-function mutations of the MYO18B gene have recently been identified in several patients exhibiting symptoms of nemaline myopathy. In mouse, mutation of Myo18B results in early developmental arrest associated with cardiomyopathy, precluding analysis of its effects on skeletal muscle development. The zebrafish, frozen (fro) mutant was identified as one of a group of immotile mutants in the 1996 Tübingen genetic screen. Mutant embryos display a loss of birefringency in their skeletal muscle, indicative of disrupted sarcomeric organization. Using meiotic mapping, we localized the fro locus to the previously unannotated zebrafish myo18b gene, the product of which shares close to 50% identity with its human ortholog. Transcription of myo18b is restricted to fast-twitch myocytes in the zebrafish embryo; consistent with this, fro mutant embryos exhibit defects specifically in their fast-twitch skeletal muscles. We show that sarcomeric assembly is blocked at an early stage in fro mutants, leading to the disorganized accumulation of actin, myosin, and α-actinin and a complete loss of myofibrillar organization in fast-twitch muscles.

Indexed as

AnimalsMuscle Fibers, Fast-TwitchMyopathies, NemalineMyosinsZebrafishZebrafish ProteinsMyosinsZebrafish Proteinsfast-twitch musclefrozenMYO18Bnemaline myopathyzebrafish

Identifiers

PMID27879346
PMCPMC5289847
OpenAlexW2552770614

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.