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June 28, 2007DevelopmentOpen Access

Zebrafishrelatively relaxedmutants have a ryanodine receptor defect, show slow swimming and provide a model of multi-minicore disease

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Key result

Introduction of antisense morpholino oligonucleotides restored normal splicing of ryr1b and recovered swimming in zebrafish relatively relaxed mutants.

Population

Zebrafish embryos (wild-type and relatively relaxed mutants)

Comparison

Antisense morpholino oligonucleotides targeting… vs Untreated relatively relaxed mutants and…

Design

Preclinical

Authors

HHHiromi HirataTWTakaki WatanabeJHJun Hatakeyama

Discussion

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Overview

May guide preclinical antisense therapy development for RYR1 myopathies; leaves open human translation.

Structured PICO

P
Population
Zebrafish embryos (wild-type and relatively relaxed mutants) used as an animal model for multi-minicore disease.
I
Intervention
Antisense morpholino oligonucleotides targeting ryr1b aberrant splicing
C
Comparator
Untreated relatively relaxed mutants and wild-type zebrafish
O
Outcome
Swimming behavior and muscle contraction physiology (Ca2+ transients, receptor localization, ultrastructure)surrogate

Zebrafish relatively relaxed mutants provide a useful preclinical model for understanding the development and physiology of multi-minicore disease caused by RYR1 mutations.

Cite This Study

Hirata et al. (2007) studied Multi-minicore disease (MmD). Antisense morpholino oligonucleotides vs. Untreated mutants was evaluated on Swimming recovery. Introduction of antisense morpholino oligonucleotides restored normal splicing of ryr1b and recovered swimming in zebrafish relatively relaxed mutants.

synapsesocial.com/papers/6a9da44a15f03e7590bf28f9https://doi.org/10.1242/dev.004531
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Also Consider

Synapse has enriched 5 closely related papers on similar clinical questions. Consider them for comparative context:

  1. 1Structural and functional properties of ryanodine receptor type 3 in zebrafish tail muscle2015 · 21 citations
  2. 2Quantitative RyR1 reduction and loss of calcium sensitivity of RyR1Q1970fsX16+A4329D cause cores and loss of muscle strength2019 · 31 citations
  3. 3Contractile impairment and structural alterations of skeletal muscles from knockout mice lacking type 1 and type 3 ryanodine receptors1998 · 42 citations
  4. 4Gene profiling of embryonic skeletal muscle lacking type I ryanodine receptor Ca2+ release channel2016 · 24 citations
  5. 5Analysis of a zebrafish behavioral mutant reveals a dominant mutation in atp2a1/SERCA12010 · 23 citations