PUBLICATION

De novo phosphoinositide synthesis in zebrafish is required for triad formation but not essential for myogenesis

Authors
Smith, L., Fabian, L., Al-Maawali, A., Noche, R.R., Dowling, J.J.
ID
ZDB-PUB-200818-16
Date
2020
Source
PLoS One   15: e0231364 (Journal)
Registered Authors
Dowling, Jim, Noche, Ramil
Keywords
none
MeSH Terms
  • Muscles/metabolism
  • Inositol Phosphates/metabolism
  • CDP-Diacylglycerol-Inositol 3-Phosphatidyltransferase/biosynthesis
  • CDP-Diacylglycerol-Inositol 3-Phosphatidyltransferase/metabolism*
  • Zebrafish/metabolism
  • Phosphatidylinositols/biosynthesis*
  • Phosphatidylinositols/metabolism*
  • Muscle Development/genetics
  • Zebrafish Proteins/metabolism
  • Animals
  • Lipogenesis
(all 11)
PubMed
32804943 Full text @ PLoS One
Abstract
Phosphoinositides (PIPs) and their regulatory enzymes are key players in many cellular processes and are required for aspects of vertebrate development. Dysregulated PIP metabolism has been implicated in several human diseases, including a subset of skeletal myopathies that feature structural defects in the triad. The role of PIPs in skeletal muscle formation, and particularly triad biogenesis, has yet to be determined. CDP-diacylglycerol-inositol 3-phosphatidyltransferase (CDIPT) catalyzes the formation of phosphatidylinositol, which is the base of all PIP species. Loss of CDIPT should, in theory, result in the failure to produce PIPs, and thus provide a strategy for establishing the requirement for PIPs during embryogenesis. In this study, we generated cdipt mutant zebrafish and determined the impact on skeletal myogenesis. Analysis of cdipt mutant muscle revealed no apparent global effect on early muscle development. However, small but significant defects were observed in triad size, with T-tubule area, inter terminal cisternae distance and gap width being smaller in cdipt mutants. This was associated with a decrease in motor performance. Overall, these data suggest that myogenesis in zebrafish does not require de novo PIP synthesis but does implicate a role for CDIPT in triad formation.
Genes / Markers
Figures
Figure Gallery (6 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
cdipthsc158/hsc158standard conditionsProtruding-mouth
cdipthsc158/hsc158standard conditionsProtruding-mouth
cdipthsc158/hsc158standard conditionsProtruding-mouth
cdipthsc158/hsc158standard conditionsProtruding-mouth
cdipthsc158/hsc158standard conditionsProtruding-mouth
cdipthsc158/hsc158standard conditionsProtruding-mouth
cdipthsc158/hsc158standard conditionsProtruding-mouth
cdipthsc158/hsc158standard conditionsDay 5
cdipthsc158/hsc158standard conditionsDay 6
cdipthsc158/hsc158standard conditionsDay 6
1 - 10 of 19
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
hi559TgTransgenic Insertion
hsc158
    		Small Deletion
    1 - 2 of 2
    Show
    Human Disease / Model
    No data available
    Sequence Targeting Reagents
    Target Reagent Reagent Type
    cdiptCRISPR1-cdiptCRISPR
    cdiptMO6-cdiptMRPHLNO
    1 - 2 of 2
    Show
    Fish
    Fish
    cdipthsc158/hsc158
    WT
    WT + MO6-cdipt
    1 - 3 of 3
    Show
    Antibodies
    No data available
    Orthology
    No data available
    Engineered Foreign Genes
    No data available
    Mapping
    No data available
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    Citation
    Smith, L., Fabian, L., Al-Maawali, A., Noche, R.R., Dowling, J.J. (2020) De novo phosphoinositide synthesis in zebrafish is required for triad formation but not essential for myogenesis. PLoS One. 15:e0231364.