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A requirement for Zic2 in the regulation of nodal expression underlies the establishment of left-sided identity

Abstract:

ZIC2 mutation is known to cause holoprosencephaly (HPE). A subset of ZIC2 HPE probands harbour cardiovascular and visceral anomalies suggestive of laterality defects. 3D-imaging of novel mouse Zic2 mutants uncovers, in addition to HPE, laterality defects in lungs, heart, vasculature and viscera. A strong bias towards right isomerism indicates a failure to establish left identity in the lateral plate mesoderm (LPM), a phenotype that cannot be explained simply by the defective ciliogenesis prev...

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Publication status:
Published
Peer review status:
Peer reviewed
Version:
Publisher's Version

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Publisher copy:
10.1038/s41598-018-28714-1

Authors


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Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
RDM
Subgroup:
RDM Cardiovascular Medicine
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
RDM
Subgroup:
RDM Cardiovascular Medicine
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
RDM
Subgroup:
RDM Cardiovascular Medicine
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
RDM
Subgroup:
RDM Cardiovascular Medicine
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
RDM
Subgroup:
RDM Cardiovascular Medicine
ORCID:
0000-0002-7007-3649
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Funding agency for:
Schneider, JE
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Funding agency for:
Bhattacharya, S
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Funding agency for:
Bhattacharya, S
Publisher:
Springer Nature Publisher's website
Journal:
Scientific reports Journal website
Volume:
8
Pages:
Article: 10439
Publication date:
2018-07-11
Acceptance date:
2018-06-25
DOI:
EISSN:
2045-2322
Pubs id:
pubs:868960
URN:
uri:b615d629-ab40-4809-9a25-22095b00d71d
UUID:
uuid:b615d629-ab40-4809-9a25-22095b00d71d
Local pid:
pubs:868960
Language:
English

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