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Sox17 is required for normal pulmonary vascular morphogenesis.

Sox17 is required for normal pulmonary vascular morphogenesis.
Sox17 is required for normal pulmonary vascular morphogenesis.
The SRY-box containing transcription factor Sox17 is required for endoderm formation and vascular morphogenesis during embryonic development. In the lung, Sox17 is expressed in mesenchymal progenitors of the embryonic pulmonary vasculature and is restricted to vascular endothelial cells in the mature lung. Conditional deletion of Sox17 in splanchnic mesenchyme-derivatives using Dermo1-Cre resulted in substantial loss of Sox17 from developing pulmonary vascular endothelial cells and caused pulmonary vascular abnormalities before birth, including pulmonary vein varices, enlarged arteries, and decreased perfusion of the microvasculature. While survival of Dermo1-Cre;Sox17?/? mice (herein termed Sox17?/?) was unaffected at E18.5, most Sox17?/? mice died by 3 weeks of age. After birth, the density of the pulmonary microvasculature was decreased in association with alveolar simplification, biventricular cardiac hypertrophy, and valvular regurgitation. The severity of the postnatal cardiac phenotype was correlated with the severity of pulmonary vasculature abnormalities. Sox17 is required for normal formation of the pulmonary vasculature and postnatal cardiovascular homeostasis.
sox17, lung, endothelial, vascular morphogenesis, dermo1-cre
0012-1606
109-120
Lange, Alexander W.
f74d0ff1-c8df-4018-a263-f256e5236cda
Haitchi, Hans Michael
68dadb29-305d-4236-884f-e9c93f4d78fe
LeCras, Timothy D.
acb90198-e9ef-4385-aa02-e43e0d81691b
Sridharan, Anusha
41c79789-05b6-4a8f-9669-acf95cfa2da5
Xu, Yan
2b4952f1-3855-4a5d-a8b0-777561a292e1
Wert, Susan E.
cdc863a5-5f42-47fd-af49-fb1e10dab174
James, Jeanne
5804be45-7208-4406-bbaf-ee316a0d83d2
Udell, Nicholas
92fe6eb1-b1ae-4a6b-a28e-05233f6fd7d7
Thurner, Philipp J.
ab711ddd-784e-48de-aaad-f56aec40f84f
Whitsett, Jeffrey A.
84fb8fc3-212e-4741-8934-d4083cd6549b
Lange, Alexander W.
f74d0ff1-c8df-4018-a263-f256e5236cda
Haitchi, Hans Michael
68dadb29-305d-4236-884f-e9c93f4d78fe
LeCras, Timothy D.
acb90198-e9ef-4385-aa02-e43e0d81691b
Sridharan, Anusha
41c79789-05b6-4a8f-9669-acf95cfa2da5
Xu, Yan
2b4952f1-3855-4a5d-a8b0-777561a292e1
Wert, Susan E.
cdc863a5-5f42-47fd-af49-fb1e10dab174
James, Jeanne
5804be45-7208-4406-bbaf-ee316a0d83d2
Udell, Nicholas
92fe6eb1-b1ae-4a6b-a28e-05233f6fd7d7
Thurner, Philipp J.
ab711ddd-784e-48de-aaad-f56aec40f84f
Whitsett, Jeffrey A.
84fb8fc3-212e-4741-8934-d4083cd6549b

Lange, Alexander W., Haitchi, Hans Michael, LeCras, Timothy D., Sridharan, Anusha, Xu, Yan, Wert, Susan E., James, Jeanne, Udell, Nicholas, Thurner, Philipp J. and Whitsett, Jeffrey A. (2014) Sox17 is required for normal pulmonary vascular morphogenesis. Developmental Biology, 387 (1), 109-120. (doi:10.1016/j.ydbio.2013.11.018). (PMID:24418654)

Record type: Article

Abstract

The SRY-box containing transcription factor Sox17 is required for endoderm formation and vascular morphogenesis during embryonic development. In the lung, Sox17 is expressed in mesenchymal progenitors of the embryonic pulmonary vasculature and is restricted to vascular endothelial cells in the mature lung. Conditional deletion of Sox17 in splanchnic mesenchyme-derivatives using Dermo1-Cre resulted in substantial loss of Sox17 from developing pulmonary vascular endothelial cells and caused pulmonary vascular abnormalities before birth, including pulmonary vein varices, enlarged arteries, and decreased perfusion of the microvasculature. While survival of Dermo1-Cre;Sox17?/? mice (herein termed Sox17?/?) was unaffected at E18.5, most Sox17?/? mice died by 3 weeks of age. After birth, the density of the pulmonary microvasculature was decreased in association with alveolar simplification, biventricular cardiac hypertrophy, and valvular regurgitation. The severity of the postnatal cardiac phenotype was correlated with the severity of pulmonary vasculature abnormalities. Sox17 is required for normal formation of the pulmonary vasculature and postnatal cardiovascular homeostasis.

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More information

Accepted/In Press date: 16 November 2013
e-pub ahead of print date: 10 January 2014
Published date: 1 March 2014
Keywords: sox17, lung, endothelial, vascular morphogenesis, dermo1-cre
Organisations: Clinical & Experimental Sciences

Identifiers

Local EPrints ID: 386152
URI: https://eprints.soton.ac.uk/id/eprint/386152
ISSN: 0012-1606
PURE UUID: 9ba6a5a4-8203-4f16-8cd3-0eaa3da52f7c
ORCID for Hans Michael Haitchi: ORCID iD orcid.org/0000-0001-8603-302X
ORCID for Philipp J. Thurner: ORCID iD orcid.org/0000-0001-7588-9041

Catalogue record

Date deposited: 28 Jan 2016 14:28
Last modified: 07 Aug 2019 00:44

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