Lymphatic function is required prenatally for lung inflation at birth
Identifieur interne : 006574 ( Ncbi/Merge ); précédent : 006573; suivant : 006575Lymphatic function is required prenatally for lung inflation at birth
Auteurs : Zoltán Jakus ; Jason P. Gleghorn ; David R. Enis ; Aslihan Sen ; Stephanie Chia ; Xi Liu ; David R. Rawnsley ; Yiqing Yang ; Paul R. Hess ; Zhiying Zou ; Jisheng Yang ; Susan H. Guttentag ; Celeste M. Nelson ; Mark L. KahnSource :
- The Journal of Experimental Medicine [ 0022-1007 ] ; 2014.
Abstract
Neonatal mice lacking lymphatic vessels due to loss of lymphangiogenic factor CCBE1 or VEGFR3 function fail to inflate their lungs, suggestive of respiratory failure in infants with congenital pulmonary lymphangiectasia.
Url:
DOI: 10.1084/jem.20132308
PubMed: 24733830
PubMed Central: 4010903
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<front><div type="abstract" xml:lang="en"><p>Neonatal mice lacking lymphatic vessels due to loss of lymphangiogenic factor CCBE1 or VEGFR3 function fail to inflate their lungs, suggestive of respiratory failure in infants with congenital pulmonary lymphangiectasia.</p>
</div>
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<pmc article-type="research-article"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">J Exp Med</journal-id>
<journal-id journal-id-type="iso-abbrev">J. Exp. Med</journal-id>
<journal-id journal-id-type="hwp">jem</journal-id>
<journal-id journal-id-type="publisher-id">jem</journal-id>
<journal-title-group><journal-title>The Journal of Experimental Medicine</journal-title>
</journal-title-group>
<issn pub-type="ppub">0022-1007</issn>
<issn pub-type="epub">1540-9538</issn>
<publisher><publisher-name>The Rockefeller University Press</publisher-name>
</publisher>
</journal-meta>
<article-meta><article-id pub-id-type="pmid">24733830</article-id>
<article-id pub-id-type="pmc">4010903</article-id>
<article-id pub-id-type="publisher-id">20132308</article-id>
<article-id pub-id-type="doi">10.1084/jem.20132308</article-id>
<article-categories><subj-group subj-group-type="hwp-journal-coll"><subject>301</subject>
</subj-group>
<subj-group subj-group-type="heading"><subject>Article</subject>
</subj-group>
</article-categories>
<title-group><article-title>Lymphatic function is required prenatally for lung inflation at birth</article-title>
<alt-title alt-title-type="short">Prenatal lymphatic function in lung inflation</alt-title>
</title-group>
<contrib-group><contrib contrib-type="author"><name><surname>Jakus</surname>
<given-names>Zoltán</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Gleghorn</surname>
<given-names>Jason P.</given-names>
</name>
<xref ref-type="aff" rid="aff2">4</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Enis</surname>
<given-names>David R.</given-names>
</name>
<xref ref-type="aff" rid="aff1">3</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Sen</surname>
<given-names>Aslihan</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Chia</surname>
<given-names>Stephanie</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Liu</surname>
<given-names>Xi</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
<xref ref-type="aff" rid="aff3">5</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Rawnsley</surname>
<given-names>David R.</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Yang</surname>
<given-names>Yiqing</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Hess</surname>
<given-names>Paul R.</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Zou</surname>
<given-names>Zhiying</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Yang</surname>
<given-names>Jisheng</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Guttentag</surname>
<given-names>Susan H.</given-names>
</name>
<xref ref-type="aff" rid="aff4">6</xref>
</contrib>
<contrib contrib-type="author"><name><surname>Nelson</surname>
<given-names>Celeste M.</given-names>
</name>
<xref ref-type="aff" rid="aff2">4</xref>
</contrib>
<contrib contrib-type="author" corresp="yes"><name><surname>Kahn</surname>
<given-names>Mark L.</given-names>
</name>
<xref ref-type="aff" rid="aff1">1</xref>
<xref ref-type="aff" rid="aff1">2</xref>
</contrib>
</contrib-group>
<aff id="aff1"><label>1</label>
<institution>Department of Medicine</institution>
,<label>2</label>
<institution>Cardiovascular Institute</institution>
, and<label>3</label>
<institution>Department of Dermatology, University of Pennsylvania, Philadelphia, PA 19104</institution>
</aff>
<aff id="aff2"><label>4</label>
<institution>Department of Chemical and Biological Engineering, Princeton University, Princeton, NJ 08544</institution>
</aff>
<aff id="aff3"><label>5</label>
<institution>Fourth Military Medical University, Xi’an 710032, China</institution>
</aff>
<aff id="aff4"><label>6</label>
<institution>Department of Neonatology, Children’s Hospital of Philadelphia, Philadelphia, PA 19104</institution>
</aff>
<author-notes><corresp>CORRESPONDENCE Mark L. Kahn: <email>markkahn@mail.med.upenn.edu</email>
</corresp>
<fn fn-type="present-address"><p>S.H. Guttentag’s present address is Dept. of Pediatrics, Vanderbilt University, Nashville, TN 37232</p>
</fn>
</author-notes>
<pub-date pub-type="ppub"><day>5</day>
<month>5</month>
<year>2014</year>
</pub-date>
<volume>211</volume>
<issue>5</issue>
<fpage>815</fpage>
<lpage>826</lpage>
<history><date date-type="received"><day>4</day>
<month>11</month>
<year>2013</year>
</date>
<date date-type="accepted"><day>19</day>
<month>3</month>
<year>2014</year>
</date>
</history>
<permissions><copyright-statement>© 2014 Jakus et al.</copyright-statement>
<copyright-year>2014</copyright-year>
<license license-type="openaccess"><license-p>This article is distributed under the terms of an Attribution–Noncommercial–Share Alike–No Mirror Sites license for the first six months after the publication date (see <ext-link ext-link-type="uri" xlink:href="http://www.rupress.org/terms">http://www.rupress.org/terms</ext-link>
). After six months it is available under a Creative Commons License (Attribution–Noncommercial–Share Alike 3.0 Unported license, as described at <ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by-nc-sa/3.0/">http://creativecommons.org/licenses/by-nc-sa/3.0/</ext-link>
).</license-p>
</license>
</permissions>
<self-uri xlink:role="icon" xlink:type="simple" xlink:href="JEM_20132308_thumb.gif"></self-uri>
<abstract abstract-type="precis"><p>Neonatal mice lacking lymphatic vessels due to loss of lymphangiogenic factor CCBE1 or VEGFR3 function fail to inflate their lungs, suggestive of respiratory failure in infants with congenital pulmonary lymphangiectasia.</p>
</abstract>
<abstract><p>Mammals must inflate their lungs and breathe within minutes of birth to survive. A key regulator of neonatal lung inflation is pulmonary surfactant, a lipoprotein complex which increases lung compliance by reducing alveolar surface tension (<xref rid="bib27" ref-type="bibr">Morgan, 1971</xref>
). Whether other developmental processes also alter lung mechanics in preparation for birth is unknown. We identify prenatal lymphatic function as an unexpected requirement for neonatal lung inflation and respiration. Mice lacking lymphatic vessels, due either to loss of the lymphangiogenic factor CCBE1 or VEGFR3 function, appear cyanotic and die shortly after birth due to failure of lung inflation. Failure of lung inflation is not due to reduced surfactant levels or altered development of the lung but is associated with an elevated wet/dry ratio consistent with edema. Embryonic studies reveal active lymphatic function in the late gestation lung, and significantly reduced total lung compliance in late gestation embryos that lack lymphatics. These findings reveal that lymphatic vascular function plays a previously unrecognized mechanical role in the developing lung that prepares it for inflation at birth. They explain respiratory failure in infants with congenital pulmonary lymphangiectasia, and suggest that inadequate late gestation lymphatic function may also contribute to respiratory failure in premature infants.</p>
</abstract>
</article-meta>
</front>
</pmc>
<affiliations><list></list>
<tree><noCountry><name sortKey="Chia, Stephanie" sort="Chia, Stephanie" uniqKey="Chia S" first="Stephanie" last="Chia">Stephanie Chia</name>
<name sortKey="Enis, David R" sort="Enis, David R" uniqKey="Enis D" first="David R." last="Enis">David R. Enis</name>
<name sortKey="Gleghorn, Jason P" sort="Gleghorn, Jason P" uniqKey="Gleghorn J" first="Jason P." last="Gleghorn">Jason P. Gleghorn</name>
<name sortKey="Guttentag, Susan H" sort="Guttentag, Susan H" uniqKey="Guttentag S" first="Susan H." last="Guttentag">Susan H. Guttentag</name>
<name sortKey="Hess, Paul R" sort="Hess, Paul R" uniqKey="Hess P" first="Paul R." last="Hess">Paul R. Hess</name>
<name sortKey="Jakus, Zoltan" sort="Jakus, Zoltan" uniqKey="Jakus Z" first="Zoltán" last="Jakus">Zoltán Jakus</name>
<name sortKey="Kahn, Mark L" sort="Kahn, Mark L" uniqKey="Kahn M" first="Mark L." last="Kahn">Mark L. Kahn</name>
<name sortKey="Liu, Xi" sort="Liu, Xi" uniqKey="Liu X" first="Xi" last="Liu">Xi Liu</name>
<name sortKey="Nelson, Celeste M" sort="Nelson, Celeste M" uniqKey="Nelson C" first="Celeste M." last="Nelson">Celeste M. Nelson</name>
<name sortKey="Rawnsley, David R" sort="Rawnsley, David R" uniqKey="Rawnsley D" first="David R." last="Rawnsley">David R. Rawnsley</name>
<name sortKey="Sen, Aslihan" sort="Sen, Aslihan" uniqKey="Sen A" first="Aslihan" last="Sen">Aslihan Sen</name>
<name sortKey="Yang, Jisheng" sort="Yang, Jisheng" uniqKey="Yang J" first="Jisheng" last="Yang">Jisheng Yang</name>
<name sortKey="Yang, Yiqing" sort="Yang, Yiqing" uniqKey="Yang Y" first="Yiqing" last="Yang">Yiqing Yang</name>
<name sortKey="Zou, Zhiying" sort="Zou, Zhiying" uniqKey="Zou Z" first="Zhiying" last="Zou">Zhiying Zou</name>
</noCountry>
</tree>
</affiliations>
</record>
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