<?xml version="1.0" encoding="utf-8"?>
<TEI xmlns="http://www.tei-c.org/ns/1.0" xmlns:xsi="http://www.w3.org/2001/XMLSchema-instance" xmlns:hal="http://hal.archives-ouvertes.fr/" xmlns:gml="http://www.opengis.net/gml/3.3/" xmlns:gmlce="http://www.opengis.net/gml/3.3/ce" version="1.1" xsi:schemaLocation="http://www.tei-c.org/ns/1.0 http://api.archives-ouvertes.fr/documents/aofr-sword.xsd">
  <teiHeader>
    <fileDesc>
      <titleStmt>
        <title>HAL TEI export of hal-01175526</title>
      </titleStmt>
      <publicationStmt>
        <distributor>CCSD</distributor>
        <availability status="restricted">
          <licence target="https://creativecommons.org/publicdomain/zero/1.0/">CC0 1.0 - Universal</licence>
        </availability>
        <date when="2026-05-20T01:26:56+02:00"/>
      </publicationStmt>
      <sourceDesc>
        <p part="N">HAL API Platform</p>
      </sourceDesc>
    </fileDesc>
  </teiHeader>
  <text>
    <body>
      <listBibl>
        <biblFull>
          <titleStmt>
            <title xml:lang="en">Multifocal Lymphangioendotheliomatosis With Thrombocytopenia: Clinical Features and Response to Sirolimus</title>
            <author role="aut">
              <persName>
                <forename type="first">Catherine</forename>
                <surname>Droitcourt</surname>
              </persName>
              <idno type="halauthorid">860044-0</idno>
              <affiliation ref="#struct-215996"/>
              <affiliation ref="#struct-2610"/>
            </author>
            <author role="aut">
              <persName>
                <forename type="first">Olivia</forename>
                <surname>Boccara</surname>
              </persName>
              <idno type="halauthorid">925618-0</idno>
            </author>
            <author role="aut">
              <persName>
                <forename type="first">Sylvie</forename>
                <surname>Fraitag</surname>
              </persName>
              <idno type="halauthorid">230713-0</idno>
              <affiliation ref="#struct-241804"/>
            </author>
            <author role="aut">
              <persName>
                <forename type="first">Géraldine</forename>
                <surname>Favrais</surname>
              </persName>
              <idno type="halauthorid">925619-0</idno>
            </author>
            <author role="aut">
              <persName>
                <forename type="first">Alain</forename>
                <surname>Dupuy</surname>
              </persName>
              <email type="md5">929b3f065eacc4ba9223dcd9d9a9e152</email>
              <email type="domain">chu-rennes.fr</email>
              <idno type="idhal" notation="numeric">1242759</idno>
              <idno type="halauthorid" notation="string">162406-1242759</idno>
              <idno type="ORCID">https://orcid.org/0000-0003-3212-7455</idno>
              <idno type="IDREF">https://www.idref.fr/074194054</idno>
              <idno type="VIAF">https://viaf.org/viaf/210208327</idno>
              <affiliation ref="#struct-2610"/>
            </author>
            <author role="aut">
              <persName>
                <forename type="first">Annabel</forename>
                <surname>Maruani</surname>
              </persName>
              <email type="md5">a19e38aeff7bca5d7a5b13b45373008c</email>
              <email type="domain">univ-tours.fr</email>
              <idno type="idhal" notation="string">annabel-maruani</idno>
              <idno type="idhal" notation="numeric">744989</idno>
              <idno type="halauthorid" notation="string">470290-744989</idno>
              <idno type="RESEARCHERID">http://www.researcherid.com/rid/L-9017-2015</idno>
              <idno type="IDREF">https://www.idref.fr/088646270</idno>
              <idno type="ORCID">https://orcid.org/0000-0003-3734-810X</idno>
              <idno type="RESEARCHERID">http://www.researcherid.com/rid/http://www.researcherid.com/rid/L-9017-2015</idno>
              <affiliation ref="#struct-360410"/>
              <affiliation ref="#struct-53727"/>
            </author>
            <editor role="depositor">
              <persName>
                <forename>Laurent</forename>
                <surname>Jonchère</surname>
              </persName>
              <email type="md5">dc1f1e9bdb3dc2065f8bf1bfd5663118</email>
              <email type="domain">univ-rennes.fr</email>
            </editor>
          </titleStmt>
          <editionStmt>
            <edition n="v1" type="current">
              <date type="whenSubmitted">2015-07-10 15:53:44</date>
              <date type="whenModified">2025-09-17 11:06:02</date>
              <date type="whenReleased">2015-07-10 15:53:44</date>
              <date type="whenProduced">2015</date>
              <ref type="externalLink" target="https://pediatrics.aappublications.org/content/pediatrics/136/2/e517.full.pdf"/>
            </edition>
            <respStmt>
              <resp>contributor</resp>
              <name key="113861">
                <persName>
                  <forename>Laurent</forename>
                  <surname>Jonchère</surname>
                </persName>
                <email type="md5">dc1f1e9bdb3dc2065f8bf1bfd5663118</email>
                <email type="domain">univ-rennes.fr</email>
              </name>
            </respStmt>
          </editionStmt>
          <publicationStmt>
            <distributor>CCSD</distributor>
            <idno type="halId">hal-01175526</idno>
            <idno type="halUri">https://univ-rennes.hal.science/hal-01175526</idno>
            <idno type="halBibtex">droitcourt:hal-01175526</idno>
            <idno type="halRefHtml">&lt;i&gt;Pediatrics&lt;/i&gt;, 2015, 136 (2), pp.e517-22. &lt;a target="_blank" href="https://dx.doi.org/10.1542/peds.2014-2410"&gt;&amp;#x27E8;10.1542/peds.2014-2410&amp;#x27E9;&lt;/a&gt;</idno>
            <idno type="halRef">Pediatrics, 2015, 136 (2), pp.e517-22. &amp;#x27E8;10.1542/peds.2014-2410&amp;#x27E9;</idno>
            <availability status="restricted"/>
          </publicationStmt>
          <seriesStmt>
            <idno type="stamp" n="UNIV-RENNES1">Université de Rennes 1</idno>
            <idno type="stamp" n="UNIV-TOURS">Université François Rabelais</idno>
            <idno type="stamp" n="APHP" corresp="INSERM">AP-HP</idno>
            <idno type="stamp" n="HL">Publications CHU Rennes</idno>
            <idno type="stamp" n="CIC">CIC-Rennes</idno>
            <idno type="stamp" n="CIC203">CIC-1414</idno>
            <idno type="stamp" n="STATS-UR1">Statistiques-HAL-UR1</idno>
            <idno type="stamp" n="UR1-UFR-SVE">UFR SVE Sciences de la vie et de l'environnement</idno>
            <idno type="stamp" n="UR1-HAL">Publications labos UR1 dans HAL-Rennes 1</idno>
            <idno type="stamp" n="UR1-SDV">UR1 - publications SDV</idno>
            <idno type="stamp" n="TEST-UNIV-RENNES">TEST Université de Rennes</idno>
            <idno type="stamp" n="TEST-UR-CSS">TEST Université de Rennes CSS</idno>
            <idno type="stamp" n="UNIV-RENNES">Université de Rennes</idno>
            <idno type="stamp" n="SORBONNE-UNIVERSITE">Sorbonne Université</idno>
            <idno type="stamp" n="SU-INF-2018" corresp="SORBONNE-UNIVERSITE">SORBONNE université &lt;2018</idno>
            <idno type="stamp" n="SU-MEDECINE" corresp="SORBONNE-UNIVERSITE">Faculté de Santé de Sorbonne Université</idno>
            <idno type="stamp" n="SU-MED" corresp="SORBONNE-UNIVERSITE">Médecine - Sorbonne Université</idno>
            <idno type="stamp" n="SU-TI">Sorbonne Université - Texte Intégral</idno>
            <idno type="stamp" n="UR1-BIO-SA">Pôle UnivRennes - Biologie-Santé</idno>
            <idno type="stamp" n="ALLIANCE-SU"> Alliance Sorbonne Université</idno>
            <idno type="stamp" n="IBRAIN">Imaging Brain &amp; Neuropsychiatry</idno>
          </seriesStmt>
          <notesStmt>
            <note type="audience" n="2">International</note>
            <note type="popular" n="0">No</note>
            <note type="peer" n="1">Yes</note>
          </notesStmt>
          <sourceDesc>
            <biblStruct>
              <analytic>
                <title xml:lang="en">Multifocal Lymphangioendotheliomatosis With Thrombocytopenia: Clinical Features and Response to Sirolimus</title>
                <author role="aut">
                  <persName>
                    <forename type="first">Catherine</forename>
                    <surname>Droitcourt</surname>
                  </persName>
                  <idno type="halauthorid">860044-0</idno>
                  <affiliation ref="#struct-215996"/>
                  <affiliation ref="#struct-2610"/>
                </author>
                <author role="aut">
                  <persName>
                    <forename type="first">Olivia</forename>
                    <surname>Boccara</surname>
                  </persName>
                  <idno type="halauthorid">925618-0</idno>
                </author>
                <author role="aut">
                  <persName>
                    <forename type="first">Sylvie</forename>
                    <surname>Fraitag</surname>
                  </persName>
                  <idno type="halauthorid">230713-0</idno>
                  <affiliation ref="#struct-241804"/>
                </author>
                <author role="aut">
                  <persName>
                    <forename type="first">Géraldine</forename>
                    <surname>Favrais</surname>
                  </persName>
                  <idno type="halauthorid">925619-0</idno>
                </author>
                <author role="aut">
                  <persName>
                    <forename type="first">Alain</forename>
                    <surname>Dupuy</surname>
                  </persName>
                  <email type="md5">929b3f065eacc4ba9223dcd9d9a9e152</email>
                  <email type="domain">chu-rennes.fr</email>
                  <idno type="idhal" notation="numeric">1242759</idno>
                  <idno type="halauthorid" notation="string">162406-1242759</idno>
                  <idno type="ORCID">https://orcid.org/0000-0003-3212-7455</idno>
                  <idno type="IDREF">https://www.idref.fr/074194054</idno>
                  <idno type="VIAF">https://viaf.org/viaf/210208327</idno>
                  <affiliation ref="#struct-2610"/>
                </author>
                <author role="aut">
                  <persName>
                    <forename type="first">Annabel</forename>
                    <surname>Maruani</surname>
                  </persName>
                  <email type="md5">a19e38aeff7bca5d7a5b13b45373008c</email>
                  <email type="domain">univ-tours.fr</email>
                  <idno type="idhal" notation="string">annabel-maruani</idno>
                  <idno type="idhal" notation="numeric">744989</idno>
                  <idno type="halauthorid" notation="string">470290-744989</idno>
                  <idno type="RESEARCHERID">http://www.researcherid.com/rid/L-9017-2015</idno>
                  <idno type="IDREF">https://www.idref.fr/088646270</idno>
                  <idno type="ORCID">https://orcid.org/0000-0003-3734-810X</idno>
                  <idno type="RESEARCHERID">http://www.researcherid.com/rid/http://www.researcherid.com/rid/L-9017-2015</idno>
                  <affiliation ref="#struct-360410"/>
                  <affiliation ref="#struct-53727"/>
                </author>
              </analytic>
              <monogr>
                <idno type="halJournalId" status="VALID">17834</idno>
                <idno type="issn">0031-4005</idno>
                <idno type="eissn">1098-4275</idno>
                <title level="j">Pediatrics</title>
                <imprint>
                  <publisher>American Academy of Pediatrics</publisher>
                  <biblScope unit="volume">136</biblScope>
                  <biblScope unit="issue">2</biblScope>
                  <biblScope unit="pp">e517-22</biblScope>
                  <date type="datePub">2015</date>
                </imprint>
              </monogr>
              <idno type="doi">10.1542/peds.2014-2410</idno>
              <idno type="pubmed">26148948</idno>
            </biblStruct>
          </sourceDesc>
          <profileDesc>
            <langUsage>
              <language ident="en">English</language>
            </langUsage>
            <textClass>
              <classCode scheme="halDomain" n="sdv">Life Sciences [q-bio]</classCode>
              <classCode scheme="halTypology" n="ART">Journal articles</classCode>
              <classCode scheme="halOldTypology" n="ART">Journal articles</classCode>
              <classCode scheme="halTreeTypology" n="ART">Journal articles</classCode>
            </textClass>
            <abstract xml:lang="en">
              <p>Multifocal lymphangioendotheliomatosis with thrombocytopenia (MLT) is a recently described glucose transporter 1-negative multifocal vascular disorder with significant morbidity and mortality. However, data are lacking on the clinical spectrum, long-term prognosis, and treatment of MLT. It is often confused with multifocal infantile hemangioma, but the conditions must be differentiated for appropriate assessment and therapeutic management. Treatments for MLT have been disappointing, and the treatments classically used for infantile hemangioma are often ineffective. We report 3 newborn cases featuring various clinical and biological phenotypes of MLT: 1 patient had severe brain involvement and died early; another had no thrombocytopenia; and the third had nearly no skin involvement. Histologically, all were negative for glucose transporter 1 and positive for the lymphatic marker lymphatic vessel endothelial hyaluronan receptor 1 or D2-40 (∼38-kDa O-linked transmembrane sialoglycoprotein podoplanin). Two cases with severe gastrointestinal bleeding were treated with sirolimus 0.1 mg/k per day, which was efficient after the first month of treatment. MLT clinically presents in various forms, and when complicated by widespread or severe extracutaneous involvement, initial aggressive therapeutic intervention is justified. The pathogenesis of MLT remains unclear, but lymphatic differentiation is widely acknowledged. Because of its antiangiogenic properties, including anti-lymphangiogenesis, sirolimus offers an adequate and targeted therapeutic approach for MLT</p>
            </abstract>
          </profileDesc>
        </biblFull>
      </listBibl>
    </body>
    <back>
      <listOrg type="structures">
        <org type="laboratory" xml:id="struct-215996" status="VALID">
          <orgName>Service de Dermatologie [Rennes] = Dermatology [Rennes]</orgName>
          <desc>
            <address>
              <addrLine>CHU Rennes, 2 Rue Henri le Guilloux, 35000 Rennes</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">https://www.chu-rennes.fr/</ref>
          </desc>
          <listRelation>
            <relation active="#struct-301327" type="direct"/>
          </listRelation>
        </org>
        <org type="laboratory" xml:id="struct-2610" status="VALID">
          <idno type="RNSR">200220115K</idno>
          <idno type="ROR">https://ror.org/02baj6743</idno>
          <orgName>Centre d'Investigation Clinique [Rennes]</orgName>
          <orgName type="acronym">CIC</orgName>
          <date type="start">2002-01-01</date>
          <desc>
            <address>
              <addrLine>Rue Henri Le Guilloux - Pavillon Clémenceau 35033 Rennes Cedex 9</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">http://www.cic-rennes.fr/</ref>
          </desc>
          <listRelation>
            <relation active="#struct-105160" type="direct"/>
            <relation active="#struct-301327" type="direct"/>
            <relation name="CIC1414" active="#struct-303623" type="direct"/>
          </listRelation>
        </org>
        <org type="laboratory" xml:id="struct-241804" status="INCOMING">
          <orgName>Cabinet de d'histopathologie cutanée</orgName>
          <desc>
            <address>
              <addrLine>Cabinet de d'histopathologie cutanée, 35, avenue Mathurin-Moreau, 75019 Paris, France</addrLine>
              <country key="FR"/>
            </address>
          </desc>
          <listRelation>
            <relation active="#struct-357974" type="direct"/>
          </listRelation>
        </org>
        <org type="institution" xml:id="struct-360410" status="VALID">
          <orgName>CHU Trousseau [APHP]</orgName>
          <desc>
            <address>
              <addrLine>26 Avenue du Dr Arnold Netter, 75012 Paris</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">http://www.aphp.fr/contenu/hopital-armand-trousseau-1</ref>
          </desc>
          <listRelation>
            <relation active="#struct-300068" type="direct"/>
            <relation active="#struct-413221" type="direct"/>
          </listRelation>
        </org>
        <org type="laboratory" xml:id="struct-53727" status="VALID">
          <idno type="IdRef">228879965</idno>
          <idno type="RNSR">201220230S</idno>
          <idno type="ROR">https://ror.org/01eem7c55</idno>
          <orgName>Imaging, Brain &amp; Neuropsychiatry</orgName>
          <orgName type="acronym">iBraiN</orgName>
          <date type="start">1988-01-01</date>
          <desc>
            <address>
              <addrLine>10 Bd TonnelléBât. Thérèse Planiol37032 Tours Cedex 1</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">https://ibrain.univ-tours.fr/</ref>
          </desc>
          <listRelation>
            <relation name="UMR U1253" active="#struct-300298" type="direct"/>
            <relation name="UMR U1253" active="#struct-303623" type="direct"/>
          </listRelation>
        </org>
        <org type="institution" xml:id="struct-301327" status="VALID">
          <idno type="ROR">https://ror.org/05qec5a53</idno>
          <orgName>Centre Hospitalier Universitaire de Rennes [CHU Rennes] = Rennes University Hospital [Pontchaillou]</orgName>
          <desc>
            <address>
              <addrLine>2 Rue Henri le Guilloux, 35000 Rennes</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">https://www.chu-rennes.fr/</ref>
          </desc>
        </org>
        <org type="institution" xml:id="struct-105160" status="VALID">
          <idno type="IdRef">26693823X</idno>
          <idno type="ROR">https://ror.org/015m7wh34</idno>
          <orgName>Université de Rennes</orgName>
          <orgName type="acronym">UR</orgName>
          <desc>
            <address>
              <addrLine>Campus de Beaulieu, 263 avenue Général Leclerc, CS 74205, 35042 RENNES CEDEX</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">https://www.univ-rennes.fr/</ref>
          </desc>
        </org>
        <org type="institution" xml:id="struct-303623" status="VALID">
          <idno type="IdRef">026388278</idno>
          <idno type="ROR">https://ror.org/02vjkv261</idno>
          <orgName>Institut National de la Santé et de la Recherche Médicale</orgName>
          <orgName type="acronym">INSERM</orgName>
          <desc>
            <address>
              <addrLine>101, rue de Tolbiac, 75013 Paris</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">http://www.inserm.fr</ref>
          </desc>
        </org>
        <org type="institution" xml:id="struct-357974" status="INCOMING">
          <orgName>Cabinet de d'histopathologie cutanée</orgName>
          <desc>
            <address>
              <country key="FR"/>
            </address>
          </desc>
        </org>
        <org type="regroupinstitution" xml:id="struct-300068" status="VALID">
          <idno type="ROR">https://ror.org/00pg5jh14</idno>
          <orgName>Assistance publique - Hôpitaux de Paris (AP-HP)</orgName>
          <orgName type="acronym">AP-HP</orgName>
          <desc>
            <address>
              <country key="FR"/>
            </address>
          </desc>
        </org>
        <org type="regroupinstitution" xml:id="struct-413221" status="VALID">
          <idno type="IdRef">221333754</idno>
          <idno type="ROR">https://ror.org/02en5vm52</idno>
          <orgName>Sorbonne Université</orgName>
          <orgName type="acronym">SU</orgName>
          <date type="start">2018-01-01</date>
          <desc>
            <address>
              <addrLine>21 rue de l’École de médecine - 75006 Paris</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">http://www.sorbonne-universite.fr/</ref>
          </desc>
        </org>
        <org type="institution" xml:id="struct-300298" status="VALID">
          <idno type="IdRef">026404478</idno>
          <idno type="ISNI">0000000121608742</idno>
          <idno type="ROR">https://ror.org/02wwzvj46</idno>
          <orgName>Université de Tours</orgName>
          <orgName type="acronym">UT</orgName>
          <date type="start">1970-12-17</date>
          <desc>
            <address>
              <addrLine>60 rue du Plat d'Étain, 37020 Tours cedex 1</addrLine>
              <country key="FR"/>
            </address>
            <ref type="url">http://www.univ-tours.fr</ref>
          </desc>
        </org>
      </listOrg>
    </back>
  </text>
</TEI>