{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,13]],"date-time":"2026-09-13T18:03:59Z","timestamp":1789322639567,"version":"build-2803163510"},"reference-count":14,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T00:00:00Z","timestamp":1786924800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T00:00:00Z","timestamp":1786924800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["obgyn.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Prenatal Diagnosis"],"published-print":{"date-parts":[[2026,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Maternal sirolimus therapy has emerged as a potential prenatal treatment for extensive fetal lymphatic malformations, particularly when associated with the risk of neonatal airway compromise. Data on prenatal indications, pharmacokinetics, and outcomes remain limited. We aimed to describe patient selection, treatment management, maternal and fetal safety, and early outcomes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Between December 2023 and May 2025, 15 fetuses with extensive cystic or mixed vascular anomalies suggestive of lymphatic malformations were referred to a national reference center. Prenatal ultrasound and fetal magnetic resonance imaging were performed. After maternal evaluation and informed consent, sirolimus was administered with weekly dose adjustments based on maternal trough levels. Tolerance, drug concentrations at delivery, obstetric outcomes, and neonatal evolution were assessed. Decrease in lesion extent was assessed using a semi\u2010quantitative 0\u20134 imaging scale.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Six fetuses received therapy after the exclusion of nine cases. Treatment began between 24 and over 30\u00a0weeks' gestation. Maternal adverse effects were mild. Transplacental drug transfer was observed with variable maternal\u2013fetal ratios. Mean gestational age at birth was 38.1\u00a0weeks. Partial or marked lesion regression occurred in five newborns.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Maternal sirolimus therapy appears feasible and well tolerated for extensive fetal lymphatic malformations, demonstrating transplacental transfer and encouraging early perinatal outcomes in carefully selected pregnancies overall.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/pd.70230","type":"journal-article","created":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T17:07:05Z","timestamp":1786986425000},"page":"1626-1636","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Maternal Sirolimus Therapy for Fetal Extensive Lymphatic Cystic Malformation: Selection Criteria, Management, and Outcomes in a Series of Six Cases"],"prefix":"10.1002","volume":"46","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5747-6654","authenticated-orcid":false,"given":"L.","family":"Guibaud","sequence":"first","affiliation":[{"name":"Pediatric and Fetal Imaging Reference Center for Superficial Vascular Anomalies H\u00f4pital Femme M\u00e8re Enfant Universit\u00e9 Claude Bernard Lyon 1  Lyon\u2010Bron France"},{"name":"INSERM U1151 Necker Enfants Malades Institute  Paris France"},{"name":"Multidisciplinary Center for Prenatal Diagnosis H\u00f4pital Femme M\u00e8re Enfant  Lyon\u2010Bron France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-1697-3777","authenticated-orcid":false,"given":"M.","family":"Bordas\u2010Fournel","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology CHU Grenoble Alpes  Nantes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0115-2586","authenticated-orcid":false,"given":"S.","family":"Cabet","sequence":"additional","affiliation":[{"name":"Pediatric and Fetal Imaging Reference Center for Superficial Vascular Anomalies H\u00f4pital Femme M\u00e8re Enfant Universit\u00e9 Claude Bernard Lyon 1  Lyon\u2010Bron France"},{"name":"Multidisciplinary Center for Prenatal Diagnosis H\u00f4pital Femme M\u00e8re Enfant  Lyon\u2010Bron France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.","family":"Le Vaillant","sequence":"additional","affiliation":[{"name":"Multidisciplinary Center for Prenatal Diagnosis CHU de Nantes  Nantes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Bruel","sequence":"additional","affiliation":[{"name":"Service de N\u00e9phro\u2010P\u00e9diatrie CHU de Nantes  Nantes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T.","family":"Lefran\u00e7ois","sequence":"additional","affiliation":[{"name":"Service d\u2019Imagerie P\u00e9diatrique CHU de Nantes  Nantes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.","family":"Megier","sequence":"additional","affiliation":[{"name":"Multidisciplinary Center for Prenatal Diagnosis H\u00f4pital Antoine\u2010B\u00e9cl\u00e8re AP\u2010HP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6045-0765","authenticated-orcid":false,"given":"A.","family":"Benachi","sequence":"additional","affiliation":[{"name":"Multidisciplinary Center for Prenatal Diagnosis H\u00f4pital Antoine\u2010B\u00e9cl\u00e8re AP\u2010HP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.","family":"Sohier","sequence":"additional","affiliation":[{"name":"Multidisciplinary Center for Prenatal Diagnosis Maternit\u00e9 Paule de Viguier CHU de Toulous  Toulouse France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3197-8290","authenticated-orcid":false,"given":"Y.","family":"Gallois","sequence":"additional","affiliation":[{"name":"ORL P\u00e9diatrique Hopital Pierre Paul Riquet CHU de Toulous  Toulouse France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T.","family":"Simon","sequence":"additional","affiliation":[{"name":"Service de N\u00e9phrologie P\u00e9diatrique H\u00f4pital des Enfants\u2010Purpan CHU de Toulous  Toulouse France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J.\u2010M.","family":"Jouannic","sequence":"additional","affiliation":[{"name":"Multidisciplinary Center for Prenatal Diagnosis H\u00f4pital Trousseau AP\u2010HP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G.","family":"Canaud","sequence":"additional","affiliation":[{"name":"INSERM U1151 Necker Enfants Malades Institute  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Guilhem","sequence":"additional","affiliation":[{"name":"Pediatric and Fetal Imaging Reference Center for Superficial Vascular Anomalies H\u00f4pital Femme M\u00e8re Enfant Universit\u00e9 Claude Bernard Lyon 1  Lyon\u2010Bron France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4805-2083","authenticated-orcid":false,"given":"A.","family":"Atallah","sequence":"additional","affiliation":[{"name":"Multidisciplinary Center for Prenatal Diagnosis H\u00f4pital Femme M\u00e8re Enfant  Lyon\u2010Bron France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Fraissenon","sequence":"additional","affiliation":[{"name":"Pediatric and Fetal Imaging Reference Center for Superficial Vascular Anomalies H\u00f4pital Femme M\u00e8re Enfant Universit\u00e9 Claude Bernard Lyon 1  Lyon\u2010Bron France"},{"name":"INSERM U1151 Necker Enfants Malades Institute  Paris France"},{"name":"Multidisciplinary Center for Prenatal Diagnosis H\u00f4pital Femme M\u00e8re Enfant  Lyon\u2010Bron France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,8,17]]},"reference":[{"key":"e_1_2_14_2_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2015\u20103257"},{"key":"e_1_2_14_3_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamadermatol.2021.3459"},{"key":"e_1_2_14_4_1","doi-asserted-by":"publisher","DOI":"10.1089\/lrb.2018.0068"},{"key":"e_1_2_14_5_1","article-title":"Management of Neonatal Extensive Cervico\u2010Facial Lymphatic Malformation Using Early Administration of Sirolimus Combined With Macro\u2010Cysts Sclerotherapies. 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