Shixiao Dong, Li Li, Jiaosheng Xu, Zhonglong Han, Xia Zheng
{"title":"西罗莫司对危及生命的新生儿淋巴管畸形反应迅速,耐受性良好。","authors":"Shixiao Dong, Li Li, Jiaosheng Xu, Zhonglong Han, Xia Zheng","doi":"10.1002/ped4.12397","DOIUrl":null,"url":null,"abstract":"<p><strong>Introduction: </strong>Lymphatic malformations (LMs) are rare vascular anomalies predominantly affecting infants, which can be debilitating or life-threatening when complicated with intralesional bleeding or infection. Effective and safe management strategies are essential in such cases.</p><p><strong>Case presentation: </strong>We report a case series involving four Chinese neonates with life-threatening LMs, initially treated with oral sirolimus. All patients achieved rapid relief and sustained remission, using a lower sirolimus dosage than previously recommended. Furthermore, adverse events were rarely recorded during follow-up.</p><p><strong>Conclusion: </strong>Sirolimus can be considered a promising choice for neonates with intricate and life-threatening LMs. Initiation with a reduced sirolimus dose is advisable.</p>","PeriodicalId":19992,"journal":{"name":"Pediatric Investigation","volume":"7 3","pages":"206-211"},"PeriodicalIF":1.9000,"publicationDate":"2023-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/65/f0/PED4-7-206.PMC10509387.pdf","citationCount":"0","resultStr":"{\"title\":\"Rapid response with good toleration of sirolimus for life-threatening neonatal lymphatic malformations.\",\"authors\":\"Shixiao Dong, Li Li, Jiaosheng Xu, Zhonglong Han, Xia Zheng\",\"doi\":\"10.1002/ped4.12397\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Introduction: </strong>Lymphatic malformations (LMs) are rare vascular anomalies predominantly affecting infants, which can be debilitating or life-threatening when complicated with intralesional bleeding or infection. Effective and safe management strategies are essential in such cases.</p><p><strong>Case presentation: </strong>We report a case series involving four Chinese neonates with life-threatening LMs, initially treated with oral sirolimus. All patients achieved rapid relief and sustained remission, using a lower sirolimus dosage than previously recommended. Furthermore, adverse events were rarely recorded during follow-up.</p><p><strong>Conclusion: </strong>Sirolimus can be considered a promising choice for neonates with intricate and life-threatening LMs. Initiation with a reduced sirolimus dose is advisable.</p>\",\"PeriodicalId\":19992,\"journal\":{\"name\":\"Pediatric Investigation\",\"volume\":\"7 3\",\"pages\":\"206-211\"},\"PeriodicalIF\":1.9000,\"publicationDate\":\"2023-08-30\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/65/f0/PED4-7-206.PMC10509387.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pediatric Investigation\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1002/ped4.12397\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2023/9/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q2\",\"JCRName\":\"PEDIATRICS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Investigation","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1002/ped4.12397","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2023/9/1 0:00:00","PubModel":"eCollection","JCR":"Q2","JCRName":"PEDIATRICS","Score":null,"Total":0}
Rapid response with good toleration of sirolimus for life-threatening neonatal lymphatic malformations.
Introduction: Lymphatic malformations (LMs) are rare vascular anomalies predominantly affecting infants, which can be debilitating or life-threatening when complicated with intralesional bleeding or infection. Effective and safe management strategies are essential in such cases.
Case presentation: We report a case series involving four Chinese neonates with life-threatening LMs, initially treated with oral sirolimus. All patients achieved rapid relief and sustained remission, using a lower sirolimus dosage than previously recommended. Furthermore, adverse events were rarely recorded during follow-up.
Conclusion: Sirolimus can be considered a promising choice for neonates with intricate and life-threatening LMs. Initiation with a reduced sirolimus dose is advisable.