Early Adjuvant Medication With the mTOR Inhibitor Sirolimus in a Preterm Neonate With Compressive Cystic Lymphatic Malformation

被引:9
作者
Honnorat, Marion [1 ]
Viremouneix, Loic [2 ,3 ]
Ayari, Sonia [4 ]
Guibaud, Laurent [2 ,3 ]
Coste, Karen [5 ,6 ]
Claris, Olivier [1 ,7 ]
Butin, Marine [1 ,8 ]
机构
[1] Hosp Civils Lyon, Serv Reanimat Neonatale, HFME, Bron, France
[2] Hosp Civils Lyon, Ctr Competence Anomalies Vasc Superficielles FAVA, HFME, Bron, France
[3] Hosp Civils Lyon, Serv Radiol Pediat, HFME, Bron, France
[4] Hosp Civils Lyon, Serv Chirurg ORL, HFME, Bron, France
[5] CHU ESTAING, Serv Reanimat Pediat & Neonatale, Clermont Ferrand, France
[6] Univ Clermont Auvergne, GReD, INSERM, CNRS,UMR 6293,U1103, Clermont Ferrand, France
[7] Univ Claude Bernard, Villeurbanne, France
[8] INSERM, Ctr Int Rech Infectiol, Equipe Pathogenese Infect Staphyloc, U1111, Lyon, France
关键词
lymphangioma; preterm; sirolimus; sclerotherapy; mTOR inhibitor;
D O I
10.3389/fped.2020.00418
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Cystic lymphatic malformations result from an abnormal embryological development of the lymphatic structures. Here we report on a case of a preterm female baby, born at 34 weeks of gestation, with a voluminous cervicofacial cystic lymphatic malformation responsible for an airway obstruction. An mTOR inhibitor, sirolimus, was started from the first day of life, and was combined with iterative sclerotherapy procedures. This case illustrates a safe and successful early administration of sirolimus in a preterm neonate.
引用
收藏
页数:5
相关论文
共 17 条
[1]   Efficacy and Safety of Sirolimus in the Treatment of Complicated Vascular Anomalies [J].
Adams, Denise M. ;
Trenor, Cameron C., III ;
Hammill, Adrienne M. ;
Vinks, Alexander A. ;
Patel, Manish N. ;
Chaudry, Gulraiz ;
Wentzel, Mary Sue ;
Mobberley-Schuman, Paula S. ;
Campbell, Lisa M. ;
Brookbank, Christine ;
Gupta, Anita ;
Chute, Carol ;
Eile, Jennifer ;
McKenna, Jesse ;
Merrow, Arnold C. ;
Fei, Lin ;
Hornung, Lindsey ;
Seid, Michael ;
Dasgupta, A. Roshni ;
Dickie, Belinda H. ;
Elluru, Ravindhra G. ;
Lucky, Anne W. ;
Weiss, Brian ;
Azizkhan, Richard G. .
PEDIATRICS, 2016, 137 (02)
[2]   Treatment of a Tongue Lymphangioma With Sirolimus after Failure of Surgical Resection and Propranolol [J].
Akyuz, Canan ;
Atas, Erman ;
Varan, Ali .
PEDIATRIC BLOOD & CANCER, 2014, 61 (05) :931-932
[3]   Airway response to sirolimus therapy for the treatment of complex pediatric lymphatic malformations [J].
Alemi, A. Sean ;
Rosbe, Kristina W. ;
Chan, Dylan K. ;
Meyer, Anna K. .
INTERNATIONAL JOURNAL OF PEDIATRIC OTORHINOLARYNGOLOGY, 2015, 79 (12) :2466-2469
[4]  
AMODEO I, 2017, MEDICINE, V0096
[5]  
Azouz Haya, 2016, JAAD Case Rep, V2, P350, DOI 10.1016/j.jdcr.2016.05.019
[6]   Lymphangiomas of the oral cavity: A clinicopathologic, immunohistochemical, and electron-microscopic study [J].
Brennan, TD ;
Miller, AS ;
Chen, SY .
JOURNAL OF ORAL AND MAXILLOFACIAL SURGERY, 1997, 55 (09) :932-935
[7]   Novel Route of Sirolimus Administration in a Neonate [J].
Clark, Ashley B. ;
Chandrasekar, Indira ;
Nickleson, Jennee .
JOURNAL OF PHARMACY PRACTICE, 2018, 31 (02) :167-168
[8]   Anterior Laryngeal Web Leading to Unanticipated Difficult Tracheal Intubation in a Neonate Diagnosed and Managed Successfully With CMAC Video Laryngoscope: A Case Report [J].
Gupta, Anju ;
Gupta, Nishkarsh .
A & A PRACTICE, 2018, 10 (01) :28-30
[9]   A Review of the Management of Lymphangiomas [J].
Ha, Jennifer ;
Yu, Yu-Ching ;
Lannigan, Francis .
CURRENT PEDIATRIC REVIEWS, 2014, 10 (03) :238-248
[10]  
Jung S., 2018, Frontiers in Immunology, V9