A case report of sirolimus use in early fetal management of lymphatic malformation.
Journal
Nature cardiovascular research
ISSN: 2731-0590
Titre abrégé: Nat Cardiovasc Res
Pays: England
ID NLM: 9918284280206676
Informations de publication
Date de publication:
Jun 2023
Jun 2023
Historique:
received:
15
12
2022
accepted:
02
05
2023
medline:
1
6
2023
pubmed:
1
6
2023
entrez:
28
8
2024
Statut:
ppublish
Résumé
Sirolimus, by targeting the mammalian target of rapamycin (mTOR) pathway, has demonstrated efficacy on lymphatic malformations (LMs) in adults and neonates. The current hypothesis is that the earlier the lesion is treated, the better it responds. This has prompted the idea that sirolimus administration might be efficacious to treat fetal LMs as well. Here we report a successful management of a cervicofacial fetal LM with sirolimus taken orally by the mother from the 22nd week of pregnancy until 2 weeks before planned delivery. Repeated cordocentesis recorded a 30% transplacental crossing of sirolimus. Continuation of sirolimus after birth allowed resection of the residual mass. We have followed the physical and neurological evolution of the child for 6 years since the fetal administration of sirolimus. We conclude that early administration of sirolimus during pregnancy with maternal serum monitoring may be proposed to high-risk fetal LMs in selected cases.
Identifiants
pubmed: 39195878
doi: 10.1038/s44161-023-00280-4
pii: 10.1038/s44161-023-00280-4
doi:
Substances chimiques
Sirolimus
W36ZG6FT64
MTOR Inhibitors
0
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
595-599Subventions
Organisme : Fonds De La Recherche Scientifique - FNRS (Belgian National Fund for Scientific Research)
ID : T.0146.16
Organisme : Fonds De La Recherche Scientifique - FNRS (Belgian National Fund for Scientific Research)
ID : P.C013.20
Organisme : Fonds De La Recherche Scientifique - FNRS (Belgian National Fund for Scientific Research)
ID : T.0247.19
Organisme : EC | Horizon 2020 Framework Programme (EU Framework Programme for Research and Innovation H2020)
ID : 874708
Informations de copyright
© 2023. The Author(s), under exclusive licence to Springer Nature Limited.
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