Efficacy and Tolerance of Sirolimus (Rapamycin) for Extracranial Arteriovenous Malformations in Children and Adults - Archive ouverte HAL Accéder directement au contenu
Article Dans Une Revue Acta Dermato-Venereologica Année : 2019

Efficacy and Tolerance of Sirolimus (Rapamycin) for Extracranial Arteriovenous Malformations in Children and Adults

Romain Gabeff
  • Fonction : Auteur
Olivia Boccara
  • Fonction : Auteur
Véronique Soupre
  • Fonction : Auteur
Gérard Lorette
  • Fonction : Auteur
Christine Bodemer
Denis Herbreteau
  • Fonction : Auteur

Résumé

Managing extracranial arteriovenous malformations is challenging. Sirolimus (rapamycin) is increasingly being used when surgery and embolization are not advised. Because of its anti-angiogenic properties here we report all extracranial arteriovenous malformation cases treated with sirolimus in 2 French tertiary centers for vascular anomalies. The outcomes were efficacy (complete, partial, no response) based on arteriovenous malformation volume and necrosis/hemorrhage and side effects. We retrospectively included 10 patients (7 children). The sirolimus dose ranged from 0.6 to 3.5 mg/m2. Median (interquartile range [IQR]) treatment time was 24.5 (4.5; 35) months. Five patients showed no response and 5 showed partial response at a median (IQR) of 3 (1; 5) months followed in 2 cases by therapeutic resistance (i.e., progressive disease after 9 and 24 months of treatment). The most frequent side effect was mouth ulcers. This study shows poor efficacy of sirolimus for treating extracranial arteriovenous malformations.

Dates et versions

hal-03158791 , version 1 (04-03-2021)

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Citer

Romain Gabeff, Olivia Boccara, Véronique Soupre, Gérard Lorette, Christine Bodemer, et al.. Efficacy and Tolerance of Sirolimus (Rapamycin) for Extracranial Arteriovenous Malformations in Children and Adults. Acta Dermato-Venereologica, 2019, 99 (12), pp.1105-1109. ⟨10.2340/00015555-3273⟩. ⟨hal-03158791⟩
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