Sirolimus for vascular anomalies associated with PTEN hamartoma tumor syndrome. Journal Articles uri icon

  •  
  • Overview
  •  
  • Research
  •  
  • Identity
  •  
  • Additional Document Info
  •  
  • View All
  •  

abstract

  • Phosphatase and tensin homolog (PTEN) hamartoma tumor syndrome (PHTS) is a rare condition associated with vascular anomalies and increased tumor risk. Sirolimus, an mTOR inhibitor used for managing vascular anomalies is underexplored in PHTS. A single-institution retrospective review of children with PHTS and vascular anomalies treated with sirolimus identified seven patients. Median age at sirolimus initiation was 10 years. After a median 2.5-year follow-up, six of seven patients (86%) showed significant clinical improvement. No significant adverse effects were observed, except mild buccal ulcers and acne. This study supports sirolimus as an effective and safe treatment for vascular anomalies in a small group of children with PHTS.

authors

  • Zabeida, Alexandra
  • Brzezinski, Jack J
  • Wasserman, Jonathan D
  • Cytrynbaum, Cheryl
  • Weksberg, Rosanna
  • Zwicker, Kelley
  • Zbuk, Kevin
  • Gasparetto, Alessandro
  • Willis, Laura
  • Fantauzzi, Michelle
  • Carcao, Manuel

publication date

  • November 2024