Scientific article
Case report
English

Sirolimus Treatment of an Infant With Intrathoracic Kaposiform Hemangioendothelioma Complicated by Life-threatening Pleural and Pericardial Effusions

Published inJournal of pediatric hematology/oncology, vol. 42, no. 1, p. 74-78
Publication date2020-01
Abstract

Kaposiform hemangioendothelioma (KHE) is a rare infiltrative vascular tumor that may be associated with Kasabach-Merritt Phenomenon (KMP), which is a consumptive coagulopathy with potentially life-threatening thrombocytopenia. Management of KHE and KMP is challenging, and currently, there are no standardized validated treatment protocols. Mammalian target of rapamycin inhibitors have been shown to be effective in the treatment of KHE. We describe a term male who presented as a diagnostic dilemma with life-threatening pleural and pericardial effusions and severe thrombocytopenia. After extensive work-up the etiology for his condition was determined to be KHE with KMP. The patient was commenced on sirolimus and responded well to therapy with resolution of KMP.

Keywords
  • Hemangioendothelioma / diagnosis
  • Hemangioendothelioma / drug therapy
  • Humans
  • Infant, Newborn
  • Kasabach-Merritt Syndrome / diagnosis
  • Kasabach-Merritt Syndrome / drug therapy
  • Male
  • Pericardial Effusion / diagnosis
  • Pericardial Effusion / drug therapy
  • Pleural Effusion, Malignant / diagnosis
  • Pleural Effusion, Malignant / drug therapy
  • Sarcoma, Kaposi / diagnosis
  • Sarcoma, Kaposi / drug therapy
  • Sirolimus / administration & dosage
Citation (ISO format)
DUAN, Lucy et al. Sirolimus Treatment of an Infant With Intrathoracic Kaposiform Hemangioendothelioma Complicated by Life-threatening Pleural and Pericardial Effusions. In: Journal of pediatric hematology/oncology, 2020, vol. 42, n° 1, p. 74–78. doi: 10.1097/MPH.0000000000001268
Main files (1)
Article (Published version)
accessLevelRestricted
Identifiers
Journal ISSN1077-4114
231views
0downloads

Technical informations

Creation18/10/2021 06:15:00
First validation18/10/2021 06:15:00
Update16/03/2023 02:33:47
Status update16/03/2023 02:33:47
Last indexation01/11/2024 00:44:50
All rights reserved by Archive ouverte UNIGE and the University of GenevaunigeBlack