Successful Propranolol Treatment of a Kaposiform Hemangioendothelioma Apparently Resistant to Propranolol.
A newborn with unresectable kaposiform hemangioendothelioma associated with Kasabach Merritt phenomenon, unresponsive to vincristine and prednisone, received second-line treatment with propranolol at a dose of 2 mg/kg/day, starting at 2 months of life and continued for 13 months. There was only slight reduction in tumor mass, but measurement of propranolol levels showed extremely low plasma concentrations. The propranolol dose was progressively increased to 3.5 mg/kg/day, leading to a substantial increase in plasma levels associated with clinically relevant tumor reduction. This case highlights the importance of relating propranolol dose to its plasma concentration before considering the treatment ineffective for this vascular tumor.
Neonatal Intensive Care Unit, Medical Surgical Fetal-Neonatal Department, "A. Meyer" University Children's Hospital, Florence, Italy.
Luca
L
Filippi
Department of Pediatric Hematology-Oncology, "A. Meyer" University Children's Hospital, Florence, Italy.
Angela
A
Tamburini
Neonatal Intensive Care Unit, Medical Surgical Fetal-Neonatal Department, "A. Meyer" University Children's Hospital, Florence, Italy.
Elettra
E
Berti
Radiology Department, "A. Meyer" University Children's Hospital, Florence, Italy.
Anna
A
Perrone
Radiology Department, "A. Meyer" University Children's Hospital, Florence, Italy.
Claudio
C
Defilippi
Department of Pediatric Hematology-Oncology, "A. Meyer" University Children's Hospital, Florence, Italy.
Claudio
C
Favre
Department of Pediatric Hematology-Oncology, "A. Meyer" University Children's Hospital, Florence, Italy.
Maura
M
Calvani
Department of Neurosciences, "A. Meyer" University Children's Hospital, Florence, Italy.
Maria Luisa
ML
Della Bona
Department of Neurosciences, "A. Meyer" University Children's Hospital, Florence, Italy.
Giancarlo
G
la Marca
Neonatal Intensive Care Unit, Medical Surgical Fetal-Neonatal Department, "A. Meyer" University Children's Hospital, Florence, Italy.
Gianpaolo
G
Donzelli
D019008:Drug Resistance, Neoplasm / Q000187:drug effects*; D006390:Hemangioendothelioma / Q000188:drug therapy*; D006801:Humans; D007231:Infant, Newborn; D007232:Infant, Newborn, Diseases / Q000188:drug therapy*; D059885:Kasabach-Merritt Syndrome / Q000188:drug therapy*; D008297:Male; D011433:Propranolol / Q000008:administration & dosage*; D012514:Sarcoma, Kaposi / Q000188:drug therapy*
D011433:Propranolol
beta-blockers; kaposiform hemangioendothelioma; propranolol