Sirolimus combined with glucocorticoids in the treatment of Kasabach-Merritt phenomenon in a neonate: A case report

Medicine (Baltimore). 2024 Apr 5;103(14):e37706. doi: 10.1097/MD.0000000000037706.

Abstract

Rationale: Kaposiform hemangioendothelioma is an aggressive vascular tumor that is often associated with life-threatening coagulopathies and Kasabach-Merritt phenomenon. Pathologic biopsies can provide a good basis for diagnosis and treatment. Therapy with srolimus combined with glucocorticoids may offer patients a favorable prognosis.

Patient concerns: A large purplish-red mass on the knee of a child with extremely progressive thrombocytopenia and refractory coagulation abnormalities. Conventional doses of glucocorticoids alone failed to improve coagulation abnormalities and the child developed large cutaneous petechiae and scalp hematomas.

Diagnosis: Kaposiform hemangioendothelioma combined with Kasabach-Merritt phenomenon.

Interventions: The patient received prednisolone 2.0 mg/kg*d for 4 days. Blood products were transfused to ensure vital signs and to complete the pathologic biopsy. Sirolimus combined with prednisolone was given after clarifying the diagnosis of Kaposiform hemangioendothelioma.

Outcomes: The tumor basically disappeared on examination and the ultrasound showed a subcutaneous hyperechoic mass with normal blood flow.

Lessons: Sirolimus combined with glucocorticoids is effective in controlling Kasabach-Merritt phenomenon and pathologic biopsy is important for definitive diagnosis.

Publication types

  • Case Reports

MeSH terms

  • Blood Coagulation Disorders* / complications
  • Glucocorticoids / therapeutic use
  • Hemangioendothelioma* / complications
  • Hemangioendothelioma* / diagnosis
  • Hemangioendothelioma* / drug therapy
  • Humans
  • Infant, Newborn
  • Kasabach-Merritt Syndrome* / drug therapy
  • Prednisolone / therapeutic use
  • Sarcoma, Kaposi* / pathology
  • Sirolimus / therapeutic use

Substances

  • Glucocorticoids
  • Prednisolone
  • Sirolimus

Supplementary concepts

  • Kaposiform Hemangioendothelioma