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A Case Report of 2 Sirolimus-Related Deaths Among Infants With Kaposiform Hemangioendotheliomas

Hanru Ying1, Congzhen Qiao1, Xi Yang1

  • 1Department of Plastic and Reconstructive Surgery, Shanghai Ninth People's Hospital and School of Medicine, Shanghai Jiao Tong University, Shanghai, China.

Pediatrics
|April 4, 2018
PubMed

Insights

Kaposiform hemangioendothelioma (KHE) with Kasabach-Merritt phenomenon (KMP) in infants can be fatal. Sirolimus treatment may increase infection risk, necessitating careful monitoring and antibiotic prophylaxis.

Area of Science:

  • Pediatric Oncology
  • Vascular Malformations
  • Pharmacology

Background:

  • Kaposiform hemangioendothelioma (KHE) is a rare, aggressive vascular tumor.
  • Kasabach-Merritt phenomenon (KMP) associated with KHE presents with severe thrombocytopenia and hypofibrinogenemia, leading to high mortality.
  • Current KHE treatments lack standardization, with medications like sirolimus showing potential but carrying risks.

Observation:

  • Two infant patients with KHE and KMP were treated with sirolimus.
  • Both patients developed respiratory complications (cough, tachypnea) shortly after sirolimus initiation.
  • These complications led to fatal infections in both cases.

Findings:

  • Sirolimus treatment in infants with KHE and KMP may be associated with an increased risk of severe infections.
  • Respiratory symptoms like cough and tachypnea can be early indicators of infection in these patients.
  • Fatal outcomes underscore the potential dangers of sirolimus's off-target effects.

Implications:

  • The findings highlight the critical need for vigilant monitoring of sirolimus-treated infants for signs of infection.
  • Prophylactic antibiotics should be strongly considered in this patient population.
  • Regular monitoring of serum sirolimus levels is crucial for optimizing treatment safety and efficacy.

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