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Pneumocystis Jirovecii Pneumonia During Sirolimus Therapy for Kaposiform Hemangioendothelioma

Thomas B Russell1, Emily K Rinker2, Claire S Dillingham3

  • 1Departments of Pediatrics and.

Pediatrics
|April 4, 2018
PubMed

Insights

Sirolimus effectively treats kaposiform hemangioendothelioma but can cause rare Pneumocystis pneumonia (PCP) in children. Prophylaxis is recommended, especially with concurrent steroid use, to prevent this life-threatening side effect.

Area of Science:

  • Pediatric Oncology
  • Immunology
  • Pharmacology

Background:

  • Kaposiform hemangioendothelioma (KHE) is a rare vascular tumor in children.
  • Sirolimus is an effective treatment for KHE, often used when other therapies fail.
  • Kasabach-Merritt phenomenon (KMP) is a complication of KHE involving thrombocytopenia and coagulopathy.

Observation:

  • A pediatric patient with KHE and KMP developed Pneumocystis pneumonia (PCP) while on sirolimus and a prednisolone taper.
  • The patient experienced respiratory failure requiring extracorporeal membrane oxygenation (ECMO).
  • PCP occurred despite initial treatment with prednisolone, propranolol, and vincristine.

Findings:

  • Sirolimus showed a positive clinical and laboratory response in the KHE patient.
  • PCP developed 4 weeks after initiating sirolimus therapy.
  • Treatment with sulfamethoxazole-trimethoprim and methylprednisolone resolved PCP, allowing sirolimus reinitiation with prophylaxis.

Implications:

  • Pneumocystis pneumonia is a rare but serious adverse event associated with sirolimus therapy in pediatric patients.
  • Concurrent steroid use may increase the risk of PCP in patients on sirolimus.
  • Pneumocystis prophylaxis should be strongly considered for children receiving sirolimus, particularly those on corticosteroids.

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