Literature DB >> 29610164

Pneumocystis Jirovecii Pneumonia During Sirolimus Therapy for Kaposiform Hemangioendothelioma.

Thomas B Russell1, Emily K Rinker2, Claire S Dillingham3, Laurence B Givner1, Thomas W McLean4.   

Abstract

Sirolimus is an effective therapy for children with kaposiform hemangioendothelioma with or without the Kasabach-Merritt phenomenon. We report the case of a child with kaposiform hemangioendothelioma and the Kasabach-Merritt phenomenon who developed Pneumocystis carinii pneumonia (PCP) while on sirolimus and a prednisolone taper, after lack of adequate response to prednisolone, propranolol, and vincristine. He had a prompt positive clinical and laboratory response to sirolimus, but 4 weeks after starting it, at the age of 4 months, he developed PCP. This led to respiratory failure, which required extracorporeal membrane oxygenation. Sirolimus was temporarily discontinued, and he was successfully treated for PCP with sulfamethoxazole-trimethoprim and methylprednisolone. He was restarted on sirolimus 3 weeks after discharge and given sulfamethoxazole-trimethoprim prophylaxis. At the age of 22 months, while still on sirolimus, the lesion continued to improve with test results revealing stable hemoglobin and platelet counts. PCP is a rare but life-threatening side effect of sirolimus therapy, especially in the setting of concurrent steroid treatment. Pneumocystis prophylaxis should be considered for patients receiving sirolimus.
Copyright © 2018 by the American Academy of Pediatrics.

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Year:  2018        PMID: 29610164     DOI: 10.1542/peds.2017-1044

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  2 in total

Review 1.  Kaposiform hemangioendothelioma: current knowledge and future perspectives.

Authors:  Yi Ji; Siyuan Chen; Kaiying Yang; Chunchao Xia; Li Li
Journal:  Orphanet J Rare Dis       Date:  2020-02-03       Impact factor: 4.123

2.  Effective low-dose sirolimus regimen for kaposiform haemangioendothelioma with Kasabach-Merritt phenomenon in young infants.

Authors:  Veroniek E M Harbers; Nathalie van der Salm; Sjoert A H Pegge; Carine J M van der Vleuten; Bas H Verhoeven; Sabine L A G Vrancken; Leo J Schultze Kool; Joris Fuijkschot; D Maroeska M W M Te Loo
Journal:  Br J Clin Pharmacol       Date:  2022-01-18       Impact factor: 3.716

  2 in total

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