, Volume 1482, Issue 1, p 36
Date: 14 Dec 2013


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A female infant [age at reaction onset not clearly stated] developed mucositis during treatment with sirolimus.

The girl had PHACE (Posterior fossa brain malformations, Haemangiomas, Arterial anomalies, Cardiac defects, and Eye abnormalities) syndrome and treatment-resistant infantile haemangioma (IH). She presented with extensive facial IH complicated by lip ulceration at the age of 3 weeks. She initially received prednisolone and then propranolol. Despite treatment, her IH rapidly progressed and she began treatment with oral sirolimus 1.5 mg/m2 daily at the age of 14 weeks. Her sirolimus was increased to 1.5 mg/m2 twice daily after 4 weeks of therapy. Her propranolol was stopped 6 weeks after beginning sirolimus and her corticosteroids were tapered and withdrawn 13 weeks after starting sirolimus. The sirolimus trough concentration range was 4.3−19.2 ng/mL. During hospitalisation for feeding intolerance, a sirolimus trough concentration of 20.3 ng/mL was noted and her sirolimus dosage ...