Ifosfamide encephalopathy is a fatal toxicity of Ifosfamide therapy. 13 cases have been reported till the year of 2018 worldwide
in children and none among Ewing’s sarcoma cases. Here we are discussing a unique case of Ifosfamide encephalopathy during
the course of treatment of Ewing’s sarcoma and was on Ifosfamide therapy and developed encephalopathy. Child improved
symptomatically with MESNA and mannitol. Child was found to be normal on follow up and chemotherapy treatment was continued..