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The immunosuppressive state in organ transplantation leads to infectious complications responsible for high mortality rate. Fungal infections account for 5% of all infections in Renal Transplantation (RT) recipients. Aspergillus species are filamentous fungi frequently causing fungal infections in RT recipients. Lungs and paranasal sinuses are the usual portal of entry from where it disseminates to other organs. Here, we are reporting a case of 14-year-old boy with RT from mother’s kidney, who had atypical presentation of isolated cerebral aspergillosis at 19 months post-transplant without identified portal of entry. Early diagnosis and prompt treatment saved the patient and the graft.