Background Involvement of the brain by Aspergillus fumigatus, named as invasive aspergillosis, is extremely rare. Definitive diagnosis needs isolation of the fungus from the tissue specimens and its culture. The disease is very resistant to treatment and quickly becomes fatal despite antifungal chemotherapy with AmB preparations, the drugs of choice for the infection. Case Description We hereby present a unique case of fungal granuloma of the brain due to A fumigatus, which was cured successfully by intensive antifungal treatment, along with a brief literature review. Conclusions Although it is rare, we draw the attention to the fact that early diagnostic procedures with rapid confirmation of Aspergillus infection and an early initiation of therapy are pivotal for a benign clinical course in such cases. A complete regression of the disease could be achieved with medical therapy alone with conventional AmB or liposomal AmB combined with oral itraconazole even if a total surgical excision of the lesions could not be performed in complicated cases, as in our patient.