Tehran University of Medical Sciences Acta Medica Iranica 0044-6025 57 7 2020 01 25 461 463 EN Amirhossein Larijani Brain and Spinal Cord Injury Research Center, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran . Amirhossein Darvishi Brain and Spinal Cord Injury Research Center, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. Kourosh Karimi-Yarandi Brain and Spinal Cord Injury Research Center, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. Seyed Ali Ahmadi Department of Pathology, Sina Hospital, Tehran University of Medical Sciences, Tehran, Iran. Maysam Alimohamadi Brain and spinal cord injury research center, Neuroscience institute, Tehran University of Medical Sciences 2018 12 29 2019 09 08 The immunosuppressive state after organ transplantation leads to infectious complications responsible for high mortality rate. Fungal infections account for 5% of all infections in Renal Transplantation (RT) recipients. A 53-year-old man was admitted with acute loss of consciousness. He had a history of renal transplantation 1 month before admission. Brain CT showed acute hydrocephalus with periventricular edema. A right frontal external ventricular drain was inserted urgently. One day later the consciousness further decreased, and CT scan showed unilateral left lateral ventricle dilatation and a hyperdense mass at the septum pellucidum. The patient was operated via endoscopic trans-middle frontal sulcus approach. The interventricular septum was pushed contralaterally, and a very adherent mesh-like thick layer covered the surface of lateral ventricle and foramen of Moro. The Monro foramen was opened, and septostomy was done. The mass inside the septum was found posterior to the foramen, punctured, and partially removed. Histopathological study revealed aspergillus infection of the lateral ventricles. CNS Aspergillosis should be considered in the differential diagnosis of altered consciousness among immunocompromised hosts. This report further implies that isolated ventricular involvement by aspergillus infection may be considered as the cause of hydrocephalus in immunocompromised patients. https://acta.tums.ac.ir/index.php/acta/article/view/7650 https://acta.tums.ac.ir/index.php/acta/article/download/7650/5292