Periostitis deformans is an uncommon condition of the skeletal system, mainly manifested as bone pain with or without bony swellings. Voriconazole-induced periostitis is widely reported in the literature, mainly in the organ transplant patients on immunosuppressant therapy. The patient in this case report, was not on any immunosuppressant therapy, but developed widespread periostitis deformans secondary to voriconazole, who was being treated for the base of skull aspergillus osteomyelitis. This report demonstrates the severity of voriconazole adverse effects and the wider impact on patient. Radiological features of periostitis deformans are still a cause of concern and the diagnosis of this condition is not easy due to its radiological features like bone tumours. This case report is an attempt to promote more awareness about this rare condition among both the orthopaedic surgeons and the radiologists, which will help in streamlining investigation and prompt diagnosis.