Abstract
BACKGROUND: The only known case report of spinal intramedullary aspergillosis in a patient who had no known immunocompromised status and no extraspinal infection is presented. A subsequent literature review was conducted to compare this case with existing cases in the literature. OBSERVATIONS: After 3 months of a worsening clinical course, the neurosurgery team was consulted to conduct a biopsy of the patient's intramedullary spinal lesion, revealing an invasive aspergillosis infection of the CNS. The patient had experienced significant morbidity and clinical symptoms, including a sensory level at T10, urinary retention, worsening of the sensory level to T6, and lower extremity weakness (2-3/5 strength on the Medical Research Council scale). Infection did not progress after appropriate antifungal treatment and diagnosis. The patient was still alive at a 4-month follow-up visit with the neurology team. LESSONS: Invasive aspergillosis is a rare diagnosis that typically arises in immunocompromised patients. This case suggests that this infectious diagnosis should be included in the differential diagnosis even if a patient is immunocompetent and there is no sign of extraspinal infection. https://thejns.org/doi/10.3171/CASE24463.