Background: Cryptococcus has a tropism for the nervous system with a higher prevalence of infection in immunosuppressed patients; it remains a major cause of human immunodeficiency virus (HIV)-related mortality worldwide. Neurological compromise caused by this microorganism mainly debuts as a meningeal syndrome, spinal involvement has been reported in literature, neuropathological assessments have found Cryptococci in spinal roots and meninges, with perineuritic adhesions probably explaining compromise lower cranial nerves and even spinal nerve roots. Case Description: 39-year-old male seronegative for HIV, with a surgical history of hydrocephalus treated with ventriculoperitoneal shut 1 year before, he presented with progressive weakness in the four extremities evolving to be disabling with bilateral accessory nerve palsy and loss of sensation below his neck. The MR imaging showed diffuse leptomeningeal thickening both supra and infratentorial and over the spinal canal up to C5 with a cystic formation shown in the craniocervical union causing compression of the medullary bulb. The patient underwent a medial suboccipital craniectomy with resection of the posterior arch of c1 for sampling and decompression, pathologically appears numerous spherical organisms that have a thick clear capsule and are surrounded by histiocytes forming a granuloma compatible with Cryptococcus. Postoperatively, the patient’s prior neurological deficits resolved. Conclusion: It is an infrequently suspected pathology in immunocompetent patients, usually requiring only antifungal treatment with adjustment of immunosuppressive or antiretroviral management. In special and rare situations like our case as presenting with lower cranial nerve and spinal involvement, surgical treatment is a priority for the resolution of the pathology and improves disabling neurological deficit.
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