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Introduction Cryptococcal meningitis often presents subacutely with fever, headaches, neck stiffness and altered mental status. While neuro-ophthalmologic manifestations are described in the literature, protracted serial cranial neuropathies secondary to chronic cryptococcus gattii meningitis is extremely rare.Case A 63-year-old immunocompetent male with a long history of recurrent cranial neuropathies of unclear aetiology, was admitted with a new left oculomotor nerve palsy. Previous episodes were separated by years and always involved different nerves; including the right facial, then left facial, right oculomotor and left abducens nerve. Serum and CSF studies demonstrated no significant results. MRI brain identified no compression. Notably, serum and CSF cryptococcus antigen (CrAg) testing completed during this episode were negative. Upon discharge, an outpatient CT chest revealed a 13mm pleural-based lesion in the right lung, with Cryptococcus gattii grown on biopsy. Repeat serum CrAg remained negative however subsequent repeat CSF now showed a positive CrAg result. The patient was admitted for anti-fungal induction therapy with amphotericin and flucytosine. Interestingly when CrAg positive, the patient’s oculomotor nerve palsy had resolved and he was asymptomatic with no neurological deficits on examination. Following antifungal induction therapy the patient has since continued maintenance therapy with fluconazole for 2 years and has not had any further episodes of cranial neuropathies to date.Conclusion This case brings awareness to a rare manifestation of chronic meningitis secondary to an often-unsuspected organism, Cryptococcus gattii. Uniquely, this patient was CrAg negative during his episode of cranial neuropathy and only became positive upon symptom resolution.