Isolated Pituitary Blastomycosis Presenting as Hypophysitis with Intracranial Hypertension and Multifocal Strokes in an Immunocompetent Patient
Objective:
To alert to a case of central nervous system (CNS) blastomycosis in an immunocompetent patient presenting as isolated hypophysitis complicated by intracranial hypertension and multifocal strokes. Our aim is to highlight the associated diagnostic challenges and management strategies.
Background:
Blastomyces dermatitidis is a thermally dimorphic fungus endemic to North America. CNS involvement occurs in approximately 4% of cases and typically manifests as meningitis, epidural abscess, or mass lesions. Isolated pituitary involvement is extremely rare and can mimic alternative pathologies such as pituitary adenoma.
Design/Methods:
Not Applicable
Results:
A 24-year-old patient presented with progressive headaches, diplopia, and papilledema. Imaging suggested a pituitary adenoma with suprasellar extension and findings consistent with elevated intracranial pressure. Neurosurgical exploration revealed an inflamed pituitary gland without adenoma; biopsy showed granulomatous inflammation. Cerebrospinal fluid studies confirmed B. dermatitidis via culture. The clinical course was complicated by multifocal ischemic strokes, possibly secondary to infectious vasculopathy. Initial treatment with amphotericin B was discontinued due to adverse effects, and the patient was transitioned to voriconazole with gradual clinical improvement. A ventriculoperitoneal shunt was placed for elevated intracranial pressure. Follow-up imaging showed resolution of papilledema and improved leptomeningeal enhancement, although residual disease required prolonged antifungal therapy and multidisciplinary follow-up.
Conclusions:
This case represents, to our knowledge, the first report of isolated hypophysitis due to Blastomyces dermatitidis without evidence of disseminated disease. The presentation initially mimicked a pituitary adenoma, delaying diagnosis. The occurrence of multifocal infarcts suggests infectious vasculopathy as a potential mechanism of stroke in CNS blastomycosis. Recognition of this atypical manifestation is essential, as timely diagnosis and antifungal therapy are critical to reducing morbidity and mortality. This case underscores the importance of maintaining a broad differential diagnosis when evaluating suspected pituitary lesions and highlights the need for further research into the pathogenesis and optimal management of CNS blastomycosis.
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