In June, 2008, a 44-year-old immunocompetent man presented to us with sinusitis associated with headache, transient diplopia, and dizziness. He had a 7-year history of chronic sinusitis occasionally treated with pseudoephedrine and antibiotics. CT of the sinuses showed opacifi cation of the paranasal sinuses which were fi lled with high density material, erosion of the sphenoid bone, and propagation of the pathological process to the sellar region, with a large sellar mass (fi gure A). MRI confi rmed a giant hypointense lesion in the sellar region. The pituitary gland was displaced upwards. Physical examination was unremarkable, except for obesity (BMI 38 kg/m2). Computerised visual-fi eld examination, and visual acuity and motility were normal. The insulin hypoglycaemia test showed a peak growth hormone (GH) of 3·4 μg/L, borderline for severe GH defi ciency (<3·0 μg/L); cortisol response was normal. Prolactin concentration was high (1150 mU/L; mean of three values). Macroprolactin was negative. Testosterone, FT4, and TSH concentrations were normal, and antibodies to thyroid peroxidase and thyroglobulin were present, compatible with a diagnosis of euthyroid chronic thyroiditis. In view of the large cystic sellar mass, and to provide symptomatic relief, the patient underwent transphenoidal surgery. Intraoperatively, thick brown material with a peanut-butter-like consistency was cleared from the sella. Histological examination showed many eosinophils within the eosinophilic mucin, but did not show a pituitary adenoma (fi gure B). In addition, septate fungal hyphae with acute angle branching embedded within the clusters of eosinophils were seen, with no fungal invasion of the tissue (fi gure C). Culture of the sellar material grew a white cotton-like mould colony; microscopically, hyphae with spicules and clamp connections were seen, suggestive of Schizophyllum commune. This was confi rmed by sequence analysis (strain number CBS 124811). Despite pituitary stalk decompression, prolactin concentrations remained high (990 mU/L). For the residual mass in the sinuses, functional endoscopic sinus surgery was done. The patient was treated with systemic antimycotics (liposomal amphotericin B, 2 weeks before and 1 week after surgery) and systemic and topical corticosteroids, followed by itraconazole for 2 months). When seen in August, 2009, he had improved clinically and prolactin concentration had decreased to 550 mU/L. We regularly inhale spores of diverse fungal species, yet fungal disease is uncommon. Fungal organisms within nasal mucin are present in up to 93% of patients with chronic sinusitis and even in healthy persons without sinonasal disease. Allergic fungal sinusitis is a noninfectious chronic disease with an immunological response to colonising fungi resulting in an eosinophilic infl ammation. S commune is a basidiomycetes fungus which has been identifi ed in some verifi ed human infections. In our case, S commune triggered an aggressive eosinophilic infl ammation. The diff erential diagnosis of large sellar masses relies mainly on pathological analysis. The most common lesions encountered in the sellar region are infective, vascular, neoplastic, or immunological. The giant hypointense lesion in the sella on MRI scans results from the very high amounts of proteins released by the eosinophils into the mucin. CT is a better diagnostic modality for allergic fungal sinusitis. In our patient, the persistence of hyperprolactinaemia after pituitary surgery, with no compression to the pituitary stalk, was intriguing. In-vitro studies show that fungal glucans may directly interact with glucan-specifi c receptors on rodent somatomammotroph cell membranes to stimulate prolactin secretion. In view of the many individuals with chronic sinusitis, physicians should be vigilant in recognising and referring patients with aggressive disease to appropriate specialists.
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Oct 11, 2024
Sandra Aulia Mardikasari + 2
Open Access
