A69-year-old male with a history of polymyalgia rheumatica presented with 4 months of intermittent sharp left retro-orbital pain and worsening vision. Two weeks prior to symptom onset, he underwent an… Click to show full abstract
A69-year-old male with a history of polymyalgia rheumatica presented with 4 months of intermittent sharp left retro-orbital pain and worsening vision. Two weeks prior to symptom onset, he underwent an ozone sinus treatment which caused significant “burning” in the area, per the patient, at an outside facility. The patient then developed progressive headache followed by increasing vision changes, nausea, and vomiting. The patient was afebrile throughout. Over the next 4 months, treatments included a frontal balloon sinuplasty and multiple doses of intravenous (i.v.) corticosteroids. In the 4 to 6 weeks prior to transfer to our facility, based on symptoms and imaging findings, he was diagnosed with Tolosa-Hunt syndrome, a rare, corticosteroid-responsive, idiopathic granulomatous inflammatory disorder of the cavernous sinus. He was continuously treated with corticosteroids at escalating doses, culminating in two admissions during which 1 g of i.v. methylprednisolone was given, followed by 60 mg of oral prednisone daily, to be tapered slowly. Immediately prior to transfer he again presented to his local emergency department due to worsening of his left eye swelling and now complete vision loss. He was given 1 g of i.v. methylprednisolone and transferred to our facility. Upon arrival, examination of the left orbit and surrounding area showed marked proptosis and lid ptosis, a relative afferent pupillary defect, mild optic nerve pallor, decreased visual acuity (ability only to perceive light) and a 23 supraduction deficit. Magnetic resonance imaging (MRI) of the orbits showed soft tissue thickening and enhancement within the left superior orbital fissure extending into the orbital apex and surrounding the optic nerve (Fig. 1, left). He was suspected to have a neoplasm, sarcoidosis, or pseudotumor and underwent surgical biopsy. No cultures were sent, and all tissue was fixed in formalin. Staining of the surgical biopsy specimen with hematoxylin and eosin showed hyaline, septate, branching fungal hyphae (Fig. 1, right). The diagnosis was later established via tissue broad-range fungal PCR.
               
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