Invasive sphenoid sinus aspergillosis with normal findings on initial diagnostic tests that mimics Tolosa-Hunt syndrome-a diagnostic dilemma: a case report

Abstract

Invasive sphenoid sinus aspergillosis can mimic Tolosa-Hunt syndrome (THS), leading to frequent misdiagnoses and potentially fatal consequences. We report a case of invasive sphenoid sinus aspergillosis initially misdiagnosed as THS. A 79-year-old man presented with right periorbital pain, ophthalmoplegia, and loss of vision. Initial evaluations including magnetic resonance imaging (MRI), were normal. He was first diagnosed with THS based on clinical features. The disease progressed despite high-dose intravenous steroid treatment, and an enhancing mass-like lesion was found in the right orbital apex, cavernous sinus, and sphenoid sinus on follow-up MRI. Aspergillosis was eventually confirmed by sphenoid sinus biopsy. The patient developed cerebral infarction and finally died despite being treated with amphotericin B. Given that invasive sphenoid sinus aspergillosis may initially resemble THS, high suspicion and rapid histological examination are important for diagnosis.

Keywords: Aspergillosis; Painful ophthalmoplegia; Sphenoid sinus; Tolosa-Hunt syndrome.

Fig. 1.

(A) Initial gadolinium-enhanced T1-weighted magnetic resonance imaging (MRI) shows no abnormalities. (B) T2 fluid-attenuated inversion recovery and (C) T1-weighted (contrast-enhanced) imaging of follow-up gadolinium-enhanced MRI show thickening and enhancement of the right optic nerve, cavernous sinus, sphenoid sinus soft tissue with partial bony destruction, and spread of enhancing tissue into the right orbital apex (arrows). (D) The source image of magnetic resonance angiography shows encasement of the right internal carotid artery (ICA) as well as ipsilateral optic canal invasion (arrows). (E) Diffusion-weighted MRI reveals acute infarction in the right ICA territory.

Fig. 2.

Histopathology of the resected tissue shows abundant Aspergillus organisms with typical branching septate hyphae with a bulbous appearance that branch at 45° (hematoxylin-eosin stain, ×400).