Background: Unilateral isolated sphenoid sinus disease (ISSD) is a rare diagnosis of the paranasal sinuses that can be associated with complications involving vascular, neurologic, and optic structures in close proximity.
Case presentation: A 62 year old female presented to a hospital in Georgetown, Guyana with right lateral rectus palsy, diplopia, and a severe progressively worsening headache. CT scan revealed an opaque left sphenoid sinus consistent with unilateral ISSD. A transnasal sphenoidotomy was performed without complication under local anesthetic in the absence of endoscopic guidance. The patient's headache resolved immediately after surgery while the diplopia and lateral rectus palsy resolved completely after 6 weeks.
Conclusion: We present a rare complication of ISSD and highlight challenges associated with diagnosis and management of ISSD in a resource-limited setting. This is the second reported case of unilateral ISSD with contralateral lateral rectus palsy in the literature.