Background: Painful ophthalmoplegia can be caused by various etiologies, and broad differential diagnosis is needed.\nCarotid-cavernous fistula (CCF) is a rare cause of painful ophthalmoplegia, and early diagnosis is quite difficult.\nCase presentation: Here, we present a case of paroxysmal painful ophthalmoplegia caused by CCF. The episodic\nsymptoms were nonstereotypical and lasted minutes to hours. Magnetic resonance imaging (MRI) and computed\ntomography angiography (CTA) results were normal, which confounded efforts to determine a diagnosis. Subsequently,\ndigital subtraction angiography (DSA) revealed a posterior-draining CCF. The CCF was treated at an early stage without\nresidual symptoms.\nConclusions: We propose that symptoms could be relapsing or remitting during an early stage of CCF and that\nposterior-draining CCF is prone to misdiagnosis due to atypical manifestations. Normal CTA results cannot exclude\ncarotid-cavernous fistula, and DSA should be performed once CCF is suspected.
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