Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) typically affects young adults and children, commonly manifesting as optic neuritis or myelitis. However, atypical presentations—particularly in the elderly—remain under-recognised. We report a rare case of recurrent painful ophthalmoplegia in an elderly woman, ultimately diagnosed with MOGAD, illustrating the importance of high clinical suspicion and prompt immunotherapy.

A 77-year-old woman with a history of hypertension, diabetes, coronary artery disease, and recurrent deep vein thrombosis on rivaroxaban presented with a two-week history of severe occipital and frontal headache, binocular diplopia, and complete ophthalmoplegia. Symptoms were accompanied by left-sided facial numbness, nausea, vomiting, and photophobia. Notably, she reported a similar episode of painful right ocular paresis at the age of 15 years old prior to that, which resolved spontaneously.

Neurological examination revealed complete bilateral ophthalmoplegia and left V1–V2 hypoesthesia, with preserved visual acuity. Brain MRI showed linear contrast enhancement near the left cavernous sinus and superior orbital fissure, without optic nerve involvement. Lumbar puncture was unremarkable. She was empirically treated for recurrent painful ophthalmoplegic neuropathy (RPON). High-dose oral prednisolone (1 mg/kg/day, tapered over 8 weeks) led to full clinical recovery. However, serum MOG-IgG returned positive at a high titer (1:1000), confirming MOGAD. She was subsequently started on rituximab as maintenance therapy.

Conclusions:

MOGAD may present with isolated, recurrent painful cranial neuropathies, even in elderly patients, mimicking RPON or ophthalmoplegic migraine. This case emphasises the need to expand diagnostic consideration for MOGAD beyond classic presentations and age groups. Early diagnosis and immunotherapy are key to achieving favourable outcomes.