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Keywords:

  • ophthalmoplegic migraine

We report the details of a patient with an unusual form of ophthalmoplegic migraine resulting in permanent vertical misalignment of the affected eye. The presentation, history, and ophthalmologic examination are reported as well as disease course and follow-up complications. We review the literature on ophthalmoplegic migraine with discussion regarding typical presentation, methods of diagnosis, and other diseases which may cause diagnostic confusion. In light of current case reports on ophthalmoplegic migraine, this is the first documented example with a permanent deficit.