A 48-year-old man with atrial septal defect abruptly developed upward and downward gaze palsy with a loss of vertical oculocephalic reflex and smooth pursuit, convergence palsy, concomitant skew deviation with left-sided hypotropia, and bilateral light-near dissociation. Cranial magnetic resonance imaging demonstrated a localized infarction in the left-sided rostral and dorsal midbrain. Neuro-ophthalmologic findings suggested that the left-sided rostral interstitial nucleus of the medial longitudinal fasciculus, left-sided interstitial nucleus of Cajal, and posterior commissure might be involved. There were not convergence retraction nystagmus, and eyelids retraction and lag, probably because the nucleus of the posterior commissure might be spared.
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