The association of internuclear ophthalmoplegia (INO) with exotropia in the contralateral eye is a rare finding, known as non-paralytic pontine exotropia (NPPE). We report a case of an 80-year-old woman with acute onset of diplopia on admission who presented with left eye exotropia with left-beating nystagmus whilst fixating with the right eye and inability to adduct the right eye on left gaze. Brain magnetic resonance imaging showed two small areas of vertebrobasilar territory ischaemic stroke, one beneath the inferior portion of the aqueduct and another in the right occipital lobe. Our case highlights an interesting clinical manifestation of brainstem infarction that, along with ocular motility examination, allowed us to review its pathophysiology, including the influence of the contralateral paramedian pontine reticular formation stimulation in the mechanism of contralateral exotropia in NPPE. The fast clinical resolution of these cases can explain the scarcity of NPPE reports.

OBJECTIVE: Unilateral gaze palsy associated with internuclear ophthalmoplegia (INO), i.e., one-and-a-half syndrome, is well known. Exotropia can also be associated with INO, but it has been reported only rarely. We sought to determine the frequencies and courses of gaze palsy and exotropia in INO.
METHODS: Patients hospitalized with acute-onset INO during the period January 2009 through December 2013 were identified from our clinical registry. Associated gaze palsy and exotropia were evaluated in the identified patients.
RESULTS: Twenty-five patients with unilateral INO and 7 patients with bilateral INO were included in this study. Of the 25 patients with unilateral INO, 4 (16.0.0%) had ipsilateral gaze palsy (one-and-a-half syndrome), 8 (32.0%) had exotropia (non-paralytic pontine exotropia), and 6 (24.0%) had both ipsilateral gaze palsy and exotropia (paralytic pontine exotropia). Six (85.7%) of the 7 patients with bilateral INO had exotropia. The gaze palsy persisted more than 1 week in 40.0% of patients, whereas the exotropia disappeared within 1 week in 92.9% of patients when the INO was unilateral.
CONCLUSIONS: Exotropia is not uncommon in the acute stage of INO. However, it is often overlooked because of its short duration.