Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus · 2013
Cyclic (alternate day) vertical deviation--possible forme fruste of ocular neuromyotonia.
Клинический случайСтатьяНазвание и аннотация: Перевод в очереди
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Roper-Hall Gill, Cruz Oscar A, Espinoza Gabriela M, Chung Sophia M
Аннотация
PURPOSE: Cyclic ocular deviations are relatively uncommon and are seldom seen in adults. We report 3 adult patients with cyclic hypotropia that has clinical characteristics similar to neuromyotonia, suggesting a possible common etiology.
METHODS: Three consecutive patients with 48-hour cyclic hypotropia underwent full neuro-ophthalmologic, oculoplastics, and orthoptic evaluations as well as appropriate medical and neurologic studies. Examinations were arranged on consecutive days on multiple visits to document the cyclic pattern.
RESULTS: All 3 patients had sustained contraction of a vertically acting extraocular muscle lasting 24 hours and demonstrated characteristics of ocular neuromyotonia. The contraction was absent for the next 24 hours. Two of the patients had thyroid eye disease; the third patient had unilateral ophthalmoparesis and had subsequent frameless robotic radiosurgery for a cavernous sinus schwannoma. Regular cycles lasting 6, 9, or 14 months were documented by all 3 patients. In 2 patients, treatment with carbamazepine and gabapentin effectively reduced or eliminated the cycle. The cyclic deviation in the third patient resolved spontaneously.
CONCLUSIONS: On the basis of the response of these patients to membrane-stabilizing medications and the behavior noted as the cycle broke each day, we propose that cyclic vertical strabismus and ocular neuromyotonia may be related conditions with similar underlying physiology.
METHODS: Three consecutive patients with 48-hour cyclic hypotropia underwent full neuro-ophthalmologic, oculoplastics, and orthoptic evaluations as well as appropriate medical and neurologic studies. Examinations were arranged on consecutive days on multiple visits to document the cyclic pattern.
RESULTS: All 3 patients had sustained contraction of a vertically acting extraocular muscle lasting 24 hours and demonstrated characteristics of ocular neuromyotonia. The contraction was absent for the next 24 hours. Two of the patients had thyroid eye disease; the third patient had unilateral ophthalmoparesis and had subsequent frameless robotic radiosurgery for a cavernous sinus schwannoma. Regular cycles lasting 6, 9, or 14 months were documented by all 3 patients. In 2 patients, treatment with carbamazepine and gabapentin effectively reduced or eliminated the cycle. The cyclic deviation in the third patient resolved spontaneously.
CONCLUSIONS: On the basis of the response of these patients to membrane-stabilizing medications and the behavior noted as the cycle broke each day, we propose that cyclic vertical strabismus and ocular neuromyotonia may be related conditions with similar underlying physiology.