Cyclic (alternate day) vertical deviation—possible forme fruste of ocular neuromyotonia

Roper-Hall, Gill; Cruz, Oscar A.; Chung, Sophia M.

doi:10.1016/j.jaapos.2012.12.156

Cited by 11 publications

(8 citation statements)

References 22 publications

(27 reference statements)

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“…In our review of the literature, 50% of reported cases of ocular neuromyotonia had ocular misalignment in primary gaze [1-3, 5-36]. There are 4 single case reports of strabismus surgery in patients with ocular neuromyotonia and an additional case series of strabismus surgery in 3 patients with a cyclic vertical deviation that the authors describe as a “forme fruste ocular neuromyotonia” (3, 6, 7, 30, 36).…”

Section: Discussionmentioning

confidence: 99%

“…There are 4 single case reports of strabismus surgery in patients with ocular neuromyotonia and an additional case series of strabismus surgery in 3 patients with a cyclic vertical deviation that the authors describe as a “forme fruste ocular neuromyotonia” (3, 6, 7, 30, 36). All seven patients had undergone recession of the neuromyotonic muscle with resolution of paroxysms post-operatively.…”

Section: Discussionmentioning

confidence: 99%

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Strabismus Surgery in Patients With Ocular Neuromyotonia: Potential Unmasking of the Condition and Effective Management Tool

Kim

Velez

Pineles

2016

Journal of Neuro-Ophthalmology

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Background Ocular neuromyotonia is a rare motility disorder in which paroxysms of tonic extraocular muscle contraction from abnormal ocular motor nerve firing result in episodic diplopia and strabismus. Medical therapy with membrane-stabilizing agents has varied success. A surgical approach to treatment has not yet been described. We report the outcomes of strabismus surgery in patients with ocular neuromyotonia. Methods We describe three patients with sixth nerve paresis and ocular neuromyotonia of the affected lateral rectus muscle who underwent strabismus surgery. All patients had a history of radiation therapy for intracranial tumors. Ophthalmologic and orthoptic examinations were performed with appropriate medical and neuroradiologic evaluation. Pre-operative and post-operative data are presented and analyzed. Results Two patients were noted to have ocular neuromyotonia after their first strabismus surgery for a sixth nerve palsy. Patients 1 and 2 had three surgeries while patient 3 had one operation. Extraocular muscles operated on included the medial rectus and lateral rectus. Pre-operative primary gaze baseline esotropia ranged from 35 to 75 prism diopters (Δ). All patients achieved improvement in ocular alignment and motility. Post-operative primary gaze deviations ranged from orthotropia to 20Δ of esotropia. Abduction deficits were unchanged or improved. The follow-up period ranged from 15 months to 2 years. Conclusions Patients with ocular neuromyotonia of a paretic rectus muscle can achieve binocular fusion with strabismus surgery. Ocular neuromyotonia may manifest post-operatively in patients with a sixth nerve palsy and a contractured medial rectus who, pre-operatively, were not noted to have ocular neuromyotonia.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Strabismus Surgery in Patients With Ocular Neuromyotonia: Potential Unmasking of the Condition and Effective Management Tool

Kim

Velez

Pineles

2016

Journal of Neuro-Ophthalmology

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“…During the follow-up period, it decreased and subsided along with spontaneous remission of her thyroid eye disease and normalization of her thyroid hormonal level without any medical or surgical management. Roper-Hall et al reported that cyclic vertical deviation could be a possible forme fruste of ocular neuromyotonia with similar neurophysiological mechanisms [ 4 ]. Ocular neuromyotonia is characterized by brief involuntary episodic contractions of ocular muscles, with a sustained tonic contraction, with episodes lasting from seconds to 3–4 min [ 7 ].…”

Section: Discussionmentioning

confidence: 99%

“…Disruption of the innate biological clock and involvement of the suprachiasmatic nucleus of the hypothalamus are two proposed mechanisms [ 2 ]. A relationship between cyclic vertical deviation and ocular neuromyotonia was recently suggested with similar neurophysiological mechanisms [ 4 ]. We herein present a patient with a 48-h cycle of vertical deviation associated with dysthyroid ophthalmopathy that spontaneously resolved.…”

Section: Introductionmentioning

confidence: 99%

A cyclic vertical deviation with dysthyroid ophthalmopathy: a case report

2016

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BackgroundCyclic strabismus is a very rare condition of ocular motility characterized by alternating strabismus and orthotropia. We report a patient with a 48-h cycle of vertical deviation associated with dysthyroid ophthalmopathy that spontaneously resolved.Case presentationA 40-year-old woman experienced sudden onset vertical double vision. She had already been diagnosed with hyperthyroidism. Asymmetric enlargement of the inferior and medial rectus muscles was demonstrated in her left eye on computed tomography, and was compatible with dysthyroid ophthalmopathy. She clearly described the “every-other-day” pattern of diplopia that spontaneously switched around 8 o’clock every night. She exhibited left hypotropia of 35 prism diopters on a bad day, and 7 prism diopters on a good day.ConclusionsThis case illustrates that cyclic vertical deviation may spontaneously resolve along with the course of the underlying diseases that initiated the cyclic deviation, especially in patients with conditions involving motor component changes such as dysthyroid ophthalmopathy.

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“…The appearance during an episode when it ''switched'' was comparable to the onset and recovery of an episode of ONM. 22 Differential diagnosis in acquired intermittent diplopia includes decompensating heterotropia, myasthenia gravis, superior oblique click syndrome, superior oblique myokymia, and ocular neuromyotonia. In patients describing episodic diplopia, careful examination should be undertaken to see if the pattern fits ONM.…”

Section: Discussionmentioning

confidence: 99%

Ocular neuromyotonia: differential diagnosis and treatment

2013

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Ocular neuromyotonia (ONM) is a rare but distinctive clinical entity characterized by involuntary episodic contraction of one or more muscles supplied by the ocular motor nerves. A retrospective review was conducted on all patients with ONM seen by the neuroophthalmology service in the past 20 years. Ten patients were identified with ONM; six affecting vertical muscles (superior oblique; inferior rectus; superior rectus) and four affecting lateral rectus muscles. Case 1 has been reported previously. Most episodes occurred every 10-40 min, lasted a few seconds to several minutes, and were repeated throughout the day. Only two patients had previously undergone cranial radiation. Two had thyroid eye disease. One patient presented with superior oblique myokymia and subsequently developed ONM. Membrane stabilizing medications were prescribed in 7 of the 10 patients with varied success. ONM episodes ceased after extraocular muscle surgery in one patient with thyroid eye disease.

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Cyclic (alternate day) vertical deviation—possible forme fruste of ocular neuromyotonia

Cited by 11 publications

References 22 publications

Strabismus Surgery in Patients With Ocular Neuromyotonia: Potential Unmasking of the Condition and Effective Management Tool

Strabismus Surgery in Patients With Ocular Neuromyotonia: Potential Unmasking of the Condition and Effective Management Tool

A cyclic vertical deviation with dysthyroid ophthalmopathy: a case report

Ocular neuromyotonia: differential diagnosis and treatment

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