FRINK Linked Data Fragments server

Matches in SemOpenAlex for { <https://semopenalex.org/work/W2654510016> ?p ?o ?g. }

• W2654510016 endingPage "e6" @default.
• W2654510016 startingPage "e4" @default.
• W2654510016 abstract "Unilateral ocular neuromyotonia (ONM) is a rare entity, and bilateral ONM even more so, with and only 1 case having been reported previously in the literature. We present a rare case of bilateral ocular neuromyotonia whose course of treatment was complicated by side effects of carbamazepine. A 35-year-old female was referred by her oncologist with a 12-month history of blurring that was most noticeable on right lateral gaze. During the episodes of blurring, she felt a tugging sensation that could last for up to 1 minute. These episodes had become more frequent over the past 6 months, increasing to 6 to 7 times per day. Notable medical history included nasopharyngeal cancer (NPC), which was treated with 4 cycles of cisplatin and 5-fluorouracil (5FU) and concurrent daily radiotherapy for 6.5 weeks, 8 years prior. One month after completing her treatment, she experienced paraesthesia in her left lower face, neck, and left arm. Investigation of this led to a diagnosis of hypopituitarism and hypothyroidism, well controlled with growth hormone injections and thyroxine. Four years later, she experienced dysphagia, which was attributed to late deterioration from radiotherapy-induced damage. Her dysphagia caused significant weight loss, and she was referred for a feeding tube. On examination, best-corrected visual acuity was –0.15 logMAR bilaterally. At rest, there was a stable near exophoria of 4 prism diopters. In primary position, the patient was orthophoric. After sustained gaze to the right, the divergent deviation increased to 25 prism diopters. After sustained gaze to the left, the divergent deviation increased to a lesser extent by 16 prism diopters. Following prolonged gaze testing, she was noted to have reduced adduction of the right eye with left abducting nystagmus. She also was noted to have reduced adduction of the left eye. Horizontal eye movements are demonstrated in Fig. 1. After sustained upgaze, she consistently showed upper lid lag in depressed positions (bilateral pseudo von Graefe sign) and a depression deficit in the left eye (Fig. 2). There was no anisocoria, and both pupils were reactive to light and accommodation.Fig. 2Vertical eye movements. Direction of gaze indicated on image by arrow or stated as being in primary position (1°). (A) Primary position: no abnormality seen; (B) left lid lag on down gaze; (C) downgaze after sustained upgaze: left levator and SR myotonia; and (D) primary position.View Large Image Figure ViewerDownload Hi-res image Download (PPT) Anterior segment intraocular pressures and fundus examination were unremarkable. Investigations included serology, electromyography (EMG), and magnetic resonance imaging (MRI). Serology, including acetylcholine receptor antibodies, was unremarkable. The divergent position of the eyes was noted during the short tau inversion recovery sequence of the MRI. Repetitive stimulation EMG and single-fibre EMG were normal. Findings were consistent with bilateral ocular neuromyotonia, and treatment with carbamazepine 200 mg twice a day was initiated. She responded well to treatment, and her symptoms resolved after 1 week. However, she developed a widespread itchy skin rash. Her treatment was reduced to 100 mg twice daily, her rash resolved, and her symptoms remained controlled. ONM is a rare clinical entity, characterized by episodic involuntary contraction of ≥1 extraocular muscles.1Roper-Hall G. Chung S.M. Cruz O.A. Ocular neuromyotonia: differential diagnosis and treatment.Strabismus. 2013; 21: 131-136Crossref PubMed Scopus (29) Google Scholar, 2Plant G.T. Putting ocular neuromyotonia in context.J Neuroophthalmol. 2006; 26: 241-243Crossref PubMed Scopus (8) Google Scholar, 3Salchow D.J. Wermund T.K. Abducens neuromyotonia as the presenting sign of an intracranial tumor.J Neuroophthalmol. 2011; 31: 34-37Crossref PubMed Scopus (15) Google Scholar The most common cranial nerve affected is the oculomotor nerve,3Salchow D.J. Wermund T.K. Abducens neuromyotonia as the presenting sign of an intracranial tumor.J Neuroophthalmol. 2011; 31: 34-37Crossref PubMed Scopus (15) Google Scholar followed by the trochlear and abducens nerves.4Choi K.D. Hwang J.M. Park S.H. Kim J.S. Primary aberrant regeneration and neuromyotonia of the third cranial nerve.J Neuroophthalmol. 2006; 26: 248-250Crossref PubMed Scopus (22) Google Scholar There is no preponderance for either sex.1Roper-Hall G. Chung S.M. Cruz O.A. Ocular neuromyotonia: differential diagnosis and treatment.Strabismus. 2013; 21: 131-136Crossref PubMed Scopus (29) Google Scholar To our knowledge, ONM has been reported to be bilateral in only 1 other case in the literature.5Morrow M.J. Kao G.W. Arnold A.C. Bilateral ocular neuromyotonia: oculographic correlations.Neurology. 1996; 46: 264-266Crossref PubMed Scopus (31) Google Scholar The spasmodic neuronal dischargers in ONM result in deviation of the affected eye accompanied by diplopia.6de Saint Sardos A. Vincent A. Aroichane M. Ospina L.H. Ocular neuromyotonia in a 15-year-old girl after radiation therapy.J AAPOS. 2008; 12: 616-617Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar Symptoms can occur spontaneously but are usually elicited after a period of prolonged gaze in the direction of the affected muscle.1Roper-Hall G. Chung S.M. Cruz O.A. Ocular neuromyotonia: differential diagnosis and treatment.Strabismus. 2013; 21: 131-136Crossref PubMed Scopus (29) Google Scholar Length of episodes can vary from seconds to several minutes.1Roper-Hall G. Chung S.M. Cruz O.A. Ocular neuromyotonia: differential diagnosis and treatment.Strabismus. 2013; 21: 131-136Crossref PubMed Scopus (29) Google Scholar, 3Salchow D.J. Wermund T.K. Abducens neuromyotonia as the presenting sign of an intracranial tumor.J Neuroophthalmol. 2011; 31: 34-37Crossref PubMed Scopus (15) Google Scholar, 6de Saint Sardos A. Vincent A. Aroichane M. Ospina L.H. Ocular neuromyotonia in a 15-year-old girl after radiation therapy.J AAPOS. 2008; 12: 616-617Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar The pathophysiology behind the aberrant firing of the affected neurons is still debated but is thought to be linked to unstable neuronal membranes, especially in view of its successful treatment with membrane-stabilizing medications.3Salchow D.J. Wermund T.K. Abducens neuromyotonia as the presenting sign of an intracranial tumor.J Neuroophthalmol. 2011; 31: 34-37Crossref PubMed Scopus (15) Google Scholar, 6de Saint Sardos A. Vincent A. Aroichane M. Ospina L.H. Ocular neuromyotonia in a 15-year-old girl after radiation therapy.J AAPOS. 2008; 12: 616-617Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar As in the case presented here, ONM often is associated with radiation therapy (≤60% of cases) and can present as late as 18 years after treatment.1Roper-Hall G. Chung S.M. Cruz O.A. Ocular neuromyotonia: differential diagnosis and treatment.Strabismus. 2013; 21: 131-136Crossref PubMed Scopus (29) Google Scholar, 2Plant G.T. Putting ocular neuromyotonia in context.J Neuroophthalmol. 2006; 26: 241-243Crossref PubMed Scopus (8) Google Scholar, 3Salchow D.J. Wermund T.K. Abducens neuromyotonia as the presenting sign of an intracranial tumor.J Neuroophthalmol. 2011; 31: 34-37Crossref PubMed Scopus (15) Google Scholar, 6de Saint Sardos A. Vincent A. Aroichane M. Ospina L.H. Ocular neuromyotonia in a 15-year-old girl after radiation therapy.J AAPOS. 2008; 12: 616-617Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar, 7Giardina A.S. Slagle W.S. Greene A.M. Musick A.N. Eckermann D.R. Novel case of ocular neuromyotonia associated with thyroid-related orbitopathy and literature review.Optom Vis Sci. 2012; 89: e124-e134Crossref PubMed Scopus (14) Google Scholar Of particular note is the suggestion by Kau et al that cisplatin and 5FU, which were used in the present case, are known to cause cranial nerve toxicity. It was hypothesized this may increase sensitivity to radiation and increase the chance of developing ONM.8Kau H.C. Tsai C.C. Abducens ocular neuromyotonia in a patient with nasopharyngeal carcinoma following concurrent chemoradiotherapy.J Neuroophthalmol. 2010; 30: 266-267Crossref PubMed Scopus (9) Google Scholar Differential diagnosis of ONM includes ocular myasthenia gravis, demyelination, cyclic oculomotor paresis, superior oblique myokymia, cyclic oculomotor paresis, and Graves’ disease.9Lee K. Aui-Aree N. DP A. A 54 year old man with intermittent diplopia.Digit J Ophthalmol. 2005; : 11Google Scholar Appropriate serologic and EMG testing should be undertaken. Imaging is imperative to exclude any space-occupying legion,3Salchow D.J. Wermund T.K. Abducens neuromyotonia as the presenting sign of an intracranial tumor.J Neuroophthalmol. 2011; 31: 34-37Crossref PubMed Scopus (15) Google Scholar, 6de Saint Sardos A. Vincent A. Aroichane M. Ospina L.H. Ocular neuromyotonia in a 15-year-old girl after radiation therapy.J AAPOS. 2008; 12: 616-617Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar particularly in those patients with a history of nasopharyngeal cancer (NPC), as NPC is associated with cranial nerve palsy in nearly 25% of cases.8Kau H.C. Tsai C.C. Abducens ocular neuromyotonia in a patient with nasopharyngeal carcinoma following concurrent chemoradiotherapy.J Neuroophthalmol. 2010; 30: 266-267Crossref PubMed Scopus (9) Google Scholar Management includes treatment with carbamazepine, with dosage varying from 100 to 600 mg.7Giardina A.S. Slagle W.S. Greene A.M. Musick A.N. Eckermann D.R. Novel case of ocular neuromyotonia associated with thyroid-related orbitopathy and literature review.Optom Vis Sci. 2012; 89: e124-e134Crossref PubMed Scopus (14) Google Scholar A good response has been reported, with 87.8% responding to this treatment.7Giardina A.S. Slagle W.S. Greene A.M. Musick A.N. Eckermann D.R. Novel case of ocular neuromyotonia associated with thyroid-related orbitopathy and literature review.Optom Vis Sci. 2012; 89: e124-e134Crossref PubMed Scopus (14) Google Scholar However, as demonstrated with the present case, side effects may complicate management. The authors have no proprietary or commercial interest in any materials discussed in this article." @default.
• W2654510016 created "2017-06-30" @default.
• W2654510016 creator A5066598911 @default.
• W2654510016 creator A5072995378 @default.
• W2654510016 creator A5088490461 @default.
• W2654510016 date "2018-02-01" @default.
• W2654510016 modified "2023-09-23" @default.
• W2654510016 title "A rare case of bilateral ocular neuromyotonia" @default.
• W2654510016 cites W1976757192 @default.
• W2654510016 cites W2033303134 @default.
• W2654510016 cites W2035366023 @default.
• W2654510016 cites W2074540430 @default.
• W2654510016 cites W2079878855 @default.
• W2654510016 cites W2150170499 @default.
• W2654510016 cites W2318997653 @default.
• W2654510016 cites W2327241740 @default.
• W2654510016 doi "https://doi.org/10.1016/j.jcjo.2017.05.011" @default.
• W2654510016 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/29426461" @default.
• W2654510016 hasPublicationYear "2018" @default.
• W2654510016 type Work @default.
• W2654510016 sameAs 2654510016 @default.
• W2654510016 citedByCount "4" @default.
• W2654510016 countsByYear W26545100162018 @default.
• W2654510016 countsByYear W26545100162021 @default.
• W2654510016 countsByYear W26545100162022 @default.
• W2654510016 countsByYear W26545100162023 @default.
• W2654510016 crossrefType "journal-article" @default.
• W2654510016 hasAuthorship W2654510016A5066598911 @default.
• W2654510016 hasAuthorship W2654510016A5072995378 @default.
• W2654510016 hasAuthorship W2654510016A5088490461 @default.
• W2654510016 hasBestOaLocation W26545100161 @default.
• W2654510016 hasConcept C118487528 @default.
• W2654510016 hasConcept C159654299 @default.
• W2654510016 hasConcept C203014093 @default.
• W2654510016 hasConcept C2778810854 @default.
• W2654510016 hasConcept C71924100 @default.
• W2654510016 hasConceptScore W2654510016C118487528 @default.
• W2654510016 hasConceptScore W2654510016C159654299 @default.
• W2654510016 hasConceptScore W2654510016C203014093 @default.
• W2654510016 hasConceptScore W2654510016C2778810854 @default.
• W2654510016 hasConceptScore W2654510016C71924100 @default.
• W2654510016 hasIssue "1" @default.
• W2654510016 hasLocation W26545100161 @default.
• W2654510016 hasLocation W26545100162 @default.
• W2654510016 hasOpenAccess W2654510016 @default.
• W2654510016 hasPrimaryLocation W26545100161 @default.
• W2654510016 hasRelatedWork W1506200166 @default.
• W2654510016 hasRelatedWork W1995515455 @default.
• W2654510016 hasRelatedWork W2048182022 @default.
• W2654510016 hasRelatedWork W2080531066 @default.
• W2654510016 hasRelatedWork W2604872355 @default.
• W2654510016 hasRelatedWork W2748952813 @default.
• W2654510016 hasRelatedWork W2899084033 @default.
• W2654510016 hasRelatedWork W3031052312 @default.
• W2654510016 hasRelatedWork W3032375762 @default.
• W2654510016 hasRelatedWork W3108674512 @default.
• W2654510016 hasVolume "53" @default.
• W2654510016 isParatext "false" @default.
• W2654510016 isRetracted "false" @default.
• W2654510016 magId "2654510016" @default.
• W2654510016 workType "article" @default.