Orthoptic and video‐oculographic analyses in oculopharyngeal muscular dystrophy

Renard, Dimitri; Ferraro, Adelaide; Lorenzini, Marie-Céline; Jeanjean, L.; Portal, Marie-Claire; Llinares, E.; Labauge, Pierre; Castelnovo, Giovanni

doi:10.1002/mus.24600

Cited by 11 publications

(12 citation statements)

References 15 publications

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“…We confirm these findings with similar frequencies of involved EOM, although variability between patients remains high. In CPEO and OPMD, the SR appears to be the most predominantly involved muscle, in line with results from previous studies 35–37…”

Section: Discussionsupporting

confidence: 91%

Diagnosing myasthenia gravis using orthoptic measurements: assessing extraocular muscle fatiguability

Keene

Nie²,

Brink³

et al. 2022

J Neurol Neurosurg Psychiatry

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IntroductionDiagnosing ocular myasthenia gravis (MG) can be challenging because serum antibodies are often not detected. We aimed to explore whether determining extraocular muscle (EOM) weakness using orthoptic measures, including an adapted Hess chart examination, can aid in diagnosing MG.MethodsWe conducted a prospective study among patients with acetylcholine receptor antibody positive MG (20 recently diagnosed, 19 chronic) and 14 seronegative MG patients. We compared orthoptic measures to 19 healthy and 18 disease controls with Graves orbitopathy, chronic progressive external ophthalmoplegia or oculopharyngeal muscular dystrophy. Maximal eye duction angles were measured using a synoptophore. Gaze deviations between eyes were measured using standard Hess chart examination with addition of 1 min persistent gaze to assess MG-associated fatiguability. Receiver operating characteristics curve analysis was performed.ResultsFor duction angles, the area under the curve (AUC) was 0.73 comparing MG to healthy, and 0.69 comparing to patient controls. For the outer field of the Hess chart, the AUC was 0.89 comparing to healthy and 0.54 to patient controls. For drift, the AUC was 0.93 comparing to healthy and 0.93 to patient controls. The sensitivity and specificity of the presence of drift was 81% and 100%.DiscussionOrthoptic measurements can be used to diagnose MG by quantifying EOM weakness and fatiguability. Drift during persistent gaze on a Hess chart is specific for MG and could be used for diagnostic purposes. The Hess chart examination is widely available, inexpensive and fast. Moreover, orthoptic measurements may be a clinically relevant outcome measure for clinical trials.

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Section: Discussionsupporting

confidence: 91%

Diagnosing myasthenia gravis using orthoptic measurements: assessing extraocular muscle fatiguability

Keene

Nie²,

Brink³

et al. 2022

J Neurol Neurosurg Psychiatry

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“…If any of the prescreening questions were answered positively and any of the vision screening shapes were not correctly identified, then the participant was excluded from the study (n = 3). Participants were excluded from the study if they reported any of the following conditions, which may have prevented successful test calibration during the prescreening process: this included vision-related issues such as extreme tropias, phorias, static visual acuity of >20/400, nystagmus, cataracts or eyelash impediments or if they had consumed drugs or alcohol within 24 h of testing (n = 1) (Han et al, 2010;Holmqvist and Nystrom, 2011;Renard et al, 2015;Kooiker et al, 2016;Niehorster et al, 2017). Participants were also excluded if they were unable to pass a nine-point calibration sequence.…”

Section: Methodsmentioning

confidence: 99%

Oculomotor Behavior as a Biomarker for Differentiating Pediatric Patients With Mild Traumatic Brain Injury and Age Matched Controls

Hunfalvay¹,

Murray

Roberts

et al. 2020

Front. Behav. Neurosci.

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“…Participants were excluded from participation in the study if they met any of the following prescreening conditions: neurological disorders (such as concussion, traumatic brain injury, Parkinson's disease, Huntington's disease, cerebral palsy); obvious vision-related issues that prevented successful calibration 24,25 of all nine points (such as extreme tropias, 26 phorias, 26,27 static visual acuity of greater than 20/400, 24 nystagmus, 24,28 cataracts, 29 or eyelash impediments 29 ); small vessel strokes; or consumption of drugs or alcohol within 24 hours of testing. Participants removed any eyewear, including glasses or contact lenses.…”

Section: Methodsmentioning

confidence: 99%

The Reliability, Validity, and Normative Data of Interpupillary Distance and Pupil Diameter Using Eye-Tracking Technology

Murray

Hunfalvay²,

Bolte

2017

Trans. Vis. Sci. Tech.

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PurposeThe purpose of this study was to determine the reliability of interpupillary distance (IPD) and pupil diameter (PD) measures using an infrared eye tracker and central point stimuli. Validity of the test compared to known clinical tools was determined, and normative data was established against which individuals can measure themselves.MethodsParticipants (416) across various demographics were examined for normative data. Of these, 50 were examined for reliability and validity. Validity for IPD measured the test (RightEye IPD/PD) against the PL850 Pupilometer and the Essilor Digital CRP. For PD, the test was measured against the Rosenbaum Pocket Vision Screener (RPVS). Reliability was analyzed with intraclass correlation coefficients (ICC) between trials with Cronbach's alpha (CA) and the standard error of measurement for each ICC. Convergent validity was investigated by calculating the bivariate correlation coefficient.ResultsReliability results were strong (CA > 0.7) for all measures. High positive significant correlations were found between the RightEye IPD test and the PL850 Pupilometer (P < 0.001) and Essilor Digital CRP (P < 0.001) and for the RightEye PD test and the RPVS (P < 0.001).ConclusionsUsing infrared eye tracking and the RightEye IPD/PD test stimuli, reliable and accurate measures of IPD and PD were found. Results from normative data showed an adequate comparison for people with normal vision development.Translational RelevanceResults revealed a central point of fixation may remove variability in examining PD reliably using infrared eye tracking when consistent environmental and experimental procedures are conducted.

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Orthoptic and video‐oculographic analyses in oculopharyngeal muscular dystrophy

Abstract: The data indicate preferential involvement of lateral and (to a lesser degree) superior rectus muscles in OPMD.

Cited by 11 publications

References 15 publications

Diagnosing myasthenia gravis using orthoptic measurements: assessing extraocular muscle fatiguability

Diagnosing myasthenia gravis using orthoptic measurements: assessing extraocular muscle fatiguability

Oculomotor Behavior as a Biomarker for Differentiating Pediatric Patients With Mild Traumatic Brain Injury and Age Matched Controls

The Reliability, Validity, and Normative Data of Interpupillary Distance and Pupil Diameter Using Eye-Tracking Technology

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