Characterization of Pupil Responses to Blue and Red Light Stimuli in Autosomal Dominant Retinitis Pigmentosa due to<i>NR2E3</i>Mutation

Kawasaki, Aki; Crippa, S.; Kardon, Randy H.; Léon, Lorette; Hamel, Christian

doi:10.1167/iovs.12-10230

Cited by 67 publications

(56 citation statements)

References 19 publications

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“…Based on previous investigations using this apparatus20394546, we defined 4 testing protocols which selected for afferent pupillomotor input derived primarily from synaptic signals from outer retinal photoreceptors (rods and cones) or from its intrinsic melanopsin activity of ipRGCs . For the first three testing protocols, each eye was tested monocularly.…”

Section: Methodsmentioning

confidence: 99%

“…For the rod-weighted and cone-weighted protocols, the immediate pupil contraction to light stimulation was determined as a maximal percentage change from baseline by the equation: (1 - minimum RPS) X 100. The calculated pupil contraction was plotted as a function of stimulus light intensity for each eye of each subject using nonlinear curve fit analysis in order to generate a stimulus response curve which characterized the retinal sensitivity to light over the intensity range of each protocol45. As the response curves appeared to reach an asymptote (maximal response), the light intensity at which a 50% pupil response occurred (logI 50 ) was determined.…”

Section: Methodsmentioning

confidence: 99%

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Differential monocular vs. binocular pupil responses from melanopsin-based photoreception in patients with anterior ischemic optic neuropathy

Tsika

Crippa

Kawasaki

2015

Sci Rep

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We examined the effect of anterior ischemic optic neuropathy (AION) on the activity of intrinsically photosensitive retinal ganglion cells (ipRGCs) using the pupil as proxy. Eighteen patients with AION (10 unilateral, 8 bilateral) and 29 age-matched control subjects underwent chromatic pupillometry. Red and blue light stimuli increasing in 0.5 log steps were presented to each eye independently under conditions of dark and light adaptation. The recorded pupil contraction was plotted against stimulus intensity to generate scotopic and photopic response curves for assessment of synaptically-mediated ipRGC activity. Bright blue light stimuli presented monocularly and binocularly were used for melanopsin activation. The post-stimulus pupil size (PSPS) at the 6th second following stimulus offset was the marker of intrinsic ipRGC activity. Finally, questionnaires were administered to assess the influence of ipRGCs on sleep. The pupil response and PSPS to all monocularly-presented light stimuli were impaired in AION eyes, indicating ipRGC dysfunction. To binocular light stimulation, the PSPS of AION patients was similar to that of controls. There was no difference in the sleep habits of the two groups. Thus after ischemic injury to one or both optic nerves, the summated intrinsic ipRGC activity is preserved when both eyes receive adequate light exposure.

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Section: Methodsmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

Differential monocular vs. binocular pupil responses from melanopsin-based photoreception in patients with anterior ischemic optic neuropathy

Tsika

Crippa

Kawasaki

2015

Sci Rep

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“…[27][28][29] These and similar protocols were successfully used to assess the function of outer and inner retinal cells in patients with retinitis pigmentosa (RP) and patients with RPE65 mutations. 30,31 However, because these methods use a wide light source that stimulates the entire retina, they are not applicable for multifocal testing to identify VF defects.…”

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confidence: 99%

Pupillometer-Based Objective Chromatic Perimetry in Normal Eyes and Patients With Retinal Photoreceptor Dystrophies

Skaat

Sher

Kolker

et al. 2013

Invest. Ophthalmol. Vis. Sci.

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Citation: Skaat A, Sher I, Kolker A, et al. Pupillometer-based objective chromatic perimetry in normal eyes and patients with retinal photoreceptor dystrophies. Invest Ophthalmol Vis Sci. 2013;54;2761 -2770 . DOI:10.1167 PURPOSE. To evaluate a novel objective perimetry using multifocal chromatic pupil light reflex in normal participants and patients with photoreceptor dysfunction, and to relate this new technique with subjective dark-adapted chromatic Goldmann perimetry.METHODS. Thirty-two eyes of 17 retinitis pigmentosa (RP) or cone-rod dystrophy patients and 20 eyes of 12 healthy individuals were tested. A computerized infrared video pupillometer was used to record changes in pupil diameter in response to short-and long-wavelength stimuli (peak 485 and 640 nm, respectively; light intensity 40 cd/m 2 ) at 13 different points of the 308 visual field (VF), under background illumination of 2.7 cd/m 2 . The pupillary response (PR) of patients was compared with PR obtained from normal control participants. In 11 patients, the pupillary responses were also compared with their findings on dark-adapted chromatic Goldmann perimetry. RESULTS.Significantly reduced pupillary responses were obtained in RP patients in response to the short-wavelength stimulus in nearly all perimetric locations (P < 0.03). By contrast, in response to the long-wavelength stimulus, RP patients demonstrated significantly reduced PR mostly in peripheral locations (P 0.02). In a cone-rod dystrophy patient, the PR to both long-and short-wavelength stimuli was significantly lower in the scotoma area identified by the dark-adapted chromatic Goldmann perimetry. In all patients that were tested by the chromatic Goldmann, minimal PR was recorded in areas that were nondetected in the chromatic Goldmann perimetry.CONCLUSIONS. This study demonstrates the potential feasibility of using pupillometer-based chromatic perimetry for objectively assessing VF defects and retinal function in patients with retinal dystrophies. (ClinicalTrials.gov number, NCT01021982.)

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“…Certain commercially available models allow for short testing times and minimal patient cooperation. Such devices are currently used in the clinic and in clinical research to measure retina, optic nerve, oculomotor, and brainstem functions 5,6…”

Section: Introductionmentioning

confidence: 99%

Abnormal pupillary light reflex with chromatic pupillometry in G aucher disease

Narita

Shirai

Kubota

et al. 2014

Ann Clin Transl Neurol

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The hallmark of neuronopathic Gaucher disease (GD) is oculomotor abnormalities, but ophthalmological assessment is difficult in uncooperative patients. Chromatic pupillometry is a quantitative method to assess the pupillary light reflex (PLR) with minimal patient cooperation. Thus, we investigated whether chromatic pupillometry could be useful for neurological evaluations in GD. In our neuronopathic GD patients, red light-induced PLR was markedly impaired, whereas blue light-induced PLR was relatively spared. In addition, patients with non-neuronopathic GD showed no abnormalities. These novel findings show that chromatic pupillometry is a convenient method to detect neurological signs and monitor the course of disease in neuronopathic GD.

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Characterization of Pupil Responses to Blue and Red Light Stimuli in Autosomal Dominant Retinitis Pigmentosa due toNR2E3Mutation

Cited by 67 publications

References 19 publications

Differential monocular vs. binocular pupil responses from melanopsin-based photoreception in patients with anterior ischemic optic neuropathy

Differential monocular vs. binocular pupil responses from melanopsin-based photoreception in patients with anterior ischemic optic neuropathy

Pupillometer-Based Objective Chromatic Perimetry in Normal Eyes and Patients With Retinal Photoreceptor Dystrophies

Abnormal pupillary light reflex with chromatic pupillometry in G aucher disease

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