Literature DB >> 23737476

The pupil light reflex in Leber's hereditary optic neuropathy: evidence for preservation of melanopsin-expressing retinal ganglion cells.

Ana Laura A Moura1, Balázs V Nagy, Chiara La Morgia, Piero Barboni, André Gustavo Fernandes Oliveira, Solange R Salomão, Adriana Berezovsky, Milton Nunes de Moraes-Filho, Carlos Filipe Chicani, Rubens Belfort, Valerio Carelli, Alfredo A Sadun, Donald C Hood, Dora Fix Ventura.   

Abstract

PURPOSE: To investigate the pupillary light reflex (PLR) of patients with severe loss of vision due to Leber's Hereditary Optic Neuropathy (LHON) in the context of a proposed preservation of melanopsin-expressing retinal ganglion cells (mRGCs).
METHODS: Ten LHON patients (7 males; 51.6 ± 14.1 years), with visual acuities ranging from 20/400 to hand motion perception and severe visual field losses, were tested and compared with 16 healthy subjects (7 males; 42.15 ± 15.4 years) tested as controls. PLR was measured with an eye tracker and the stimuli were controlled with a Ganzfeld system. Pupil responses were measured monocularly, to 1 second of blue (470 nm) and red (640 nm) flashes with 1, 10, 100, and 250 cd/m² luminances. The normalized amplitude of peak of the transient PLR and the amplitude of the sustained PLR at 6 seconds after the flash offset were measured. In addition, optical coherence topography (OCT) scans of the peripapillary retinal nerve fiber layer were obtained.
RESULTS: The patient's peak PLR responses were on average 15% smaller than controls (P < 0.05), but 5 out of 10 patients had amplitudes within the range of controls. The patients' sustained PLRs were comparable with controls at lower flash intensities, but on average, 27% smaller to the 250 cd/m² blue light, although there was considerable overlap with the PLR amplitudes of control. All patients had severe visual field losses and the retinal nerve fiber layer thickness was reduced to a minimum around the optic disc in 8 of the 10 patients.
CONCLUSIONS: The PLR is maintained overall in LHON patients despite the severity of optic atrophy. These results are consistent with previous evidence of selective preservation of mRGCs.

Entities:  

Keywords:  LHON; OCT; ganglion cells; melanopsin; pupillometry

Mesh:

Substances:

Year:  2013        PMID: 23737476      PMCID: PMC4322722          DOI: 10.1167/iovs.12-11137

Source DB:  PubMed          Journal:  Invest Ophthalmol Vis Sci        ISSN: 0146-0404            Impact factor:   4.799


  33 in total

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Authors:  Beatrix Feigl; Dietmar Mattes; Ravi Thomas; Andrew J Zele
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3.  Toward a clinical protocol for assessing rod, cone, and melanopsin contributions to the human pupil response.

Authors:  Jason C Park; Ana L Moura; Ali S Raza; David W Rhee; Randy H Kardon; Donald C Hood
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6.  Quantification of optic nerve axon loss associated with a relative afferent pupillary defect in the monkey.

Authors:  J B Kerrison; K Buchanan; M L Rosenberg; R Clark; K Andreason; D V Alfaro; H E Grossniklaus; L A Kerrigan-Baumrind; D F Kerrigan; N R Miller; H A Quigley
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9.  A very large Brazilian pedigree with 11778 Leber's hereditary optic neuropathy.

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2.  Effect of stimulus size and luminance on the rod-, cone-, and melanopsin-mediated pupillary light reflex.

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3.  The diagnostic accuracy of chromatic pupillary light responses in diseases of the outer and inner retina.

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

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Review 8.  Medical management of hereditary optic neuropathies.

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9.  Dissociation of Pupillary Post-Illumination Responses from Visual Function in Confirmed OPA1 c.983A > G and c.2708_2711delTTAG Autosomal Dominant Optic Atrophy.

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10.  Melanopsin retinal ganglion cell loss in Alzheimer disease.

Authors:  Chiara La Morgia; Fred N Ross-Cisneros; Yosef Koronyo; Jens Hannibal; Roberto Gallassi; Gaetano Cantalupo; Luisa Sambati; Billy X Pan; Kevin R Tozer; Piero Barboni; Federica Provini; Pietro Avanzini; Michele Carbonelli; Annalisa Pelosi; Helena Chui; Rocco Liguori; Agostino Baruzzi; Maya Koronyo-Hamaoui; Alfredo A Sadun; Valerio Carelli
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