Photophobia and abnormally sustained pupil responses in a mouse model of bradyopsia

Adisa Kuburas; Stewart Thompson; Nikolai O Artemyev; Randy H Kardon; Andrew F Russo

doi:10.1167/iovs.14-14784

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Photophobia and abnormally sustained pupil responses in a mouse model of bradyopsia

Journal article

Open access

Peer reviewed

Photophobia and abnormally sustained pupil responses in a mouse model of bradyopsia

Adisa Kuburas, Stewart Thompson, Nikolai O Artemyev, Randy H Kardon and Andrew F Russo

Investigative ophthalmology & visual science, Vol.55(10), pp.6878-6885

09/25/2014

DOI: 10.1167/iovs.14-14784

PMCID: PMC4214208

PMID: 25257059

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Abstract

Mutations in the RGS9 gene cause the visual disorder bradyopsia, which includes difficulty adapting to changes in light and photophobia. The purpose of this study was to determine whether lack of Rgs9 also caused photophobia-like behavior in Rgs9 knockout (Rgs9-/-) mice and to identify useful diagnostic measures of Rgs9 dysfunction. We measured two behavioral responses to light and the pupillary light reflex to determine the form and basis of photophobia in Rgs9-/- mice. Rgs9-/- mice spent less time than wild-type mice in both dim and bright light. The mice also showed increased sensitivity to light in negative masking behavior, with a half maximal response at 0.08 lux (0.01 μW·cm(-2)) in Rgs9-/- mice compared to 5.0 lux (0.85 μW·cm(-2)) in wild-type mice. These behaviors were not due to increased anxiety or increased pupil size causing more light to enter the eye. Rather, constriction of the pupil showed that Rgs9-/- mice had an abnormally sustained response to light across multiple irradiance measurement pathways. Rgs9-/- mice recapitulate a photophobia phenotype of bradyopsia, and the pupil light reflex identifies a simple means to screen for irradiance measurement abnormalities in bradyopsia and potentially other genetic disorders involving photophobia.

Photophobia - physiopathology

Retinal Ganglion Cells - physiology

Eye Diseases, Hereditary - physiopathology

Photophobia - etiology

Eye Diseases, Hereditary - complications

Mice, Inbred C57BL

Male

Reflex, Pupillary - physiology

Animals

Photophobia - metabolism

Female

Mice

Photic Stimulation

Eye Diseases, Hereditary - metabolism

Disease Models, Animal

Details

Title: Subtitle: Photophobia and abnormally sustained pupil responses in a mouse model of bradyopsia
Creators: Adisa Kuburas - Department of Molecular Physiology and Biophysics, University of Iowa, Iowa City, Iowa, United States
Stewart Thompson - Department of Ophthalmology and Visual Sciences, University of Iowa, Iowa City, Iowa, United States
Nikolai O Artemyev - Department of Molecular Physiology and Biophysics, University of Iowa, Iowa City, Iowa, United States
Randy H Kardon - Department of Ophthalmology and Visual Sciences, University of Iowa, Iowa City, Iowa, United States Veterans Affairs Health Care System, Iowa City, Iowa, United States
Andrew F Russo - Department of Molecular Physiology and Biophysics, University of Iowa, Iowa City, Iowa, United States Veterans Affairs Health Care System, Iowa City, Iowa, United States Department of Neurology, University of Iowa, Iowa City, Iowa, United States
Resource Type: Journal article
Publication Details: Investigative ophthalmology & visual science, Vol.55(10), pp.6878-6885
DOI: 10.1167/iovs.14-14784
PMID: 25257059
PMCID: PMC4214208
NLM abbreviation: Invest Ophthalmol Vis Sci
ISSN: 0146-0404
eISSN: 1552-5783
Publisher: United States
Grant note: NS075599 / NINDS NIH HHS R01 NS075599 / NINDS NIH HHS R01 EY012682 / NEI NIH HHS EY12682 / NEI NIH HHS
Language: English
Date published: 09/25/2014
Academic Unit: Neurology; Molecular Physiology and Biophysics; Iowa Neuroscience Institute; Craniofacial Anomalies Research Center; Ophthalmology and Visual Sciences
Record Identifier: 9983979958202771

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