March 2012
Volume 53, Issue 14
Free
ARVO Annual Meeting Abstract  |   March 2012
Hypopigmented Mice as an Animal Model for Infantile Nystagmus Syndrome
Author Affiliations & Notes
  • Ghislaine L. Traber
    Lab for Retinal Cell Biology, Department of Ophthalmology,
    University Hospital Zurich, Zurich, Switzerland
  • Chien-Cheng Chen
    Department of Neurology,
    University Hospital Zurich, Zurich, Switzerland
  • Ying-Yu Huang
    Department of Neurology,
    University Hospital Zurich, Zurich, Switzerland
  • Marcella Spoor
    Department of Neuroscience, Erasmus MC Rotterdam, Rotterdam, The Netherlands
  • Jeanine Roos
    Department of Neuroscience, Erasmus MC Rotterdam, Rotterdam, The Netherlands
  • Maarten Frens
    Department of Neuroscience, Erasmus MC Rotterdam, Rotterdam, The Netherlands
  • Dominik Straumann
    Department of Neurology,
    University Hospital Zurich, Zurich, Switzerland
  • Christian Grimm
    Lab for Retinal Cell Biology, Department of Ophthalmology,
    University Hospital Zurich, Zurich, Switzerland
  • Footnotes
    Commercial Relationships  Ghislaine L. Traber, None; Chien-Cheng Chen, None; Ying-Yu Huang, None; Marcella Spoor, None; Jeanine Roos, None; Maarten Frens, None; Dominik Straumann, None; Christian Grimm, None
  • Footnotes
    Support  Bangerter-Rhyner-Stiftung; Stiftung OPOS; Hans Imholz-Stiftung, Schweizerischer Fonds zur Verhütung und Bekämpfung der Blindheit; ZIHP, University of Zurich; SNSF
Investigative Ophthalmology & Visual Science March 2012, Vol.53, 521. doi:
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      Ghislaine L. Traber, Chien-Cheng Chen, Ying-Yu Huang, Marcella Spoor, Jeanine Roos, Maarten Frens, Dominik Straumann, Christian Grimm; Hypopigmented Mice as an Animal Model for Infantile Nystagmus Syndrome. Invest. Ophthalmol. Vis. Sci. 2012;53(14):521.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose: : Individuals with oculocutaneous albinism are predisposed to visual system abnormalities affecting the retina and the retinofugal projections, which may lead to reduced visual acuity and infantile nystagmus syndrome (INS). Due to absence of an established mammalian animal model, mechanisms underlying infantile nystagmus syndrome remain incompletely understood. In this study, we screened wildtype mice of varying pigmentation for ocular motor abnormalities in order to identify a possible animal model for INS.

Methods: : Two albino strains (Crl:CD1(ICR), BALB/cAnNTac), the grey DBA/1JbomTac, the brown 129S6/SvEvTac and the black C57BL/6NTac strains were screened using infrared-oculography. Varying visual stimuli (white background, stationary pattern, rotating pattern) were displayed to the full or anterior visual field of the restrained mouse.

Results: : We found spontaneous nystagmus, specifically jerks and oscillations, in hypopigmented mice under all experimental conditions, while ocular positions in pigmented mice were nearly stable (median absolute eye velocity in pigmented strains: mean < 0.4 deg/s; in hypopigmented strains: mean 0.8-3.4 deg/s).In hypopigmented mice, full visual field optokinetic stimuli elicited an optokinetic response (OKR) in the correct direction, albeit with superimposed oscillations. Anterior visual field optokinetic stimuli, however, evoked reversed OKR in these strains. The latter has been reported before for albino mice and is a well known feature of INS.

Conclusions: : Based on our results, we endorse the investigated hypopigmented mouse strains as new animal models for INS.

Keywords: eye movements • nystagmus • ocular motor control 
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