April 2014
Volume 55, Issue 13
Free
ARVO Annual Meeting Abstract  |   April 2014
Efficacy and Safety of New Topical Sodium Pump Inhibitor (NSPI) in Reducing Eye Oscillations in A Canine model of Infantile Nystagmus Syndrome (INS)
Author Affiliations & Notes
  • Richard W Hertle
    Ophthalmology, Children's Hosp Medical Ctr of Akron, Akron, OH
    Ophthalmology, SUMMA Medical Center, Akron, OH
  • Jeffrey Dunmire
    Ophthalmology, SUMMA Medical Center, Akron, OH
  • Louis F Dell'Osso
    Neurology, Case Western Reserve Univ, Cleveland, OH
    Daroff-Dellosso Ocular Motility Lab, Cleveland VA Med Ctr, Cleveland, OH
  • Jonathan B Jacobs
    Neurology, Case Western Reserve Univ, Cleveland, OH
    Daroff-Dellosso Ocular Motility Lab, Cleveland VA Med Ctr, Cleveland, OH
  • Lauren A Dalvin
    Graduate Medical Education, Mayo Clinic, Rochester, MN
  • Dongsheng Yang
    Ophthalmology, Children's Hosp Medical Ctr of Akron, Akron, OH
  • Michelle Evano-Chapman
    Ophthalmology, SUMMA Medical Center, Akron, OH
  • Footnotes
    Commercial Relationships Richard Hertle, US FDA (P), Vision of Children (F); Jeffrey Dunmire, None; Louis Dell'Osso, None; Jonathan Jacobs, None; Lauren Dalvin, None; Dongsheng Yang, None; Michelle Evano-Chapman, None
  • Footnotes
    Support None
Investigative Ophthalmology & Visual Science April 2014, Vol.55, 4546. doi:https://doi.org/
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      Richard W Hertle, Jeffrey Dunmire, Louis F Dell'Osso, Jonathan B Jacobs, Lauren A Dalvin, Dongsheng Yang, Michelle Evano-Chapman; Efficacy and Safety of New Topical Sodium Pump Inhibitor (NSPI) in Reducing Eye Oscillations in A Canine model of Infantile Nystagmus Syndrome (INS). Invest. Ophthalmol. Vis. Sci. 2014;55(13):4546. doi: https://doi.org/.

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

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Abstract
 
Purpose
 

To determine the ocular and systemic safety and efficacy of using a new topical medication in a canine model of INS. The rationale for this proposal is based on a case study of a patient whose INS improved after inadvertent exposure to a agent containing the NSPI.

 
Methods
 

After in vitro safety testing and IUCAC approval we studied increasing concentrations of topical NSPI drops (.002% to 0.7%) in canines with a purposely bred defect in the RPE65 gene resulting in both retinal degeneration and INS. We collected data on ocular and systemic effects and performed eye-movement recordings (EMR) using a remote video eye movement system with a protocol previously shown valid and reliable in canines.

 
Results
 

At the 0.7% concentration dose of NSPI there was minimal, reversible, conjunctival hyperemia. There was no other ocular or systemic toxicity. At the 0.6% dose there was a visible decrease in the INS and EMR showed a 150%-250% increase in NAFX and 30%-70% decrease (p<0.001) in amplitude across gaze. There was also a sustained (40%-60%) significant (p<.05) decrease in intraocular pressure from baseline.

 
Conclusions
 

This animal study suggests this new pharmacological agent has potential for topical treatment of both INS and diseases with raised intraocular pressure. Further, this new treatment approach confirms the importance of extraocular muscle proprioception in ocular motor diseases and their treatment.

  
Keywords: 619 nystagmus • 522 eye movements • 725 strabismus: treatment  
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