Effects of acetazolamide on infantile nystagmus syndrome waveforms: comparisons to contact lenses and convergence in a well-studied subject

M J Thurtell; L F Dell'osso; R J Leigh; M Matta; J B Jacobs; R L Tomsak

doi:10.2174/1874364101004010042

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Effects of acetazolamide on infantile nystagmus syndrome waveforms: comparisons to contact lenses and convergence in a well-studied subject

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Effects of acetazolamide on infantile nystagmus syndrome waveforms: comparisons to contact lenses and convergence in a well-studied subject

M J Thurtell, L F Dell'osso, R J Leigh, M Matta, J B Jacobs and R L Tomsak

The open ophthalmology journal, Vol.4(1), pp.42-51

07/29/2010

DOI: 10.2174/1874364101004010042

PMCID: PMC3027082

PMID: 21270949

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Abstract

To determine if acetazolamide, an effective treatment for certain inherited channelopathies, has therapeutic effects on infantile nystagmus syndrome (INS) in a well-studied subject, compare them to other therapies in the same subject and to tenotomy and reattachment (T&R) in other subjects. Eye-movement data were taken using a high-speed digital video recording system. Nystagmus waveforms were analyzed by applying an eXpanded Nystagmus Acuity Function (NAFX) at different gaze angles and determining the Longest Foveation Domain (LFD). Acetazolamide improved foveation by both a 59.7% increase in the peak value of the NAFX function (from 0.395 to 0.580) and a 70% broadening of the NAFX vs Gaze Angle curve (the LFD increased from 20° to 34°). The resulting U-shaped improvement in the percent NAFX vs Gaze Angle curve, varied from ~60% near the NAFX peak to over 1000% laterally. The therapeutic improvements in NAFX from acetazolamide (similar to T&R) were intermediate between those of soft contact lenses and convergence, the latter was best; for LFD improvements, acetazolamide and contact lenses were equivalent and less effective than convergence. Computer simulations suggested that damping the central oscillation driving INS was insufficient to produce the foveation improvements and increased NAFX values. Acetazolamide resulted in improved-foveation INS waveforms over a broadened range of gaze angles, probably acting at more than one site. This raises the question of whether hereditary INS involves an inherited channelopathy, and whether other agents with known effects on ion channels should be investigated as therapy for this condition.

foveation

Infantile nystagmus syndrome

waveforms

drug

Details

Title: Subtitle: Effects of acetazolamide on infantile nystagmus syndrome waveforms: comparisons to contact lenses and convergence in a well-studied subject
Creators: M J Thurtell - Daroff-Dell'Osso Ocular Motility Laboratory, Louis Stokes Cleveland Department of Veterans Affairs Medical Center and CASE Medical School, USA
L F Dell'osso
R J Leigh
M Matta
J B Jacobs
R L Tomsak
Resource Type: Journal article
Publication Details: The open ophthalmology journal, Vol.4(1), pp.42-51
DOI: 10.2174/1874364101004010042
PMID: 21270949
PMCID: PMC3027082
NLM abbreviation: Open Ophthalmol J
ISSN: 1874-3641
eISSN: 1874-3641
Publisher: United Arab Emirates
Language: English
Date published: 07/29/2010
Academic Unit: Neurology; Ophthalmology and Visual Sciences
Record Identifier: 9983980061402771

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