Document Detail


In vivo imaging reveals novel aspects of retinal disease progression in Rs1h(-/Y) mice but no therapeutic effect of carbonic anhydrase inhibition.
MedLine Citation:
PMID:  22672048     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
Purpose  X-linked juvenile retinoschisis (XLRS) is the most common juvenile maculopathy in men and is caused by mutations in the gene encoding retinoschisin (RS1). Evidence in the literature on the therapeutic effect of carboanhydrase inhibitors (CAIs) to treat schisis formation in the retina has remained equivocal. Here, we evaluate the effect of the CAI dorzolamide on the structural and functional disease progression in the mouse model for XLRS (Rs1h(-/y) ). Methods  Rs1h ( -/y ) mice were treated unilaterally with dorzolamide eye drops (Trusopt(®) 20 mg/mL) every 12 h for 2 weeks starting on postnatal day 14 (n = 27). Changes of retinal structure were monitored by confocal scanning laser ophthalmoscopy and spectral domain optical coherence tomography 12 h, 14 days, 4 weeks, 2 months, and 6 months after completion of the treatment. Results  Schisis formation (peak at 3 months) preceded photoreceptor degeneration and hyper-fluorescence (peak at 7 months). Structural pathology was most severe in the superior hemi-retina with previously unreported hyper-fluorescent lesions. Quantitative analysis showed no significant differences regarding the inner or outer retinal thickness of the treated vs. untreated eyes 12 h after the completion of treatment (IRT(12 h)  = -1.29 ± 1.89 μm; ORT(12 h)  = 0.61 ± 2.08 μm; mean ± 95%CI) or at any later time point. Conclusion  Time line analysis after short-term treatment with CAI failed to show short-, intermediate-, or long-term evidence of structural improvement in Rs1h(-/y) mice. Schisis formation in the inner retina peaked at the age of 3 months and was followed by photoreceptor degeneration predominantly in the superior hemi-retina. Previously unreported hyper-fluorescent lesions co-register with structural retinal pathologies.
Authors:
Ahmad Zhour; Sylvia Bolz; Christian Grimm; Gabriel Willmann; Andreas Schatz; Bernhard H F Weber; Eberhart Zrenner; M Dominik Fischer
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Publication Detail:
Type:  JOURNAL ARTICLE     Date:  2012-6-4
Journal Detail:
Title:  Veterinary ophthalmology     Volume:  -     ISSN:  1463-5224     ISO Abbreviation:  -     Publication Date:  2012 Jun 
Date Detail:
Created Date:  2012-6-7     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  100887377     Medline TA:  Vet Ophthalmol     Country:  -    
Other Details:
Languages:  ENG     Pagination:  -     Citation Subset:  -    
Copyright Information:
© 2012 American College of Veterinary Ophthalmologists.
Affiliation:
Centre for Ophthalmology, University of Tübingen, Schleichstr, 12-16, 72076 Tübingen, Germany Laboratory for Retinal Cell Biology, University of Zurich, Wagistrasse 14, 8952 Schlieren, Switzerland Institute of Human Genetics, University of Regensburg, Franz-Josef-Strauß-Allee 11, 93053 Regensburg, Germany.
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