Underdeveloped RPE Apical Domain Underlies Lesion Formation in Canine Bestrophinopathies View Full Text


Ontology type: schema:Chapter      Open Access: True


Chapter Info

DATE

2018-05-03

AUTHORS

Karina E. Guziewicz , Emily McTish , Valerie L. Dufour , Kathryn Zorych , Anuradha Dhingra , Kathleen Boesze-Battaglia , Gustavo D. Aguirre

ABSTRACT

Canine bestrophinopathy (cBest) is an important translational model for BEST1-associated maculopathies in man that recapitulates the broad spectrum of clinical and molecular disease aspects observed in patients. Both human and canine bestrophinopathies are characterized by focal to multifocal separations of the retina from the RPE. The lesions can be macular or extramacular, and the specific pathomechanism leading to formation of these lesions remains unclear. We used the naturally occurring canine BEST1 model to examine factors that underlie formation of vitelliform lesions and addressed the susceptibility of the macula to its primary detachment in BEST1-linked maculopathies. More... »

PAGES

309-315

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/978-3-319-75402-4_38

DOI

http://dx.doi.org/10.1007/978-3-319-75402-4_38

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1103767275

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/29721958


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