RT Journal Article SR Electronic T1 HDAC inhibition protects degenerating cone photoreceptors in vivo JF bioRxiv FD Cold Spring Harbor Laboratory SP 049742 DO 10.1101/049742 A1 Dragana Trifunović A1 Blanca Arango-Gonzalez A1 Antonella Comitato A1 Melanie Barth A1 Ayse Sahaboglu A1 Eva M. del Amo A1 Manoj Kulkarni A1 Stefanie M. Hauck A1 Marius Ueffing A1 Arto Urtti A1 Yvan Arsenijevic A1 Valeria Marigo A1 François Paquet-Durand YR 2016 UL http://biorxiv.org/content/early/2016/04/22/049742.abstract AB Retinal diseases caused by cone photoreceptor cell death are devastating as the patients are experiencing loss of accurate and color vision. Understanding the mechanisms of cone cell death and the identification of key players therein could provide new treatment options. We studied the neuroprotective effects of a histone deacetylase inhibitor, Trichostatin A (TSA), in a mouse model of inherited, primary cone degeneration (cpfl1). We show that HDAC inhibition protects cones in vitro, in retinal explant cultures. More importantly, in vivo a single TSA injection increased cone survival for up to 10 days post-injection. In addition, the abnormal, incomplete cone migration pattern in the cpfl1 retina was significantly improved by HDAC inhibition. These findings suggest a crucial role for HDAC activity in primary cone degeneration and highlight a new avenue for future therapy developments for cone dystrophies and diseases associated with impaired cone migration.