Sostdc1 is expressed in all major compartments of developing and adult mammalian eyes.

Valensi, Maud; Goldman, Gabrielle; Marchant, Dominique; Van Den Berghe, Loïc; Jonet, Laurent; Daruich, Alejandra; Robert, Matthieu P.; Krejci, Eric; Klein, Christophe; Mascarelli, Frédéric; Versaux-Botteri, Claudine; Moulin, Alexandre; Putterman, Marc; Guimiot, Fabien; Molina, Thierry; Terris, Benoît; Brémond-Gignac, Dominique; Behar-Cohen, Francine; Abitbol, Marc M.

Purpose: This study was conducted in order to study Sostdc1 expression in rat and human developing and adult eyes. Methods: Using the yeast signal sequence trap screening method, we identified the Sostdc1 cDNA encoding a protein secreted by the adult rat retinal pigment epithelium. We determined by in situ hybridization, RT-PCR, immunohistochemistry, and western blot analysis Sostdc1 gene and protein expression in developing and postnatal rat ocular tissue sections. We also investigated Sostdc1 immunohistolocalization in developing and adult human ocular tissues. Results: We demonstrated a prominent Sostdc1 gene expression in the developing rat central nervous system (CNS) and eyes at early developmental stages from E10.5 days postconception (dpc) to E13 dpc. Specific Sostdc1 immunostaining was also detected in most adult cells of rat ocular tissue sections. We also identified the rat ocular embryonic compartments characterized by a specific Sostdc1 immunohistostaining and specific Pax6, Sox2, Otx2, and Vsx2 immunohistostaining from embryonic stages E10.5 to E13 dpc. Furthermore, we determined the localization of SOSTDC1 immunoreactivity in ocular tissue sections of developing and adult human eyes. Indeed, we detected SOSTDC1 immunostaining in developing and adult human retinal pigment epithelium (RPE) and neural retina (NR) as well as in several developing and adult human ocular compartments, including the walls of choroidal and scleral vessels. Of utmost importance, we observed a strong SOSTDC1 expression in a pathological ocular specimen of type 2 Peters' anomaly complicated by retinal neovascularization as well in the walls ofother pathological extra-ocular vessels. Conclusion: As rat Sostdc1 and human SOSTDC1 are dual antagonists of the Wnt/β-catenin and BMP signaling pathways, these results underscore the potential crucial roles of these pathways and their antagonists, such as Sostdc1 and SOSTDC1, in developing and adult mammalian normal eyes as well as in syndromic and nonsyndromic congenital eye diseases.

WESTERN immunoblotting; RHODOPSIN; CENTRAL nervous system; IN situ hybridization; EYE

Graefe's Archive of Clinical & Experimental Ophthalmology, 2019, Vol 257, Issue 11, p2401

0721-832X

Academic Journal

10.1007/s00417-019-04462-4