Article
Inhibition of ceramide accumulation in AdipoR1-/- mice increases photoreceptor survival and improves vision.
JCI insight - 22 Feb 2022
Lewandowski Dominik, Foik Andrzej T, Smidak Roman, Choi Elliot H, Zhang Jianye, Hoang Thanh, Tworak Aleksander, Suh Susie, Leinonen Henri, Dong Zhiqian, Pinto Antonio Fm, Tom Emily, Luu Jennings, Lee Joan, Ma Xiuli, Bieberich Erhard, Blackshaw Seth, Saghatelian Alan, Lyon David C, Skowronska-Krawczyk Dorota, Tabaka Marcin, Palczewski Krzysztof
Abstract excerpt
Adiponectin receptor 1 (ADIPOR1) is a lipid and glucose metabolism regulator that possesses intrinsic ceramidase activity. Mutations of the ADIPOR1 gene have been associated with nonsyndromic and syndromic retinitis pigmentosa. Here, we show that the absence of AdipoR1 in mice leads to progressive photoreceptor degeneration, significant reduction of electroretinogram amplitudes, decreased retinoid content in the...
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