Scientific reports · 2020
Sfrp1 deficiency makes retinal photoreceptors prone to degeneration.
СтатьяПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод пока не подготовлен
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Cisneros Elsa, di Marco Fabiana, Rueda-Carrasco Javier, Lillo Concepción, Pereyra Guadalupe, Martín-Bermejo María Jesús, Vargas Alba, Sanchez Rocío, Sandonís África, Esteve Pilar, Bovolenta Paola
Аннотация
Millions of individuals worldwide suffer from impaired vision, a condition with multiple origins that often impinge upon the light sensing cells of the retina, the photoreceptors, affecting their integrity. The molecular components contributing to this integrity are however not yet fully understood. Here we have asked whether Secreted Frizzled Related Protein 1 (SFRP1) may be one of such factors. SFRP1 has a context-dependent function as modulator of Wnt signalling or of the proteolytic activity of A Disintegrin And Metalloproteases (ADAM) 10, a main regulator of neural cell-cell communication. We report that in Sfrp1-/- mice, the outer limiting membrane (OLM) is discontinuous and the photoreceptors disorganized and more prone to light-induced damage. Sfrp1 loss significantly enhances the effect of the Rpe65Leu450Leu genetic variant -present in the mouse genetic background- which confers sensitivity to light-induced stress. These alterations worsen with age, affect visual function and are associated to an increased proteolysis of Protocadherin 21 (PCDH21), localized at the photoreceptor outer segment, and N-cadherin, an OLM component. We thus propose that SFRP1 contributes to photoreceptor fitness with a mechanism that involves the maintenance of OLM integrity. These conclusions are discussed in view of the broader implication of SFRP1 in neurodegeneration and aging.
Предметные рубрики MeSH
ADAM10 ProteinAmyloid Precursor Protein SecretasesAnimalsCadherinsCell CommunicationCell MembraneLightMembrane ProteinsMiceMice, Inbred C57BLMice, KnockoutPhotoreceptor Cells, VertebrateRetinal DegenerationVision DisordersWnt ProteinsWnt Signaling Pathwaycis-trans-IsomerasesRetinoid IsomerohydrolaseSecreted Frizzled-Related Proteins