Literature DB >> 21266465

Mutation in the βA3/A1-crystallin gene impairs phagosome degradation in the retinal pigmented epithelium of the rat.

J Samuel Zigler1, Cheng Zhang, Rhonda Grebe, Gitanjali Sehrawat, Laszlo Hackler, Souvonik Adhya, Stacey Hose, D Scott McLeod, Imran Bhutto, Walid Barbour, Geetha Parthasarathy, Donald J Zack, Yuri Sergeev, Gerard A Lutty, James T Handa, Debasish Sinha.   

Abstract

Phagocytosis of the shed outer segment discs of photoreceptors is a major function of the retinal pigmented epithelium (RPE). We demonstrate for the first time that βA3/A1-crystallin, a major structural protein of the ocular lens, is expressed in RPE cells. Further, by utilizing the Nuc1 rat, in which the βA3/A1-crystallin gene is mutated, we show that this protein is required by RPE cells for proper degradation of outer segment discs that have been internalized in phagosomes. We also demonstrate that in wild-type RPE, βA3/A1-crystallin is localized to the lysosomes. However, in the Nuc1 RPE, βA3/A1-crystallin fails to translocate to the lysosomes, perhaps because misfolding of the mutant protein masks sorting signals required for proper trafficking. The digestion of phagocytized outer segments requires a high level of lysosomal enzyme activity, and cathepsin D, the major enzyme responsible for proteolysis of the outer segments, is decreased in mutant RPE cells. Interestingly, our results also indicate a defect in the autophagy process in the Nuc1 RPE, which is probably also linked to impaired lysosomal function, because phagocytosis and autophagy might share common mechanisms in degradation of their targets. βA3/A1-crystallin is a novel lysosomal protein in RPE, essential for degradation of phagocytosed material.

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Year:  2011        PMID: 21266465      PMCID: PMC3031366          DOI: 10.1242/jcs.078790

Source DB:  PubMed          Journal:  J Cell Sci        ISSN: 0021-9533            Impact factor:   5.285


  47 in total

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  34 in total

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Review 7.  Functions of crystallins in and out of lens: roles in elongated and post-mitotic cells.

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Review 9.  Rat models of human diseases and related phenotypes: a systematic inventory of the causative genes.

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Review 10.  Lysosomes: Regulators of autophagy in the retinal pigmented epithelium.

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