Journal
NATURE COMMUNICATIONS
Volume 13, Issue 1, Pages -Publisher
NATURE PORTFOLIO
DOI: 10.1038/s41467-021-27935-9
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Funding
- NIH [RO1 EY 032966, EY 029428, RR0293T00]
- RPB
- Stein Innovation Award from Research To Prevent Blindness (RPB)
- Alcon Research Award
- Research to Prevent Blindness [EY015240, EY028916]
- Paul and Evanina Bell Mackall Foundation Trust
- Intramural Research Program of the NIH
- National Cancer Institute
- Intramural Research Funds from the N.E.I
- Simons Foundation [SF349247]
- NYSTAR
- NIH National Institute of General Medical Sciences [GM103310]
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In this study, researchers observed that mice lacking chloride intracellular channel 4 in retinal pigment epithelium exhibited a full spectrum of functional and pathological features of dry AMD, including drusen formation. Multidisciplinary longitudinal studies revealed a mechanistic link between RPE cell-autonomous aberrant lipid metabolism and transport and drusen formation.
Age-related macular degeneration (AMD) is the leading cause of blindness among the elderly. Dry AMD has unclear etiology and no treatment. Lipid-rich drusen are the hallmark of dry AMD. An AMD mouse model and insights into drusenogenesis are keys to better understanding of this disease. Chloride intracellular channel 4 (CLIC4) is a pleomorphic protein regulating diverse biological functions. Here we show that retinal pigment epithelium (RPE)-specific Clic4 knockout mice exhibit a full spectrum of functional and pathological hallmarks of dry AMD. Multidisciplinary longitudinal studies of disease progression in these mice support a mechanistic model that links RPE cell-autonomous aberrant lipid metabolism and transport to drusen formation. Age-related macular degeneration (AMD) is a leading cause of blindness and is characterised by the accumulation of lipid deposits, called drusen. Here, the authors show that mice lacking chloride intracellular channel 4 in retinal pigment epithelium have defective lipid processing in the eye and pathological features mirroring human AMD, including drusen formation.
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