机构:
UCL, Inst Ophthalmol, London EC1V 9EL, EnglandUCL, Inst Ophthalmol, London EC1V 9EL, England
Jaroszynska, Natalia
[1
]
Harding, Philippa
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h-index: 0
机构:
UCL, Inst Ophthalmol, London EC1V 9EL, EnglandUCL, Inst Ophthalmol, London EC1V 9EL, England
Harding, Philippa
[1
]
Moosajee, Mariya
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机构:
UCL, Inst Ophthalmol, London EC1V 9EL, England
Moorfields Eye Hosp NHS Fdn Trust, London EC1V 2PD, England
Great Ormond St Hosp Children NHS Fdn Trust, London WC1N 3JH, England
Francis Crick Inst, London NW1 1AT, EnglandUCL, Inst Ophthalmol, London EC1V 9EL, England
Moosajee, Mariya
[1
,2
,3
,4
]
机构:
[1] UCL, Inst Ophthalmol, London EC1V 9EL, England
[2] Moorfields Eye Hosp NHS Fdn Trust, London EC1V 2PD, England
[3] Great Ormond St Hosp Children NHS Fdn Trust, London WC1N 3JH, England
Retinal photoreceptors are amongst the most metabolically active cells in the body, consuming more glucose as a metabolic substrate than even the brain. This ensures that there is sufficient energy to establish and maintain photoreceptor functions during and after their differentiation. Such high dependence on glucose metabolism is conserved across vertebrates, including zebrafish from early larval through to adult retinal stages. As the zebrafish retina develops rapidly, reaching an adult-like structure by 72 hours post fertilisation, zebrafish larvae can be used to study metabolism not only during retinogenesis, but also in functionally mature retinae. The interplay between rod and cone photoreceptors and the neighbouring retinal pigment epithelium (RPE) cells establishes a metabolic ecosystem that provides essential control of their individual functions, overall maintaining healthy vision. The RPE facilitates efficient supply of glucose from the choroidal vasculature to the photoreceptors, which produce metabolic products that in turn fuel RPE metabolism. Many inherited retinal diseases (IRDs) result in photoreceptor degeneration, either directly arising from photoreceptor-specific mutations or secondary to RPE loss, leading to sight loss. Evidence from a number of vertebrate studies suggests that the imbalance of the metabolic ecosystem in the outer retina contributes to metabolic failure and disease pathogenesis. The use of larval zebrafish mutants with disease-specific mutations that mirror those seen in human patients allows us to uncover mechanisms of such dysregulation and disease pathology with progression from embryonic to adult stages, as well as providing a means of testing novel therapeutic approaches.
机构:
Fudan Univ, Zhong Shan Hosp, Sch Life Sci, State Key Lab Genet Engn, Shanghai 200438, Peoples R China
East China Normal Univ, Sch Life Sci, Inst Mol Med, Shanghai Key Lab Regulatory Biol, Shanghai 200241, Peoples R ChinaFudan Univ, Zhong Shan Hosp, Sch Life Sci, State Key Lab Genet Engn, Shanghai 200438, Peoples R China
Li, Wenyan
Jin, Daqing
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机构:
East China Normal Univ, Sch Life Sci, Inst Mol Med, Shanghai Key Lab Regulatory Biol, Shanghai 200241, Peoples R ChinaFudan Univ, Zhong Shan Hosp, Sch Life Sci, State Key Lab Genet Engn, Shanghai 200438, Peoples R China
Jin, Daqing
Zhong, Tao P.
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机构:
Fudan Univ, Zhong Shan Hosp, Sch Life Sci, State Key Lab Genet Engn, Shanghai 200438, Peoples R China
East China Normal Univ, Sch Life Sci, Inst Mol Med, Shanghai Key Lab Regulatory Biol, Shanghai 200241, Peoples R ChinaFudan Univ, Zhong Shan Hosp, Sch Life Sci, State Key Lab Genet Engn, Shanghai 200438, Peoples R China
机构:
Univ Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Tsantilas, Kristine A.
Cleghorn, Whitney M.
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Univ Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Cleghorn, Whitney M.
Bisbach, Celia M.
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机构:
Univ Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Bisbach, Celia M.
Whitson, Jeremy A.
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h-index: 0
机构:
Davidson Coll, Dept Biol, Davidson, NC 28036 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Whitson, Jeremy A.
Hass, Daniel T.
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机构:
Univ Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Hass, Daniel T.
Robbings, Brian M.
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机构:
Univ Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Univ Washington, UW Diabet Inst, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Robbings, Brian M.
Sadilek, Martin
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机构:
Univ Washington, Dept Chem, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Sadilek, Martin
Linton, Jonathan D.
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机构:
Univ Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Linton, Jonathan D.
Rountree, Austin M.
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机构:
Univ Washington, UW Diabet Inst, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Rountree, Austin M.
Valencia, Ana P.
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机构:
Univ Washington, Dept Radiol, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Valencia, Ana P.
Sweetwyne, Mariya T.
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机构:
Univ Washington, Dept Lab Med & Pathol, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Sweetwyne, Mariya T.
Campbell, Matthew D.
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机构:
Univ Washington, Dept Radiol, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Campbell, Matthew D.
Zhang, Huiliang
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机构:
Univ Arkansas Med Sci, Coll Med, Dept Pharmacol & Toxicol, Little Rock, AR 72205 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Zhang, Huiliang
Jankowski, Connor S. R.
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机构:
Princeton Univ, Dept Mol Biol, Princeton, NJ USA
Princeton Univ, Lewis Sigler Inst Integrat Genom, Princeton, NJ 08544 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Jankowski, Connor S. R.
Sweet, Ian R.
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h-index: 0
机构:
Univ Washington, UW Diabet Inst, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Sweet, Ian R.
Marcinek, David J.
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机构:
Univ Washington, Dept Radiol, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Marcinek, David J.
Rabinovitch, Peter S.
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机构:
Univ Washington, Dept Lab Med & Pathol, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Rabinovitch, Peter S.
Hurley, James B.
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机构:
Univ Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA
Univ Washington, Dept Ophthalmol, Seattle, WA 98195 USAUniv Washington, Dept Biochem, UW Box 357350,1705 NE Pacific St, Seattle, WA 98195 USA