Literature DB >> 33477551

A Splice Variant in SLC16A8 Gene Leads to Lactate Transport Deficit in Human iPS Cell-Derived Retinal Pigment Epithelial Cells.

Laurence Klipfel1, Marie Cordonnier1, Léa Thiébault1, Emmanuelle Clérin1, Frédéric Blond1, Géraldine Millet-Puel1, Saddek Mohand-Saïd2, Olivier Goureau1, José-Alain Sahel1,2, Emeline F Nandrot1, Thierry Léveillard1.   

Abstract

Age-related macular degeneration (AMD) is a blinding disease for which most of the patients remain untreatable. Since the disease affects the macula at the center of the retina, a structure specific to the primate lineage, rodent models to study the pathophysiology of AMD and to develop therapies are very limited. Consequently, our understanding relies mostly on genetic studies highlighting risk alleles at many loci. We are studying the possible implication of a metabolic imbalance associated with risk alleles within the SLC16A8 gene that encodes for a retinal pigment epithelium (RPE)-specific lactate transporter MCT3 and its consequences for vision. As a first approach, we report here the deficit in transepithelial lactate transport of a rare SLC16A8 allele identified during a genome-wide association study. We produced induced pluripotent stem cells (iPSCs) from the unique patient in our cohort that carries two copies of this allele. After in vitro differentiation of the iPSCs into RPE cells and their characterization, we demonstrate that the rare allele results in the retention of intron 2 of the SLC16A8 gene leading to the absence of MCT3 protein. We show using a biochemical assay that these cells have a deficit in transepithelial lactate transport.

Entities:  

Keywords:  MCT3; age-related macular degeneration; induced pluripotent stem cells; lactate transport; retinal pigment epithelium; splicing

Year:  2021        PMID: 33477551      PMCID: PMC7831140          DOI: 10.3390/cells10010179

Source DB:  PubMed          Journal:  Cells        ISSN: 2073-4409            Impact factor:   6.600


  39 in total

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Journal:  Cell       Date:  2015-05-07       Impact factor: 41.582

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5.  Defined Xeno-free and Feeder-free Culture Conditions for the Generation of Human iPSC-derived Retinal Cell Models.

Authors:  Amélie Slembrouck-Brec; Céline Nanteau; José-Alain Sahel; Olivier Goureau; Sacha Reichman
Journal:  J Vis Exp       Date:  2018-09-06       Impact factor: 1.355

6.  Complement factor H genetic variant and age-related macular degeneration: effect size, modifiers and relationship to disease subtype.

Authors:  Reecha Sofat; Juan P Casas; Andrew R Webster; Alan C Bird; Samantha S Mann; John R W Yates; Anthony T Moore; Tiina Sepp; Valentina Cipriani; Catey Bunce; Jane C Khan; Humma Shahid; Anand Swaroop; Gonçalo Abecasis; Kari E H Branham; Sepideh Zareparsi; Arthur A Bergen; Caroline C W Klaver; Dominique C Baas; Kang Zhang; Yuhong Chen; Daniel Gibbs; Bernhard H F Weber; Claudia N Keilhauer; Lars G Fritsche; Andrew Lotery; Angela J Cree; Helen L Griffiths; Shomi S Bhattacharya; Li L Chen; Sharon A Jenkins; Tunde Peto; Mark Lathrop; Thierry Leveillard; Michael B Gorin; Daniel E Weeks; Maria Carolina Ortube; Robert E Ferrell; Johanna Jakobsdottir; Yvette P Conley; Mati Rahu; Johan H Seland; Gisele Soubrane; Fotis Topouzis; Jesus Vioque; Laura Tomazzoli; Ian Young; John Whittaker; Usha Chakravarthy; Paulus T V M de Jong; Liam Smeeth; Astrid Fletcher; Aroon D Hingorani
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7.  Human iPSC-Derived Retinal Pigment Epithelium: A Model System for Prioritizing and Functionally Characterizing Causal Variants at AMD Risk Loci.

Authors:  Erin N Smith; Agnieszka D'Antonio-Chronowska; William W Greenwald; Victor Borja; Lana R Aguiar; Robert Pogue; Hiroko Matsui; Paola Benaglio; Shyamanga Borooah; Matteo D'Antonio; Radha Ayyagari; Kelly A Frazer
Journal:  Stem Cell Reports       Date:  2019-05-09       Impact factor: 7.765

Review 8.  Functional Genomics of the Retina to Elucidate its Construction and Deconstruction.

Authors:  Frédéric Blond; Thierry Léveillard
Journal:  Int J Mol Sci       Date:  2019-10-04       Impact factor: 5.923

9.  Seven new loci associated with age-related macular degeneration.

Authors:  Lars G Fritsche; Wei Chen; Matthew Schu; Brian L Yaspan; Yi Yu; Gudmar Thorleifsson; Donald J Zack; Satoshi Arakawa; Valentina Cipriani; Stephan Ripke; Robert P Igo; Gabriëlle H S Buitendijk; Xueling Sim; Daniel E Weeks; Robyn H Guymer; Joanna E Merriam; Peter J Francis; Gregory Hannum; Anita Agarwal; Ana Maria Armbrecht; Isabelle Audo; Tin Aung; Gaetano R Barile; Mustapha Benchaboune; Alan C Bird; Paul N Bishop; Kari E Branham; Matthew Brooks; Alexander J Brucker; William H Cade; Melinda S Cain; Peter A Campochiaro; Chi-Chao Chan; Ching-Yu Cheng; Emily Y Chew; Kimberly A Chin; Itay Chowers; David G Clayton; Radu Cojocaru; Yvette P Conley; Belinda K Cornes; Mark J Daly; Baljean Dhillon; Albert O Edwards; Evangelos Evangelou; Jesen Fagerness; Henry A Ferreyra; James S Friedman; Asbjorg Geirsdottir; Ronnie J George; Christian Gieger; Neel Gupta; Stephanie A Hagstrom; Simon P Harding; Christos Haritoglou; John R Heckenlively; Frank G Holz; Guy Hughes; John P A Ioannidis; Tatsuro Ishibashi; Peronne Joseph; Gyungah Jun; Yoichiro Kamatani; Nicholas Katsanis; Claudia N Keilhauer; Jane C Khan; Ivana K Kim; Yutaka Kiyohara; Barbara E K Klein; Ronald Klein; Jaclyn L Kovach; Igor Kozak; Clara J Lee; Kristine E Lee; Peter Lichtner; Andrew J Lotery; Thomas Meitinger; Paul Mitchell; Saddek Mohand-Saïd; Anthony T Moore; Denise J Morgan; Margaux A Morrison; Chelsea E Myers; Adam C Naj; Yusuke Nakamura; Yukinori Okada; Anton Orlin; M Carolina Ortube; Mohammad I Othman; Chris Pappas; Kyu Hyung Park; Gayle J T Pauer; Neal S Peachey; Olivier Poch; Rinki Ratna Priya; Robyn Reynolds; Andrea J Richardson; Raymond Ripp; Guenther Rudolph; Euijung Ryu; José-Alain Sahel; Debra A Schaumberg; Hendrik P N Scholl; Stephen G Schwartz; William K Scott; Humma Shahid; Haraldur Sigurdsson; Giuliana Silvestri; Theru A Sivakumaran; R Theodore Smith; Lucia Sobrin; Eric H Souied; Dwight E Stambolian; Hreinn Stefansson; Gwen M Sturgill-Short; Atsushi Takahashi; Nirubol Tosakulwong; Barbara J Truitt; Evangelia E Tsironi; André G Uitterlinden; Cornelia M van Duijn; Lingam Vijaya; Johannes R Vingerling; Eranga N Vithana; Andrew R Webster; H-Erich Wichmann; Thomas W Winkler; Tien Y Wong; Alan F Wright; Diana Zelenika; Ming Zhang; Ling Zhao; Kang Zhang; Michael L Klein; Gregory S Hageman; G Mark Lathrop; Kari Stefansson; Rando Allikmets; Paul N Baird; Michael B Gorin; Jie Jin Wang; Caroline C W Klaver; Johanna M Seddon; Margaret A Pericak-Vance; Sudha K Iyengar; John R W Yates; Anand Swaroop; Bernhard H F Weber; Michiaki Kubo; Margaret M Deangelis; Thierry Léveillard; Unnur Thorsteinsdottir; Jonathan L Haines; Lindsay A Farrer; Iris M Heid; Gonçalo R Abecasis
Journal:  Nat Genet       Date:  2013-03-03       Impact factor: 38.330

10.  Evaluating the potential of hyperpolarised [1-13C] L-lactate as a neuroprotectant metabolic biosensor for stroke.

Authors:  Jean-Noël Hyacinthe; Lara Buscemi; Thanh Phong Lê; Mario Lepore; Lorenz Hirt; Mor Mishkovsky
Journal:  Sci Rep       Date:  2020-03-26       Impact factor: 4.379

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  3 in total

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2.  The Emergence of Rod-Cone Cellular Interaction.

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3.  Transcriptome Sequencing Analysis of the Effect of β-Elemene on Colorectal Cancer from the lncRNA-miRNA-mRNA Perspective.

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