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Dysfunctional LAT2 Amino Acid Transporter Is Associated With Cataract in Mouse and Humans.
Knöpfel, Emilia Boiadjieva; Vilches, Clara; Camargo, Simone M R; Errasti-Murugarren, Ekaitz; Stäubli, Andrina; Mayayo, Clara; Munier, Francis L; Miroshnikova, Nataliya; Poncet, Nadège; Junza, Alexandra; Bhattacharya, Shomi S; Prat, Esther; Berry, Vanita; Berger, Wolfgang; Heon, Elise; Moore, Anthony T; Yanes, Óscar; Nunes, Virginia; Palacín, Manuel; Verrey, Francois; Kloeckener-Gruissem, Barbara.
Afiliação
  • Knöpfel EB; Institute of Physiology, University of Zurich, Zurich, Switzerland.
  • Vilches C; Zurich Center for Integrative Human Physiology, University of Zurich, Zurich, Switzerland.
  • Camargo SMR; Swiss National Centre of Competence in Research Kidney.CH, University of Zurich, Zurich, Switzerland.
  • Errasti-Murugarren E; Genes, Disease and Therapy Program, Molecular Genetics Laboratory - IDIBELL, Barcelona, Spain.
  • Stäubli A; U730 and U731, Centro de Investigación Biomédica en Red de Enfermedades Raras, Barcelona, Spain.
  • Mayayo C; Institute of Physiology, University of Zurich, Zurich, Switzerland.
  • Munier FL; Zurich Center for Integrative Human Physiology, University of Zurich, Zurich, Switzerland.
  • Miroshnikova N; U730 and U731, Centro de Investigación Biomédica en Red de Enfermedades Raras, Barcelona, Spain.
  • Poncet N; Institute for Research in Biomedicine, The Barcelona Institute of Science and Technology, Barcelona, Spain.
  • Junza A; Institute of Medical Molecular Genetics, University of Zurich, Zurich, Switzerland.
  • Bhattacharya SS; Department of Biology, ETH Zurich, Zurich, Switzerland.
  • Prat E; Genes, Disease and Therapy Program, Molecular Genetics Laboratory - IDIBELL, Barcelona, Spain.
  • Berry V; Institute for Research in Biomedicine, The Barcelona Institute of Science and Technology, Barcelona, Spain.
  • Berger W; Jules-Gonin Eye Hospital, University of Lausanne, Lausanne, Switzerland.
  • Heon E; Institute of Medical Molecular Genetics, University of Zurich, Zurich, Switzerland.
  • Moore AT; Institute of Physiology, University of Zurich, Zurich, Switzerland.
  • Yanes Ó; Zurich Center for Integrative Human Physiology, University of Zurich, Zurich, Switzerland.
  • Nunes V; Metabolomics Platform, IISPV, Department of Electronic Engineering, Universitat Rovira i Virgili, Tarragona, Spain.
  • Palacín M; CIBER of Diabetes and Associated Metabolic Diseases (CIBERDEM), Madrid, Spain.
  • Verrey F; Andalusian Molecular Biology and Regenerative Medicine Centre - CABIMER, Seville, Spain.
  • Kloeckener-Gruissem B; UCL Institute of Ophthalmology, London, United Kingdom.
Front Physiol ; 10: 688, 2019.
Article em En | MEDLINE | ID: mdl-31231240
Cataract, the loss of ocular lens transparency, accounts for ∼50% of worldwide blindness and has been associated with water and solute transport dysfunction across lens cellular barriers. We show that neutral amino acid antiporter LAT2 (Slc7a8) and uniporter TAT1 (Slc16a10) are expressed on mouse ciliary epithelium and LAT2 also in lens epithelium. Correspondingly, deletion of LAT2 induced a dramatic decrease in lens essential amino acid levels that was modulated by TAT1 defect. Interestingly, the absence of LAT2 led to increased incidence of cataract in mice, in particular in older females, and a synergistic effect was observed with simultaneous lack of TAT1. Screening SLC7A8 in patients diagnosed with congenital or age-related cataract yielded one homozygous single nucleotide deletion segregating in a family with congenital cataract. Expressed in HeLa cells, this LAT2 mutation did not support amino acid uptake. Heterozygous LAT2 variants were also found in patients with cataract some of which showed a reduced transport function when expressed in HeLa cells. Whether heterozygous LAT2 variants may contribute to the pathology of cataract needs to be further investigated. Overall, our results suggest that defects of amino acid transporter LAT2 are implicated in cataract formation, a situation that may be aggravated by TAT1 defects.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Tipo de estudo: Risk_factors_studies Idioma: En Revista: Front Physiol Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Suíça

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Tipo de estudo: Risk_factors_studies Idioma: En Revista: Front Physiol Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Suíça