SLC4A11 Three-Dimensional Model Explains Structural Basis for Endothelial Corneal Dystrophy-Causing Mutations

نویسندگان

  • Kumari Alka
  • Katherine E. Badior
  • Joseph R. Casey
  • Alice E. Davidson
  • Petra Liskova
  • Cerys J. Evans
  • Lenka Nosková
  • Nikolas Pontikos
  • Vincent Plagnol
  • Martin Filipec
  • Stanislav Kmoch
  • Stephen J. Tuft
  • Alison J. Hardcastle
چکیده

These abstracts are licensed under a Creative Commons Attribution-NonCommercial-No Derivatives 4.0 International License. Go to http://iovs.arvojournals.org/ to access the versions of record. 438 Corneal Genetics and Myopia Wednesday, May 04, 2016 11:00 AM–12:45 PM Exhibit/Poster Hall Poster Session Program #/Board # Range: 4820–4840/A0036–A0056 Organizing Section: Genetics Group Contributing Section(s): Anatomy/Pathology, Clinical/ Epidemiologic Research

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SLC4A11 mutations in Fuchs endothelial corneal dystrophy.

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SLC4A11 and the Pathophysiology of Congenital Hereditary Endothelial Dystrophy

Congenital hereditary endothelial dystrophy (CHED) is a rare autosomal recessive disorder of the corneal endothelium characterized by nonprogressive bilateral corneal edema and opacification present at birth. Here we review the current knowledge on the role of the SLC4A11 gene, protein, and its mutations in the pathophysiology and clinical presentation of CHED. Individuals with CHED have mutati...

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Conditionally Immortal Slc4a11−/− Mouse Corneal Endothelial Cell Line Recapitulates Disrupted Glutaminolysis Seen in Slc4a11−/− Mouse Model

Purpose To establish conditionally immortal mouse corneal endothelial cell lines with genetically matched Slc4a11+/+ and Slc4a11-/- mice as a model for investigating pathology and therapies for SLC4A11 associated congenital hereditary endothelial dystrophy (CHED) and Fuchs' endothelial corneal dystrophy. Methods We intercrossed H-2Kb-tsA58 mice (Immortomouse) expressing an IFN-γ dependent and...

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تاریخ انتشار 2016