Molecular basis of ocular abnormalities associated with proximal renal tubular acidosis

J Clin Invest. 2001 Jul;108(1):107-15. doi: 10.1172/JCI11869.

Abstract

Proximal renal tubular acidosis associated with ocular abnormalities such as band keratopathy, glaucoma, and cataracts is caused by mutations in the Na(+)-HCO(3)(-) cotransporter (NBC-1). However, the mechanism by which NBC-1 inactivation leads to such ocular abnormalities remains to be elucidated. By immunological analysis of human and rat eyes, we demonstrate that both kidney type (kNBC-1) and pancreatic type (pNBC-1) transporters are present in the corneal endothelium, trabecular meshwork, ciliary epithelium, and lens epithelium. In the human lens epithelial (HLE) cells, RT-PCR detected mRNAs of both kNBC-1 and pNBC-1. Although a Na(+)-HCO(3)-cotransport activity has not been detected in mammalian lens epithelia, cell pH (pH(i)) measurements revealed the presence of Cl(-)-independent, electrogenic Na(+)-HCO(3)-cotransport activity in HLE cells. In addition, up to 80% of amiloride-insensitive pH(i) recovery from acid load in the presence of HCO(3)(-)/CO(2) was inhibited by adenovirus-mediated transfer of a specific hammerhead ribozyme against NBC-1, consistent with a major role of NBC-1 in overall HCO(3)-transport by the lens epithelium. These results indicate that the normal transport activity of NBC-1 is indispensable not only for the maintenance of corneal and lenticular transparency but also for the regulation of aqueous humor outflow.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • 4,4'-Diisothiocyanostilbene-2,2'-Disulfonic Acid / pharmacology
  • Acidosis, Renal Tubular / complications*
  • Acidosis, Renal Tubular / genetics
  • Amiloride / pharmacology
  • Animals
  • Bicarbonates / metabolism*
  • Blotting, Western
  • Carrier Proteins / genetics*
  • Carrier Proteins / metabolism
  • Cataract / etiology*
  • Cataract / genetics
  • Cells, Cultured
  • Chlorides / metabolism
  • Cornea / metabolism*
  • Cornea / pathology
  • Corneal Opacity / etiology*
  • Corneal Opacity / genetics
  • Epithelial Cells / drug effects
  • Epithelial Cells / metabolism
  • Eye Proteins / genetics*
  • Eye Proteins / metabolism
  • Glaucoma / etiology*
  • Glaucoma / genetics
  • Humans
  • Ion Transport / genetics
  • Kidney Tubules, Proximal / metabolism
  • Lens, Crystalline / metabolism*
  • Lens, Crystalline / pathology
  • Pancreas / metabolism
  • Protein Isoforms / deficiency
  • Protein Isoforms / genetics*
  • Protein Isoforms / metabolism
  • RNA, Catalytic / chemistry
  • RNA, Catalytic / pharmacology
  • RNA, Messenger / biosynthesis
  • Rats
  • Reverse Transcriptase Polymerase Chain Reaction
  • Sodium / metabolism*
  • Sodium-Bicarbonate Symporters
  • Valinomycin / pharmacology

Substances

  • Bicarbonates
  • Carrier Proteins
  • Chlorides
  • Eye Proteins
  • Protein Isoforms
  • RNA, Catalytic
  • RNA, Messenger
  • Sodium-Bicarbonate Symporters
  • hammerhead ribozyme RZ-NBC
  • Valinomycin
  • Amiloride
  • Sodium
  • 4,4'-Diisothiocyanostilbene-2,2'-Disulfonic Acid