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Proteinuria precedes podocyte abnormalities inLamb2–/– mice, implicating the glomerular basement membrane as an albumin barrier
George Jarad, … , Andrey S. Shaw, Jeffrey H. Miner
George Jarad, … , Andrey S. Shaw, Jeffrey H. Miner
Published August 1, 2006
Citation Information: J Clin Invest. 2006;116(8):2272-2279. https://doi.org/10.1172/JCI28414.
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Research Article Nephrology

Proteinuria precedes podocyte abnormalities inLamb2–/– mice, implicating the glomerular basement membrane as an albumin barrier

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Abstract

Primary defects in either podocytes or the glomerular basement membrane (GBM) cause proteinuria, a fact that complicates defining the barrier to albumin. Laminin β2 (LAMB2) is a GBM component required for proper functioning of the glomerular filtration barrier. To investigate the GBM’s role in glomerular filtration, we characterized GBM and overlying podocyte architecture in relation to development and progression of proteinuria in Lamb2–/– mice, which model Pierson syndrome, a rare congenital nephrotic syndrome. We found ectopic deposition of several laminins and mislocalization of anionic sites in the GBM, which together suggest that the Lamb2–/– GBM is severely disorganized, although it is ultrastructurally intact. Importantly, albuminuria was detectable shortly after birth and preceded podocyte foot process effacement and loss of slit diaphragms by at least 7 days. Expression and localization of slit diaphragm and foot process–associated proteins appeared normal at early stages. GBM permeability to the electron-dense tracer ferritin was dramatically elevated in Lamb2–/– mice, even before widespread foot process effacement. Increased ferritin permeability was not observed in nephrotic CD2-associated protein–null (Cd2ap–/–) mice, which have a primary podocyte defect. Together these data show that the GBM serves as a barrier to protein in vivo and that the glomerular slit diaphragm alone is not sufficient to prevent the passage of albumin into the urinary space.

Authors

George Jarad, Jeanette Cunningham, Andrey S. Shaw, Jeffrey H. Miner

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Figure 5

Increased GBM permeability to ferritin inLamb2–/– mice.

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                  Increased GBM permeability to ferritin inLamb2–/–
   ...
Ferritin was detected in the GBM either 1 (A) or 2 (B) hour after a single intravenous injection into control/mutant littermate pairs, at the indicated ages. Ferritin particles were counted in the total surface area of the GBM (expressed as number of ferritin particles/10,000 nm2) or only at the subepithelial (podocyte) aspect (expressed as number of ferritin particles/μm of GBM length). There was an increase in total and in subepithelial ferritin at 1 hour in all Lamb2–/– mice compared with control or non-nephrotic, rescued Lamb2–/– mice carrying the NEPH-B2 transgene (Res). The increase was more remarkable after 2 hours in the older mice. (C–F) Representative electron micrographs showing ferritin particles in the GBM of normal (C and D) and Lamb2–/– (E and F) mice at P11. Note the increased ferritin in the mutant GBM despite the normal FP architecture. D and F are higher magnifications of C and E, respectively. Scale bars: 125 nm.

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