TNFα pathway blockade ameliorates toxic effects of FSGS plasma on podocyte cytoskeleton and β3 integrin activation.

Background: In the absence of mutant genes encoding components of the podocyte slit diaphragm, about 30-50 % of children with primary glucocorticoid-resistant focal segmental glomerulosclerosis (FSGS) develop recurrent proteinuria and slowly progressive FSGS lesions following renal transplantation....

Descripción completa

Detalles Bibliográficos
Publicado en:Pediatric Nephrology Vol. 27; no. 12; pp. 2217 - 2227
Autores principales: Bitzan, Martin, Babayeva, Sima, Vasudevan, Anil, Goodyer, Paul, Torban, Elena
Formato: pictorial research tables/charts Journal Article
Publicado: Springer Nature Dec2012
Acceso en línea:Ver este registro en EBSCOhost
fields @attributes:
  recordID: 1
pdfLink:
plink: https://search.ebscohost.com/login.aspx?direct=true&db=ccm&AN=83184825&site=ehost-live
header:
  @attributes:
    shortDbName: ccm
    uiTerm: 83184825
    longDbName: CINAHL Complete
    uiTag: AN
  controlInfo:
    bkinfo:
    dissinfo:
    jinfo:
      jid:
        0931041X
        EF1
      jtl: Pediatric Nephrology
      issn: 0931041X
      maglogo: N
    pubinfo:
      dt: Dec2012
      vid: 27
      iid: 12
      pid: 237
      pub: Springer Nature
      place: New York, New York
    artinfo:
      ui:
        83184825
        104434499
        104434499
        10.1007/s00467-012-2163-3
        83184825
      ppf: 2217
      ppct: 10
      formats:
        fmt:
          @attributes:
            type: P
      tig:
        atl: TNFα pathway blockade ameliorates toxic effects of FSGS plasma on podocyte cytoskeleton and β3 integrin activation.
      aug:
        au:
          Bitzan, Martin
          Babayeva, Sima
          Vasudevan, Anil
          Goodyer, Paul
          Torban, Elena
        affil: Department of Pediatrics, Montreal Children's Hospital, McGill University, Montreal Canada
      sug:
        subj:
          Tumor Necrosis Factor
          Antigens, Surface
          Glomerulonephritis Pathology
          Epithelial Cells
          Signal Transduction
          Human
          Cell Culture Techniques
          Child
          Female
          Fluorescent Antibody Technique
          T-Tests
          Analysis of Variance
          Descriptive Statistics
          In Vitro Studies
          Funding Source
          Data Analysis Software
          Child: 6-12 years
          Female
      ab: Background: In the absence of mutant genes encoding components of the podocyte slit diaphragm, about 30-50 % of children with primary glucocorticoid-resistant focal segmental glomerulosclerosis (FSGS) develop recurrent proteinuria and slowly progressive FSGS lesions following renal transplantation. Recurrence of FSGS in the allograft strongly suggests a circulating factor that disturbs normal podocyte biology. To date, the nature of the circulating factor is unclear, and there is no cure for the recurrent form of FSGS (R-FSGS). Methods: Cultured differentiated human podocytes were exposed to the plasmapheresis effluent or blood plasma samples from pediatric patients with recurrent or primary FSGS; in some cases, podocytes were pre-incubated with specific antibodies to block the tumor necrosis factor-alpha (TNFα) signaling pathway. Integrity of focal adhesion complexes and actin cytoskeleton were investigated by immunofluorescent microscopy. Results: Plasmapheresis effluent from an R-FSGS child or fresh plasma from two children with primary FSGS rapidly disturbed the cytoskeleton of normal human podocytes in vitro. Plasma from a child with R-FSGS also activated β3 integrin and dispersed focal adhesion complexes. The effects were reversed by pre-incubation with antibodies against TNFα or either of the two TNFα receptors. When our patient with R-FSGS became resistant to plasmapheresis, we initiated treatment with twice weekly etanercept injections and then infliximab. Within 3 weeks of regular anti-TNFα therapy, the patient achieved sustained partial remission of proteinuria, allowing us to wean her off plasmapheresis completely. Conclusions: We suggest that in some FSGS patients, disruption of the podocyte cytoskeleton and β3 integrin-mediated podocyte attachment are driven by the TNFα pathway.
      pubtype: Academic Journal
      doctype:
        pictorial
        research
        tables/charts
        Journal Article
      ougenre: Article
    language: English
    refInfo:
    holdings:
      @attributes:
        islocal: N