Recurrence of Goodpasture syndrome without circulating anti-glomerular basement membrane antibodies after kidney transplant, a case report.
Adult
Anti-Glomerular Basement Membrane Disease
/ etiology
Autoantibodies
/ blood
Female
Glomerular Basement Membrane
/ immunology
Humans
Immunosuppressive Agents
/ therapeutic use
Kidney Glomerulus
/ pathology
Kidney Transplantation
Living Donors
Plasma Exchange
Postoperative Complications
/ etiology
Recurrence
Renal Dialysis
Reoperation
Anti-glomerular basement membrane (GBM) disease
End-stage renal disease (ESRD)
Goodpasture syndrome (GS)
Kidney transplant
Journal
BMC nephrology
ISSN: 1471-2369
Titre abrégé: BMC Nephrol
Pays: England
ID NLM: 100967793
Informations de publication
Date de publication:
08 01 2019
08 01 2019
Historique:
received:
21
08
2018
accepted:
26
12
2018
entrez:
10
1
2019
pubmed:
10
1
2019
medline:
15
2
2020
Statut:
epublish
Résumé
Goodpasture Syndrome (GS) is an autoimmune disease caused by the development of auto-antibodies against the Glomerular Basement Membrane (GBM). Linear deposit of immunoglobulins G on the GBM detected by immunofluorescence analysis of renal biopsies is a GS pathognomonic finding. GS is commonly monophasic and its incidence is 1.6 case per million per year. This report describes and discusses the case of a 40-year-old woman who one year after allograft kidney transplant, presented with acute pulmonary and renal symptoms of GS, leading to acute graft dysfunction, without circulating anti-GBM antibody detection in laboratory assays. She received a living donor kidney transplant 4 years after the first diagnosis of GS without circulating anti-GBM antibodies, when considered in remission. In both episodes, the diagnosis of GS was based exclusively on the kidney biopsy that showed rapidly progressing glomerulonephritis with deposition of immunoglobulins G on the GBM. Although rare, the management of patients with GS without circulating anti-GBM antibodies is difficult due to the lack of standardized follow-up guidelines to reduce the risk of GS recurrence after kidney transplantation.
Sections du résumé
BACKGROUND
Goodpasture Syndrome (GS) is an autoimmune disease caused by the development of auto-antibodies against the Glomerular Basement Membrane (GBM). Linear deposit of immunoglobulins G on the GBM detected by immunofluorescence analysis of renal biopsies is a GS pathognomonic finding. GS is commonly monophasic and its incidence is 1.6 case per million per year.
CASE PRESENTATION
This report describes and discusses the case of a 40-year-old woman who one year after allograft kidney transplant, presented with acute pulmonary and renal symptoms of GS, leading to acute graft dysfunction, without circulating anti-GBM antibody detection in laboratory assays. She received a living donor kidney transplant 4 years after the first diagnosis of GS without circulating anti-GBM antibodies, when considered in remission.
CONCLUSIONS
In both episodes, the diagnosis of GS was based exclusively on the kidney biopsy that showed rapidly progressing glomerulonephritis with deposition of immunoglobulins G on the GBM. Although rare, the management of patients with GS without circulating anti-GBM antibodies is difficult due to the lack of standardized follow-up guidelines to reduce the risk of GS recurrence after kidney transplantation.
Identifiants
pubmed: 30621605
doi: 10.1186/s12882-018-1197-6
pii: 10.1186/s12882-018-1197-6
pmc: PMC6323659
doi:
Substances chimiques
Autoantibodies
0
Immunosuppressive Agents
0
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
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