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Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab

Classic antiglomerular basement membrane (anti-GBM) disease is an exceedingly rare but extremely aggressive form of glomerulonephritis, typically caused by autoantibodies directed against cryptic, conformational epitopes within the noncollagenous domain of the type IV collagen alpha-3 subunit. Patho...

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Autores principales: Jen, Kuang-Yu, Auron, Ari
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8313325/
https://www.ncbi.nlm.nih.gov/pubmed/34336318
http://dx.doi.org/10.1155/2021/2586693
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author Jen, Kuang-Yu
Auron, Ari
author_facet Jen, Kuang-Yu
Auron, Ari
author_sort Jen, Kuang-Yu
collection PubMed
description Classic antiglomerular basement membrane (anti-GBM) disease is an exceedingly rare but extremely aggressive form of glomerulonephritis, typically caused by autoantibodies directed against cryptic, conformational epitopes within the noncollagenous domain of the type IV collagen alpha-3 subunit. Pathologic diagnosis is established by the presence of strong, diffuse, linear staining for immunoglobulin on immunofluorescence microscopy. Recently, patients with atypical clinical and pathologic findings of anti-GBM disease have been described. These patients tend to have an indolent clinical course, without pulmonary involvement, and laboratory testing rarely reveals the presence of anti-GBM antibodies. Specific guidelines for the treatment and management of these patients are unclear. Here, we describe a case of atypical anti-GBM disease in a young child who presented with hematuria and prominent proteinuria. Throughout the course of his illness, creatinine remained normal. He was conservatively treated with steroids and rituximab, resulting in resolution of his clinical symptoms and normalization of laboratory findings.
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spelling pubmed-83133252021-07-31 Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab Jen, Kuang-Yu Auron, Ari Case Rep Nephrol Case Report Classic antiglomerular basement membrane (anti-GBM) disease is an exceedingly rare but extremely aggressive form of glomerulonephritis, typically caused by autoantibodies directed against cryptic, conformational epitopes within the noncollagenous domain of the type IV collagen alpha-3 subunit. Pathologic diagnosis is established by the presence of strong, diffuse, linear staining for immunoglobulin on immunofluorescence microscopy. Recently, patients with atypical clinical and pathologic findings of anti-GBM disease have been described. These patients tend to have an indolent clinical course, without pulmonary involvement, and laboratory testing rarely reveals the presence of anti-GBM antibodies. Specific guidelines for the treatment and management of these patients are unclear. Here, we describe a case of atypical anti-GBM disease in a young child who presented with hematuria and prominent proteinuria. Throughout the course of his illness, creatinine remained normal. He was conservatively treated with steroids and rituximab, resulting in resolution of his clinical symptoms and normalization of laboratory findings. Hindawi 2021-07-17 /pmc/articles/PMC8313325/ /pubmed/34336318 http://dx.doi.org/10.1155/2021/2586693 Text en Copyright © 2021 Kuang-Yu Jen and Ari Auron. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Jen, Kuang-Yu
Auron, Ari
Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab
title Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab
title_full Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab
title_fullStr Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab
title_full_unstemmed Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab
title_short Atypical Antiglomerular Basement Membrane Disease in a Pediatric Patient Successfully Treated with Rituximab
title_sort atypical antiglomerular basement membrane disease in a pediatric patient successfully treated with rituximab
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8313325/
https://www.ncbi.nlm.nih.gov/pubmed/34336318
http://dx.doi.org/10.1155/2021/2586693
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