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C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?

We describe a 16-year-old Caucasian boy who presented with steroid-sensitive nephrotic syndrome aged 2 years. His clinical course was one of frequent relapses and severe steroid dependence. To manage this, he was sequentially treated with levamisole, then oral cyclophosphamide before being started o...

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Autores principales: Muorah, Mordi, Sinha, Manish D., Horsfield, Catherine, O’Donnell, Patrick J.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2009
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4421234/
https://www.ncbi.nlm.nih.gov/pubmed/25984017
http://dx.doi.org/10.1093/ndtplus/sfp055
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author Muorah, Mordi
Sinha, Manish D.
Horsfield, Catherine
O’Donnell, Patrick J.
author_facet Muorah, Mordi
Sinha, Manish D.
Horsfield, Catherine
O’Donnell, Patrick J.
author_sort Muorah, Mordi
collection PubMed
description We describe a 16-year-old Caucasian boy who presented with steroid-sensitive nephrotic syndrome aged 2 years. His clinical course was one of frequent relapses and severe steroid dependence. To manage this, he was sequentially treated with levamisole, then oral cyclophosphamide before being started on ciclosporin. A renal biopsy performed prior to commencement of ciclosporin confirmed minimal change disease on light microscopy. The immunohistochemistry and electron microscopy findings were in keeping with this. His complement levels were normal and his lupus serology negative. He remained on ciclosporin therapy for 8 years and had two further renal biopsies to detect ciclosporin-induced renal damage. Both biopsies showed evidence of increasing amounts of C1q deposition on immunohistochemistry and the presence of immune deposits on electron microscopy. As he had continued negative lupus serology, this was compatible with a diagnosis of C1q nephropathy. In addition both biopsies had changes compatible with chronic mild ciclosporin nephrotoxicity. This case is the first report describing in detail a paediatric patient with evolving C1q nephropathy who was treated successfully with rituximab. We discuss the role of C1q in this clinicopathological entity and question its significance.
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spelling pubmed-44212342015-05-15 C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon? Muorah, Mordi Sinha, Manish D. Horsfield, Catherine O’Donnell, Patrick J. NDT Plus Case Report We describe a 16-year-old Caucasian boy who presented with steroid-sensitive nephrotic syndrome aged 2 years. His clinical course was one of frequent relapses and severe steroid dependence. To manage this, he was sequentially treated with levamisole, then oral cyclophosphamide before being started on ciclosporin. A renal biopsy performed prior to commencement of ciclosporin confirmed minimal change disease on light microscopy. The immunohistochemistry and electron microscopy findings were in keeping with this. His complement levels were normal and his lupus serology negative. He remained on ciclosporin therapy for 8 years and had two further renal biopsies to detect ciclosporin-induced renal damage. Both biopsies showed evidence of increasing amounts of C1q deposition on immunohistochemistry and the presence of immune deposits on electron microscopy. As he had continued negative lupus serology, this was compatible with a diagnosis of C1q nephropathy. In addition both biopsies had changes compatible with chronic mild ciclosporin nephrotoxicity. This case is the first report describing in detail a paediatric patient with evolving C1q nephropathy who was treated successfully with rituximab. We discuss the role of C1q in this clinicopathological entity and question its significance. Oxford University Press 2009-08 2009-05-09 /pmc/articles/PMC4421234/ /pubmed/25984017 http://dx.doi.org/10.1093/ndtplus/sfp055 Text en © The Author [2009]. Published by Oxford University Press on behalf of ERA-EDTA. All rights reserved. For Permissions, please e-mail: journals.permissions@oxfordjournals.org http://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Case Report
Muorah, Mordi
Sinha, Manish D.
Horsfield, Catherine
O’Donnell, Patrick J.
C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?
title C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?
title_full C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?
title_fullStr C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?
title_full_unstemmed C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?
title_short C1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?
title_sort c1q nephropathy: a true immune complex disease or an immunologic epiphenomenon?
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4421234/
https://www.ncbi.nlm.nih.gov/pubmed/25984017
http://dx.doi.org/10.1093/ndtplus/sfp055
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