Clinical characteristics and outcomes of childhood-onset ANCA-associated vasculitis: a French nationwide study Article Swipe
Anne‐Sylvia Sacri
,
T. Chambaraud
,
Bruno Ranchin
,
Benoît Florkin
,
H. See
,
Stéphane Decramer
,
Hugues Flodrops
,
Tim Ulinski
,
Emma Allain‐Launay
,
Olivia Boyer
,
Olivier Dunand
,
Michel Fischbach
,
É. Hachulla
,
Christine Piétrement
,
P. Le Pogamp
,
JL Stéphan
,
Alexandre Bélot
,
Hubert Nivet
,
François Nobili
,
Loı̈c Guillevin
,
Pierre Quartier
,
Georges Deschênes
,
Rémi Salomon
,
Marie Essig
,
J. Harambat
·
YOU?
·
· 2015
· Open Access
·
· DOI: https://doi.org/10.1093/ndt/gfv011
YOU?
·
· 2015
· Open Access
·
· DOI: https://doi.org/10.1093/ndt/gfv011
Background Data on anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis are scarce in children. The current study is aimed at describing the clinical features and outcomes of childhood-onset ANCA-associated vasculitis (AAV). Methods We conducted a retrospective French multicentre study involving patients in whom AAV was diagnosed before the age of 18 years. Inclusion criteria were (i) granulomatosis with polyangiitis (GPA) or microscopic polyangiitis (MPA) according to classification criteria of the European League Against Rheumatism/Paediatric Rheumatology European Society, and (ii) ANCA positivity. Patient and renal survival were analysed. Results Among 66 children included, 80% were female, 42% had GPA and 58% MPA including renal-limited vasculitis, 67% were pANCA+ and 33% cANCA+. The mean incidence of reported cases increased to 0.45 per million children/year in the period 2006–10. Median age at diagnosis was 11.5 years, and median time to diagnosis was 1 month. Initial symptoms included fever and fatigue (79%), skin lesions (41%), arthritis (42%), pulmonary (45%) and renal involvement (88%). Clinical features were similar between GPA and MPA with the exception of upper airway impairment (28%) specific of GPA. Ninety percent of the patients achieved remission after induction treatment. After a median follow-up of 5.2 years, 4 patients (6%) died, corresponding to a mortality rate of 1.2 per 100 person-years, and 22 patients (34%) developed end-stage renal disease (ESRD). Renal survival was 74, 70 and 59% at 1, 5 and 10 years, respectively. In a multivariable Cox regression model, baseline glomerular filtration rate, ethnic origin, histopathological classification and era of treatment were associated with the occurrence of ESRD. Relapse-free survival was 57% at 5 years and 34% at 10 years of follow-up. Patient and renal outcome did not significantly differ between GPA and MPA. Conclusion Childhood-onset AAV is a rare disease characterized by female predominance, delayed diagnosis, frequent renal impairment and a high remission rate. Baseline GFR and new histopathological classification system are strong predictors of ESRD. Renal survival in childhood AAV has improved over time.
Related Topics
Concepts
Medicine
Microscopic polyangiitis
Internal medicine
Vasculitis
Granulomatosis with polyangiitis
Rheumatology
Anti-neutrophil cytoplasmic antibody
Incidence (geometry)
Pediatrics
Surgery
Disease
Physics
Optics
Metadata
- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.1093/ndt/gfv011
- https://academic.oup.com/ndt/article-pdf/30/suppl_1/i104/6838943/gfv011.pdf
- OA Status
- bronze
- Cited By
- 103
- References
- 59
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W2000378313
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
-
https://openalex.org/W2000378313Canonical identifier for this work in OpenAlex
- DOI
-
https://doi.org/10.1093/ndt/gfv011Digital Object Identifier
- Title
-
Clinical characteristics and outcomes of childhood-onset ANCA-associated vasculitis: a French nationwide studyWork title
- Type
-
articleOpenAlex work type
- Language
-
enPrimary language
- Publication year
-
2015Year of publication
- Publication date
-
2015-02-12Full publication date if available
- Authors
-
Anne‐Sylvia Sacri, T. Chambaraud, Bruno Ranchin, Benoît Florkin, H. See, Stéphane Decramer, Hugues Flodrops, Tim Ulinski, Emma Allain‐Launay, Olivia Boyer, Olivier Dunand, Michel Fischbach, É. Hachulla, Christine Piétrement, P. Le Pogamp, JL Stéphan, Alexandre Bélot, Hubert Nivet, François Nobili, Loı̈c Guillevin, Pierre Quartier, Georges Deschênes, Rémi Salomon, Marie Essig, J. HarambatList of authors in order
- Landing page
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https://doi.org/10.1093/ndt/gfv011Publisher landing page
- PDF URL
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https://academic.oup.com/ndt/article-pdf/30/suppl_1/i104/6838943/gfv011.pdfDirect link to full text PDF
- Open access
-
YesWhether a free full text is available
- OA status
-
bronzeOpen access status per OpenAlex
- OA URL
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https://academic.oup.com/ndt/article-pdf/30/suppl_1/i104/6838943/gfv011.pdfDirect OA link when available
- Concepts
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Medicine, Microscopic polyangiitis, Internal medicine, Vasculitis, Granulomatosis with polyangiitis, Rheumatology, Anti-neutrophil cytoplasmic antibody, Incidence (geometry), Pediatrics, Surgery, Disease, Physics, OpticsTop concepts (fields/topics) attached by OpenAlex
- Cited by
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103Total citation count in OpenAlex
- Citations by year (recent)
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2025: 7, 2024: 9, 2023: 13, 2022: 16, 2021: 14Per-year citation counts (last 5 years)
- References (count)
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59Number of works referenced by this work
- Related works (count)
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10Other works algorithmically related by OpenAlex
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| publication_date | 2015-02-12 |
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| abstract_inverted_index.1 | 138 |
| abstract_inverted_index.4 | 194 |
| abstract_inverted_index.5 | 226, 261 |
| abstract_inverted_index.a | 33, 188, 200, 232, 286, 299 |
| abstract_inverted_index.1, | 225 |
| abstract_inverted_index.10 | 228, 266 |
| abstract_inverted_index.18 | 49 |
| abstract_inverted_index.22 | 209 |
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| abstract_inverted_index.70 | 221 |
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| abstract_inverted_index.We | 31 |
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| abstract_inverted_index.42% | 94 |
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| abstract_inverted_index.57% | 259 |
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| abstract_inverted_index.MPA | 99, 165 |
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| abstract_inverted_index.not | 275 |
| abstract_inverted_index.per | 118, 205 |
| abstract_inverted_index.the | 20, 46, 68, 122, 167, 180, 252 |
| abstract_inverted_index.was | 43, 129, 137, 219, 258 |
| abstract_inverted_index.(6%) | 196 |
| abstract_inverted_index.(ii) | 77 |
| abstract_inverted_index.0.45 | 117 |
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| abstract_inverted_index.ANCA | 78 |
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| abstract_inverted_index.MPA. | 281 |
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| abstract_inverted_index.(34%) | 211 |
| abstract_inverted_index.(45%) | 153 |
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