Favourable renal survival in paediatric microscopic polyangiitis: efficacy of a novel treatment algorithm

Basu, Biswanath; Mahapatra, Tks; Mondal, Nirmal

doi:10.1093/ndt/gfv016

Cited by 9 publications

(12 citation statements)

References 18 publications

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“…We analysed data from eight retrospective cohorts [ 11 , 12 , 25 – 30 ] for a total of 130 patients (86/109 patients were female [79 %]). The main organ/system involvement are summarised in Table 2 and the main characteristics of studies included in Table 3 .…”

Section: Microscopic Polyangiitismentioning

confidence: 99%

“…Information about immunofluorescence findings for ANCA positivity was available in five studies [ 11 , 25 , 28 – 30 ]. Positive results were obtained for c-ANCA in 4 % of patients (95 % CI 1–11, I 2 = 50 %), and for p-ANCA in 94 % of patients (95 % CI 86–99, I 2 = 35 %).…”

Section: Microscopic Polyangiitismentioning

confidence: 99%

“…Six studies reported the drugs used to induce remission [ 11 , 25 , 27 – 30 ]. In five of these studies, oral or intravenous cyclophosphamide was used with corticosteroids (62 %, 95 % CI 20–96, I 2 = 90 %).…”

Section: Microscopic Polyangiitismentioning

confidence: 99%

“…In five of these studies, oral or intravenous cyclophosphamide was used with corticosteroids (62 %, 95 % CI 20–96, I 2 = 90 %). In one study [ 30 ], only rituximab was used. If we excluded this study from the analysis, the prevalence of cyclophosphamide use increased to 77 % (95 % CI 45–99, I 2 82 %).…”

Section: Microscopic Polyangiitismentioning

confidence: 99%

“…One study stated that no cases of impaired fertility or cancer occurred during follow-up [ 29 ]. In another, only infections were described [ 30 ].…”

Section: Microscopic Polyangiitismentioning

confidence: 99%

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Childhood-onset granulomatosis with polyangiitis and microscopic polyangiitis: systematic review and meta-analysis

Iudici

Quartier

Terrier

et al. 2016

Orphanet J Rare Dis

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BackgroundThe data from cohorts of childhood-onset granulomatosis with polyangiitis (GPA) and microscopic polyangiitis (MPA) remain scarce and heterogeneous. We aimed to analyse the features at presentation, therapeutic approaches and the disease course of these rare diseases.MethodsElectronic searches of Medline and the Cochrane Central Register of Controlled trials database were conducted. We also checked the reference lists of the studies included and other systematic reviews, to identify additional reports. We included all cohorts, cross-sectional studies or registries reporting features at presentation or outcomes in patients with a diagnosis of childhood-onset GPA or MPA (age <18 years). The pooled prevalence of clinical manifestations at presentation, ANCA and induction therapies for GPA and MPA was calculated.ResultsWe reviewed 570 full texts and identified 14 studies on GPA and 8 on MPA. Childhood-onset GPA and MPA occurred predominantly in female subjects during adolescence. For GPA, ear-nose-throat (ENT) disease (pooled prevalence 82 % [95 % CI 78–87]), constitutional symptoms (73 % [95 % CI 55–88]), renal (65 % [95 % CI 49–79]), and lower respiratory tract (61 % [95 % CI 48–74]) manifestations were the most frequently reported at presentation. Renal disease was a hallmark of MPA (94 % [95 % CI 89–97]). ANCA were detected in >90 % of children with GPA or MPA. Combined corticosteroids and cyclophosphamide was the most frequently used first remission-inducing treatment for GPA (76 % [95 % CI 69–82]) and MPA (62 % [95 % CI 20–96]). Relapses occurred more frequently in GPA (67–100 %) than in MPA (25–50 %). The leading causes of death were the disease itself, and infections.ConclusionsChildhood-onset MPA and GPA remain severe diseases with frequent relapses and a high cumulative morbidity. Survival and disease-free survival need to be improved.Electronic supplementary materialThe online version of this article (doi:10.1186/s13023-016-0523-y) contains supplementary material, which is available to authorized users.

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Section: Microscopic Polyangiitismentioning

confidence: 99%

Section: Microscopic Polyangiitismentioning

confidence: 99%

Section: Microscopic Polyangiitismentioning

confidence: 99%

Section: Microscopic Polyangiitismentioning

confidence: 99%

“…One study stated that no cases of impaired fertility or cancer occurred during follow-up [ 29 ]. In another, only infections were described [ 30 ].…”

Section: Microscopic Polyangiitismentioning

confidence: 99%

See 3 more Smart Citations

Childhood-onset granulomatosis with polyangiitis and microscopic polyangiitis: systematic review and meta-analysis

Iudici

Quartier

Terrier

et al. 2016

Orphanet J Rare Dis

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Incidence of Pneumocystis Jiroveci Pneumonia in Patients With ANCA‐Associated Vasculitis Initiating Therapy With Rituximab or Cyclophosphamide

Nettleton,

Sattui,

Wallace

et al. 2023

Arthritis Care & Research

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ObjectiveThis manuscript assesses the incidence of Pneumocystis jiroveci pneumonia (PJP) among patients receiving contemporary treatment regimens for antineutrophil cytoplasmic antibody–associated vasculitis (AAV) and adverse events associated with PJP prophylaxis.MethodsIncident users of rituximab or cyclophosphamide for AAV were identified in the TriNetX electronic health records database from 2011 to 2022. The incidence rates (IRs) of PJP in the first 6 months of induction therapy with rituximab and/or cyclophosphamide and during postinduction maintenance therapy with rituximab were calculated. Cox proportional hazard models were used to estimate hazard ratios (HRs) and confidence intervals (CIs) for the risk of adverse events commonly associated with PJP prophylaxis.ResultsWe identified 1,461 AAV cases who received induction therapy with rituximab (69.7%), cyclophosphamide (18.9%), or both (11.4%). Prophylaxis prescribed within 30 days of induction included trimethoprim‐sulfamethoxazole (30.7%), atovaquone (5.4%), dapsone (3.8%), and pentamidine (0.8%). During induction therapy, 10 cases of PJP were identified (IR 15.0 cases per 1,000 patient‐years); no deaths occurred. In adjusted analyses, those who received prophylaxis had a higher risk of leukopenia (HR 3.1; 95% CI 1.1–8.6), rash (HR 1.9; 95% CI 1.0–3.6), and nephropathy (HR 2.6; 95% CI 1.3–5.1) than those who did not. During rituximab maintenance therapy (n = 709), five cases of PJP were identified (IR 2.1 cases per 1,000 person‐years), one of whom died during the hospitalization associated with a PJP diagnosis.ConclusionRates of PJP in patients with AAV were lower than previously observed, and few cases occurred during rituximab maintenance therapy. PJP prophylaxis was associated with adverse events.

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Clinical presentation and outcome of pediatric ANCA-associated glomerulonephritis

Kouri

Andreoli

2016

Pediatr Nephrol

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Favourable renal survival in paediatric microscopic polyangiitis: efficacy of a novel treatment algorithm

Abstract: Efficacy and medium-term safety of rituximab- and MMF-based protocol in managing children with MPA was evident in this study.

Cited by 9 publications

References 18 publications

Childhood-onset granulomatosis with polyangiitis and microscopic polyangiitis: systematic review and meta-analysis

Childhood-onset granulomatosis with polyangiitis and microscopic polyangiitis: systematic review and meta-analysis

Incidence of Pneumocystis Jiroveci Pneumonia in Patients With ANCA‐Associated Vasculitis Initiating Therapy With Rituximab or Cyclophosphamide

Clinical presentation and outcome of pediatric ANCA-associated glomerulonephritis

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