Late recovery of renal function by rituximab in a patient with Wegener’s granulomatosis

Malina, Michal; Schaefer, Betti; Waldherr, Rüdiger; Wühl, Elke; Schaefer, Franz; Schmitt, Claus Peter

doi:10.1007/s00467-012-2332-4

Cited by 4 publications

(4 citation statements)

References 15 publications

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“…Following the first report of the successful treatment of AAV with RTX, different rare clinical manifestations were reported as having been successfully treated with RTX. Some of the most important rare clinical presentations in GPA patients, which resolved following RTX, include retrobulbar granuloma [ 78 ], refractory ophthalmic [ 126 ], pachymeningitis [ 146 ], the presence of orbital inflammation [ 122 ], long-established end-stage renal disease [ 147 ], post-kidney-transplantation glomerulonephritis-related AAV [ 148 ], pituitary GPA [ 149 ], renal mass [ 150 ], gastric ulcer [ 151 ], pancreatitis [ 152 ], ophthalmoplegia [ 153 ], pyoderma gangrenosum [ 154 , 155 , 156 ], prostatitis [ 157 ] aortic inflammation [ 158 ], intestinal involvement [ 159 ], mastitis [ 160 ], aortitis [ 161 ], gingivitis [ 162 ], Isolated Pauci-Immune Pulmonary Capillaritis [ 163 ], isolated orbital GPA [ 164 ], cranial neuropathy [ 165 ], severe necrotizing scleritis [ 166 ], genital necrosis and inflammation [ 167 ], GPA-associated subcutaneous cheek odule [ 168 ], pseudo-tumoral digital nodules [ 169 ], hypertrophic pachymeningitis [ 170 , 171 ], acute myocarditis [ 172 ], palpable purpura [ 173 ], central nervous system vasculitis [ 174 ], intra-cranial hypertension [ 175 , 176 ], tracheobronchial stenosis [ 142 ], acute aortic valve regurgitation [ 177 ], GPA-mimicking meningeal tuberculosis [ 178 ], gynecological involvement [ 179 ], GPA-mimicking lung mal...…”

Section: Resultsmentioning

confidence: 99%

The Efficacy and Safety of Rituximab in ANCA-Associated Vasculitis: A Systematic Review

Habibi

Alesaeidi

Zahedi

et al. 2022

Biology

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Background and aim: Antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) is a rare multisystem autoimmune disease developed by autoantibody production against human neutrophilic granulocytes, including proteinase-3 (PR3) and myeloperoxidase (MPO). The management of AAV patients is difficult due to the multiorgan involvement, high rate of relapse, and complications of immunosuppressive agents that make it challenging. This study aims to investigate the efficacy and safety of rituximab (RTX) therapy in patients with granulomatosis with polyangiitis (GPA) or microscopic polyangiitis (MPA) subtypes. Method: The PubMed/Medline database was searched for any studies related to RTX therapy in ANCA-associated vasculitis (GPA and MPA subtypes), from inception to 1 August 2022, and proceeded in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA). Results: Our search resulted in 1082 initial records. After the elimination of review papers, irrelevant studies, and non-English records, 223 articles were included, and the data related to the efficacy and safety of RTX therapy were extracted. Several randomized and non-randomized studies showed that RTX is an effective treatment option for patients with AAV. Most of the studies showed the very effective effect of RTX in controlling disease in AAV patients, including pediatrics, adults, and elderlies, although RTX cannot completely prevent relapse. However, maintenance therapy helps delay the disease’s relapse and causes sustained remission. Not only the licensed dose (375 mg/m2 intravenous per week for 4 weeks) could induce disease remission, but studies also showed that a single infusion of RTX could be effective. Although RTX could resolve many rare manifestations in AAV patients, there are few reports showing treatment failure. Additionally, few sudies have reported the unexpeted worsening of the disease after RTX administration. Generally, RTX is relatively safe compared to conventional therapies, but some serious adverse effects, mainly infections, cytopenia, hypogammaglobinemia, malignancy, and hypersensitivity have been reported. Conclusions: RTX is an effective and relatively safe therapeutic option for AAV. Studies on the evaluation of the safety profiles of RTX and the prevention of severe RTX-related side effects in AAV patients are required.

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Section: Resultsmentioning

confidence: 99%

The Efficacy and Safety of Rituximab in ANCA-Associated Vasculitis: A Systematic Review

Habibi

Alesaeidi

Zahedi

et al. 2022

Biology

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“…While vascular diseases play a major role in the large series and in registry data, case reports, which are more prone to publication biases, mainly regard the more “charming” glomerular diseases and are mainly targeted at describing exceptional cases, highlighting the possibility of long-term recovery after various treatments, whether started in the pre-dialysis phase or performed during dialysis (29–37, 39–40) (Tab. II).…”

Section: Discussionmentioning

confidence: 99%

“…Table II summarizes the 13 case reports retrieved by our search strategy; they deal with one to two cases, and are reported from different sources (8 from Europe, 2 from Japan, 1 from Canada, and 2 from USA). The distribution of the case reports differs from that of the case series and registry data, highlighting the possibility of renal function recovery in immunologic or hematologic diseases, encompassing all the major glomerulopathies, from the rare ones, like Wegener's disease and atypical HUS, to the common ones, like IgA nephropathy or FSGS (29)(30)(31)(32)(33)(34)(35)39). However, in keeping with the important role of vascular diseases in this context, two cases (cholesterol emboli and renal artery stenosis) are also reported (38,41).…”

Section: Large Seriesmentioning

confidence: 93%

Chronic Dialysis Discontinuation: A Systematic Narrative Review of the Literature in the New Millennium

et al. 2014

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Introduction and aims Renal function recovery (RFR), defined as the discontinuation of dialysis after 3 months of replacement therapy, is an uncommon occurrence. At a time when the “too early” start of dialysis is in discussion, a systematic review of the literature for cases in which patients recovered renal function after starting dialysis with chronic indications, including single cases and large series, may lead to attention being focused on this interesting issue. Methods The search strategy was built in Medline on Pubmed, in EMBASE and in the Cochrane Collaboration (August 2013) combining Mesh, Emtree and free terms: dialysis or hemodialysis, kidney function, renal function and recovery (publication date 2000-2013). The following tasks were performed in duplicate: titles and abstracts were manually screened, the data were extracted: title, author, objective, year, journal, period of study, multi-center, country, type of study. Results The systematic review retrieved 1 894 titles; 58 full papers were retrieved and the final selection included 24 papers: 11 case series or Registry data (4 from ANZdata) and 13 case reports. In spite of the high heterogeneity of the studies, overall they suggest that RFR occurs in about 1% of patients, without differences between PD and HD. RFR appears to be more frequent in elderly patients with renal vascular disease (up to 10% RFR in cholesterol emboli or scleroderma), but is reported in all types of primary and secondary kidney diseases. Conclusions RFR is a clinical event that should be looked for, particularly in elderly patients with vascular comorbidity.

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“…97 Interestingly, RTX led to clinical remission in a dialysis-dependent adolescent with GPA who had been dialysis dependent for 22 months despite the use of CYC. 98 Danish investigators randomized 32 patients with GPA to PLEX or no PLEX as part of initial induction therapy (both groups received CYC þ CS). 99 Multivariate analysis demonstrated that PLEX improved renal survival among patients with initial serum creatinine greater than 250 μmol/L (2.85 mg/dL; p < 0.01).…”

Section: Renal Involvementmentioning

confidence: 99%

Granulomatosis with Polyangiitis (Wegener's Granulomatosis): Evolving Concepts in Treatment

Lynch

Derhovanessian

Tazelaar

et al. 2018

Semin Respir Crit Care Med

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Granulomatosis with polyangiitis (GPA), formerly termed Wegener's granulomatosis, is the most common of the pulmonary vasculitides. GPA typically involves the upper respiratory tract, lower respiratory tract (bronchi and lung), and kidney, with varying degrees of disseminated vasculitis. Cardinal histologic features include a necrotizing vasculitis involving small vessels, extensive “geographic” necrosis, and granulomatous inflammation. The spectrum and severity of the disease is heterogeneous, ranging from indolent disease involving only one site to fulminant, multiorgan vasculitis. Circulating antibodies against cytoplasmic components of neutrophils (ANCAs) play a role in the pathogenesis, and often correlate with activity of the disease. Treatment strategies are evolving. Cyclophosphamide (CYC) plus corticosteroids was the mainstay of therapy for generalized, multisystemic GPA since the 1970s. However, within the past decade, rituximab (RTX), a monoclonal antibody directed against B cells, has been shown to be at least as effective (and possibly more effective) as CYC. Furthermore, the use of RTX may reduce the need for maintenance immunosuppression. Optimal therapy for GPA remains controversial, and additional studies are required to determine the role and duration of maintenance therapy following successful induction therapy.

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Late recovery of renal function by rituximab in a patient with Wegener’s granulomatosis

Abstract: This case documents a potent beneficial effect of rituximab on renal manifestation of GPA even in long-established ESRD.

Cited by 4 publications

References 15 publications

The Efficacy and Safety of Rituximab in ANCA-Associated Vasculitis: A Systematic Review

The Efficacy and Safety of Rituximab in ANCA-Associated Vasculitis: A Systematic Review

Chronic Dialysis Discontinuation: A Systematic Narrative Review of the Literature in the New Millennium

Granulomatosis with Polyangiitis (Wegener's Granulomatosis): Evolving Concepts in Treatment

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