Rituximab Therapy for Severe Cutaneous Leukocytoclastic Angiitis Refractory to Corticosteroids, Cellcept and Cyclophosphamide

El-Reshaid, Kamel; Madda, John Patrick

doi:10.1159/000350559

Cited by 7 publications

(4 citation statements)

References 13 publications

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“…Other risk factors associated with relapse of LCV were hepatic cytolysis, positive ANCA serology, and peripheral neuropathy, all of which are associated with systemic vasculitides related to an underlying disease, thus rationally contrasting with SoCSVV, which was the only protective factor for relapse in our study. Even though relapses have been described in SoCSVV, [ 16 , 26 ] our work shows that the prognosis is better in LCV patients who meet the criteria for SoCSVV. As direct immunofluorescence was done in 85% of biopsies, we performed the analysis of factors associated with relapse after exclusion of the 13 patients for whom immunofluorescence was not available.…”

Section: Discussionmentioning

confidence: 71%

Etiologies and prognostic factors of leukocytoclastic vasculitis with skin involvement

Bouiller

Audia

Devilliers³

et al. 2016

Medicine

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In this study, outcomes of patients with leukocytoclastic vasculitis (LCV) were analyzed focusing on clinical, histopathology and laboratory findings, relapses, and survival.Data from patients with cutaneous vasculitis diagnosed between January 1, 2000, and December 31, 2010, at Dijon University Hospital (France) were retrospectively reviewed. LCV was defined as perivascular neutrophilic infiltrate, endothelial cell nuclear swelling, extravasation of red blood cells, and/or fibrin deposition in vessels. Patients were classified according to the 2012 Chapel Hill Consensus Conference. Relapses were defined as the recurrence of vasculitis symptoms after a period of remission >1 month. Time to relapse and/or death was calculated from the date of diagnosis. Univariate and multivariate (Cox model) analyses were performed.A total of 112 patients (57 males and 55 females), with a mean age of 60 ± 19 (18–98) years, were analyzed. Overall follow-up was 61 ± 38 months. At diagnosis, all patients had skin lesions, purpura being the most common (n = 83). Lesions were associated with systemic involvement in 55 (51%) patients. Only 41 (36.6%) patients received specific treatment: glucocorticoids in 29 of 41 (70.7%) and immunosuppressants in 9 of 41 (22%). Sixty-two patients (55%) had LCV due to underlying causes, 29 (25.9%) had single-organ cutaneous small vessel vasculitis (SoCSVV), and 21 (18.8%) had unclassifiable LCV. Twenty patients of the cohort (18%) experienced relapse, 14 ± 13 (1–40) months after the diagnosis of LCV. None of the 29 patients with SoCSVV relapsed. Independent risk factors for relapse were vascular thrombosis in the biopsy [hazard ratio (HR) = 4.9; P = 0.017], peripheral neuropathy (HR = 9.8; P = 0.001), hepatitis (HR = 3.1; P = 0.004), and positive antineutrophil cytoplasm antibodies (ANCA, HR = 5.9 P = 0.005). In contrast, SoCSVV was a protective factor for relapse (HR = 0.12; P = 0.043).The 1-, 3-, and 6-year overall survival rates were 99%, 83%, and 71%, respectively, with no difference between relapsers and nonrelapsers (P = 0.960) or between SoCSVV and unclassifiable LCV (P = 0.588).This study demonstrates that global survival for LCV patients is good but relapses remain frequent, especially when the cutaneous biopsy shows vascular thrombosis, or in patients with peripheral neuropathy or hepatitis. Conversely, SoCSVV is a protective factor for relapse.

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Section: Discussionmentioning

confidence: 71%

Etiologies and prognostic factors of leukocytoclastic vasculitis with skin involvement

Bouiller

Audia

Devilliers³

et al. 2016

Medicine

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“…'nın çalışmalarında ESR en sık rastlanan patolojik laboratuar bulgusu olmasına rağmen, yüksek ESR ile sistemik tutulum arasında bir ilişki bulunamamıştır. [22,24,28] [6,30,31]…”

Section: Discussionunclassified

Demographic, etiological and clinical characteristics of patients with leukocytoclastic vasculitis single center experience

Tezcan

Limon

Gülcemal

et al. 2019

Journal of Contemporary Medicine

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Lökositoklastik vaskülitli hastaların demografik, etyolojik ve klinik özellikleri tek merkez deneyimi Demographic, etiological and clinical characteristics of patients with leukocytoclastic vasculitis single center experience Amaç: Lökositoklastik vaskülit, primer deri tutulumu ile seyreden, esas olarak post kapiller venülleri etkileyen bir küçük damar vaskülit formudur. Etiyolojide ilaçlar, enfeksiyonlar, maligniteler, sistemik inflamatuvar hastalıklar gibi çeşitli nedenler yer almaktadır. Ancak olguların bir kısmında neden saptanamamakta ve bu olgular idiyopatik olarak değerlendirilmektedir. Klinik olarak özellikle alt ekstremitelerde sınırlı palpe edilebilen purpuralar ile karakterize olup, nadiren yaygın tutulum da gözlenebilmektedir. Bu çalışmada, kliniğimizde lökositoklastik vaskülit tanısı almış hastalarda etiyolojik faktörler, demografik ve klinik özelliklerin belirlenmesi amaçlanmıştır.

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“…Rituximab has been used successfully in different types of vasculitis, including urticaria vasculitis, type II mixed cryoglobulinemia, anti-neutrophil cytoplasmic antibody–associated vasculitis, and cutaneous small vessel vasculitis, as well as in several autoimmune dermatoses and cutaneous IgG4-RD …”

Section: Discussionmentioning

confidence: 99%

Assessment of Treatment of Refractory Granuloma Faciale With Intralesional Rituximab

et al. 2018

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IMPORTANCE Granuloma faciale (GF) is a rare, benign inflammatory dermatosis of unknown cause. Some reports have suggested that it could be part of the spectrum of IgG4-related sclerosing diseases. Granuloma faciale is characterized by single or multiple red-brown nodules, most frequently occurring on the face, and it can produce severe disfigurement. Treatment is difficult, and poor outcomes are often seen. Rituximab is a monoclonal antibody against CD-20 approved by the US Food and Drug Administration for treatment of some autoimmune and tumoral diseases.OBJECTIVE To evaluate the treatment of refractory GF with intralesional rituximab. DESIGN, SETTING, AND PARTICIPANTSIn this case series, 3 patients with biopsy-proved refractory GF who underwent treatment at a dermatology outpatient clinic of a tertiary referral hospital were evaluated. The study was conducted from August 2015 to December 2017.INTERVENTIONS Doses of 0.5 to 1 mL/cm 2 of intralesional rituximab, 10 mg/mL, were administered monthly for 6 months and thereafter depending on clinical response. In 2 patients, peripheral blood B-cell counts were monitored before and during treatment, and in 1 patient, only during treatment. MAIN OUTCOMES AND MEASURESReduction in size of the lesions, ultrasonography evaluation, subjective improvement, and adverse events were monitored throughout the course of therapy.RESULTS All 3 of the patients were men (ages from 30s to 60s). They showed a significant reduction in the size and thickness of GF both clinically and on ultrasonography evaluation. Two patients had a complete response and the third, a partial response. A reduction in peripheral blood B-cell count was observed in the 3 patients, suggesting that the action of rituximab could be systemic. No severe adverse reactions were reported.CONCLUSIONS AND RELEVANCE Intralesional rituximab may represent a novel and well-tolerated therapy for refractory GF.

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Rituximab Therapy for Severe Cutaneous Leukocytoclastic Angiitis Refractory to Corticosteroids, Cellcept and Cyclophosphamide

Cited by 7 publications

References 13 publications

Etiologies and prognostic factors of leukocytoclastic vasculitis with skin involvement

Etiologies and prognostic factors of leukocytoclastic vasculitis with skin involvement

Demographic, etiological and clinical characteristics of patients with leukocytoclastic vasculitis single center experience

Assessment of Treatment of Refractory Granuloma Faciale With Intralesional Rituximab

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