B‐cell depletion with rituximab for refractory head and neck Wegener’s granulomatosis: a cohort study

Pero, Marcos Martinez Del; Chaudhry, Afzal; Jones, Rachel; Sivasothy, Pasupathy; Jani, Piyush; Jayne, David

doi:10.1111/j.1749-4486.2009.01968.x

Cited by 86 publications

(58 citation statements)

References 25 publications

(31 reference statements)

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“…In one of the patients, dyspnoea and subglottic stenosis improved significantly after fourth RTX pulse and the disease went into remission, whereas the second patient displayed further disease progression [23]. In some studies, patients with endobronchial and subglottic lesions were not studied in detail [10,22]. We observed no clinical improvement in 3 patients with endobronchial disease nor in two patients with tracheal-subglottic stenosis in response to RTX treatment.…”

Section: Discussionmentioning

confidence: 58%

“…Of note, ocular biopsy samples from two patients obtained pre-RTX therapy showed the presence of numerous CD20+ cells in the ocular tissue, whereas these cells were undetectable posttreatment [21]. Four of five patients with treatment refractory retro-orbital granuloma responded well to RTX as observed in another retrospective cohort study [22]. However, Aries et al [23] reported in a prospective open-labelled study enlargement of the retro-orbital granulomas in three of five patients, and in other two patients, the granuloma size remained unchanged.…”

Section: Discussionmentioning

confidence: 65%

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Off‐Trial Evaluation of the B cell–Targeting Treatment in the Refractory Cases of Antineutrophil Cytoplasmic Antibodies (ANCA)‐Associated Vasculitis: Long‐Term Follow‐Up from a Single Centre

et al. 2012

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The aim of the study was to evaluate long‐term clinical and immunological effects of anti‐B cell treatment in patients with antineutrophil cytoplasmic antibodies (ANCA)‐associated vasculitis refractory to conventional immunosuppressive treatment. Rituximab (RTX) was added to the ongoing immunosuppressive treatment in 29 patients with refractory ANCA‐associated vasculitis. The disease activity was measured using Birmingham Vasculitis Activity Score/Wegener’s granulomatosis (BVAS/WG score), and clinical laboratory variables were recorded. The median BVAS/WG score before treatment was 6 (IQR 3–8), and 28 patients (97%) had disease flare classified either severe (62%) or limited (34%). Six of 29 patients (21%) achieved a complete remission, and 12 (41%) had a treatment response with ≥50% decrease in BVAS/WG score at 6 months. Fourteen patients (64%) with kidney involvement achieved remission, and in seven patients (50%), no flare was seen during the follow‐up period. Three patients had renal flare and were successfully re‐treated with RTX. Seventeen patients had disease symptoms from airways and eyes at RTX initiation, whereas only 29% displayed ≥50% treatment response. Limited clinical improvement was seen in patients with endobronchial lesions and trachea‐subglottic granulomatous disease. RTX is a potent therapeutic option for ANCA‐associated vasculitis refractory to conventional treatment. Best response may be expected in patients with vasculitic manifestations.

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Section: Discussionmentioning

confidence: 58%

Section: Discussionmentioning

confidence: 65%

Off‐Trial Evaluation of the B cell–Targeting Treatment in the Refractory Cases of Antineutrophil Cytoplasmic Antibodies (ANCA)‐Associated Vasculitis: Long‐Term Follow‐Up from a Single Centre

et al. 2012

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“…m given every fourth week. Martinez del Pero et al also demonstrated positive response with 1 g dosing in four of five patients with retro-orbital GPA, although two of four responders experienced recurrence after 6 months and required alternative therapy [82]. Similarly, in a recent study evaluating maintenance therapy for systemic GPA, more patients experienced sustained remission with rituximab than azathioprine after induction therapy with corticosteroids and cyclophosphamide [83].…”

Section: Granulomatosis With Polyangitismentioning

confidence: 91%

Orbital inflammatory disease management

Moore

Rootman

2016

Expert Review of Ophthalmology

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“…A full clinical response was reported in all cases but 1, who died because of interstitial pneumonia in course ofcyclophosphamide therapy (10). Other cases ofrefractory meningitis -in course of cyclophosphamide therapy -successfully treated with infliximab or rituximab have been reported in recent years (16)(17)(18).…”

Section: Fig 1 Bone Thickening and Hypoattenuation Ofthe Fronto-ethmentioning

confidence: 99%

Meningeal Involvement in Wegener Granulomatosis: Case Report and Review of the Literature

Soriano

Vullo

Casale

et al. 2012

Int J Immunopathol Pharmacol

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Wegener Granulomatosis (WG) is a multisystem autoimmune disorder characterized by necrotizing granulomatous vasculitis that most commonly involves the upper respiratory tract, lungs, and kidneys. The involvement of the central nervous system (CNS) is infrequent and can cause stroke, cranial nerve abnormalities, cerebrovascular events, seizures, and meningeal involvement. Meningeal involvement is rare and may occur due to local vasculitis, directly spread from adjacent disease in the skull base, paranasal or orbital region. We describe the case ofa 20-year-old Caucasian man who was diagnosed with sinonasal WG with frontal focal meningeal involvement. A literature review on diagnosis and treatment of meningeal involvement in course of WG was carried out. The importance of an early diagnosis and treatment of localized WG has been emphasized, in order to avoid the progression to a severe form of disease, especially in younger patients and in paucisymptomatic cases. Case reportA twenty-year-old Caucasian man with history of parietaria allergy presented with nasal congestion, occasional epistaxis and purulent rhinorrhea that had started 1 year previously. He had been seen at another hospital and had received a diagnosis of allergic rhinosinusitis. Large doses of intranasal steroids and multiple courses ofantibiotic therapy did not result in an improvement. An X-ray of the paranasal sinuses carried out six months previously was negative. His familial history was unremarkable; his parents were both living and well; the patient was the only child, he was a medical student at University in his own city, he was single and lived with his parents. He had no allergies to medications, and he denied use of alcohol, illicit drugs and smoking.On admission to our Department, headache, neck stiffness, dyspnoea, cough, fever, night sweats, fatigue, myalgia or arthralgia were absent. On examination, the patient was alert, oriented, and cooperative. There was nasal congestion and copious discharge. The lungs were clear on the auscultation, the cardiac and abdominal examinations were normal. Neurologic examination was negative for focal motor or sensory deficits as well as examination of cranial nerves II through XII. A complete blood count revealed a white-cell count of 8,430 per cubic millimeter, with 46% polymorphonuclear cells. The hematocrit was 41%, and the platelet count 296,000 per cubic millimeter. The blood urea nitrogen level was 27 mg per deciliter, and the creatinine level 0.70 mg per deciliter. Erythrocyte sedimentationrate (ESR)

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B‐cell depletion with rituximab for refractory head and neck Wegener’s granulomatosis: a cohort study

Cited by 86 publications

References 25 publications

Off‐Trial Evaluation of the B cell–Targeting Treatment in the Refractory Cases of Antineutrophil Cytoplasmic Antibodies (ANCA)‐Associated Vasculitis: Long‐Term Follow‐Up from a Single Centre

Off‐Trial Evaluation of the B cell–Targeting Treatment in the Refractory Cases of Antineutrophil Cytoplasmic Antibodies (ANCA)‐Associated Vasculitis: Long‐Term Follow‐Up from a Single Centre

Orbital inflammatory disease management

Meningeal Involvement in Wegener Granulomatosis: Case Report and Review of the Literature

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