Successful reconstruction of an ocular defect resulting from granulomatosis with polyangiitis, following treatment with rituximab

Kenny, Grace; Holl‐Ulrich, Konstanze; Fulcher, Timothy; McElnea, Elizabeth; Kavanagh, Eoin C.; Mulligan, Niall; Molloy, Eamonn; McCarthy, Géraldine

doi:10.1016/j.ajoc.2018.03.014

Cited by 4 publications

(3 citation statements)

References 16 publications

(20 reference statements)

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“…discontinuation [13]. In the present case, rituximab effectively re-induced and maintained remission after recurrence.…”

Section: Case Reports In Ophthalmologysupporting

confidence: 56%

“…Although the sample size of that study was small, the findings suggest the effectiveness of rituximab for ANCA-associated vasculitis with ocular lesions. In a previous report of GPA with retinal detachment (case 4), rituximab therapy permitted prednisolone and cyclophosphamide discontinuation [13] . In the present case, rituximab effectively re-induced and maintained remission after recurrence.…”

Section: Discussionmentioning

confidence: 92%

“…In the current case, detailed examinations, including MRI and comprehensive blood tests, and consultation with rheumatologists led to the final diagnosis of GPA with retinal detachment. Only 9 cases of non-rhegmatogenous retinal detachment due to GPA have been reported [6][7][8][9][10][11][12][13], and detailed information is available for only 4 cases (Table 2). Cyclophosphamide was administered in all 4 cases and was effective in three.…”

Section: Discussionmentioning

confidence: 99%

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Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report

Sugisawa¹,

Takase²,

Sugihara³

et al. 2023

Case Rep Ophthalmol

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We report a rare case of severe exudative retinal detachment with orbital granuloma associated with granulomatosis with polyangiitis (GPA). A 42-year-old man developed bilateral conjunctival hyperemia and eye pain 15 months before presenting to us. Because vitreous cells and retinal detachment were detected in his left eye, he was referred to us for further evaluation. The left eye showed scleral edema, cells in the anterior chamber and anterior vitreous, exudative retinal detachment, and elevated white subretinal lesions from the nasal to the inferior parts of the eye fundus. Orbital contrast-enhanced magnetic resonance imaging revealed a granulomatous lesion, retinal detachment, and fluid retention in the left eyeball. Comprehensive rheumatological evaluation revealed proteinase 3 anti-neutrophil cytoplasmic antibody positivity and a history of otitis media, leading to a GPA diagnosis. Methylprednisolone 1,000 mg/day was administered intravenously for 3 days, followed by oral prednisolone and intravenous cyclophosphamide. Although the retinal detachment decreased, scleritis and choroidal detachment relapse were observed in the left eye after the fifth cyclophosphamide administration. After switching from cyclophosphamide to rituximab, the scleritis and choroidal detachment resolved. Remission was successfully maintained with biannual rituximab administration. In this case, we conclude that rituximab was important to re-induce and maintain remission after recurrence. Collaboration with a rheumatologist is essential for proper treatment in related cases. This is the first report of ultra-widefield and multimodal imaging for retinal detachment associated with GPA.

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“…discontinuation [13]. In the present case, rituximab effectively re-induced and maintained remission after recurrence.…”

Section: Case Reports In Ophthalmologysupporting

confidence: 56%

Section: Discussionmentioning

confidence: 92%

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report

Sugisawa¹,

Takase²,

Sugihara³

et al. 2023

Case Rep Ophthalmol

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Current clinical and therapeutic approach to tumour-like mass lesions in granulomatosis with polyangiitis

Padoan

Campaniello

Gatto

et al. 2022

Autoimmunity Reviews

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Rituximab for treatment of non-infectious and non-malignant orbital inflammatory disease

Cunningham

2021

J Ophthal Inflamm Infect

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Purpose To provide a comprehensive review of rituximab use for the treatment of non-infectious/non-malignant orbital inflammation. Methods Review of literature through January 2021. Results Individual data was available for 167 patients with refractory non-infectious/non-malignant orbital inflammation who received treatment with rituximab (RTX). Rituximab was generally utilized as third-line or later treatment (108/149, 72.5%) at a mean of 44.6 months following the diagnosis of orbital inflammation (range = 0 to 360 months; median = 13.7 months). Patients with non-infectious/non-malignant orbital inflammation either received prior treatment with corticosteroids only (27/122, 22.1%), or with one (31/122, 25.4%), two (25/122, 20.5%), or three or more (25/122, 20.5%) corticosteroid-sparing immunosuppressive agents with or without corticosteroids before initiation of RTX treatment. The rheumatologic protocol (two infusions of 1 gram of RTX separated by 14 days) was utilized most frequently (80/144, 55.6%), followed by the oncologic protocol (four weekly infusions of 375 mg/m2 RTX; 51/144, 35.4%). Various other off-label regimens were used infrequently (13/144, 9.0%). Rituximab treatments resulted in a positive therapeutic response for the majority of patients with orbital inflammation (146/166, 88.0%). Commonly treated diagnoses included granulomatosis with polyangiitis (99/167, 59.3%), IgG-4 related disease (36/167, 21.6%), and orbital inflammation of indeterminate cause (25/167, 15.0%). No side effects were reported in 83.3% (55/66) of cases. The most common RTX-induced adverse event was an infusion-related temporary exacerbation of orbital disease (4/66, 6.1%), which occurred prior to the routine use of systemic corticosteroids as pre-conditioning. Conclusions Overall, RTX appears to be both efficacious and well-tolerated as second- or third-line therapy for patients with non-infectious/non-malignant orbital inflammation.

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Successful reconstruction of an ocular defect resulting from granulomatosis with polyangiitis, following treatment with rituximab

Cited by 4 publications

References 16 publications

Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report

Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report

Current clinical and therapeutic approach to tumour-like mass lesions in granulomatosis with polyangiitis

Rituximab for treatment of non-infectious and non-malignant orbital inflammatory disease

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