Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report
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...
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| Format: | Article |
| Language: | English |
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Karger Publishers
2023-05-01
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| Series: | Case Reports in Ophthalmology |
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| Online Access: | https://beta.karger.com/Article/FullText/530007 |
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| author | Keigo Sugisawa Hiroshi Takase Takahiko Sugihara Tetsuya Saito Yuko Iwasaki Koju Kamoi Tatsushi Kawaguchi Shinsuke Yasuda Kyoko Ohno-Matsui |
| author_facet | Keigo Sugisawa Hiroshi Takase Takahiko Sugihara Tetsuya Saito Yuko Iwasaki Koju Kamoi Tatsushi Kawaguchi Shinsuke Yasuda Kyoko Ohno-Matsui |
| author_sort | Keigo Sugisawa |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-03-13T06:43:19Z |
| format | Article |
| id | doaj.art-6a286e2f8a2a49eeb4b0693c48022e45 |
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| issn | 1663-2699 |
| language | English |
| last_indexed | 2024-03-13T06:43:19Z |
| publishDate | 2023-05-01 |
| publisher | Karger Publishers |
| record_format | Article |
| series | Case Reports in Ophthalmology |
| spelling | doaj.art-6a286e2f8a2a49eeb4b0693c48022e452023-06-08T12:42:07ZengKarger PublishersCase Reports in Ophthalmology1663-26992023-05-0114121422210.1159/000530007530007Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case ReportKeigo Sugisawa0https://orcid.org/0000-0002-4437-8473Hiroshi Takase1https://orcid.org/0000-0001-8327-803XTakahiko Sugihara2Tetsuya Saito3Yuko Iwasaki4https://orcid.org/0000-0003-0584-0407Koju Kamoi5Tatsushi Kawaguchi6https://orcid.org/0000-0002-1330-8894Shinsuke Yasuda7Kyoko Ohno-Matsui8Department of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Rheumatology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Rheumatology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Rheumatology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanDepartment of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, JapanWe 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.https://beta.karger.com/Article/FullText/530007anti-neutrophil cytoplasmic antibody-associated vasculitisgranulomatosis with polyangiitisretinal detachmentrituximab |
| spellingShingle | Keigo Sugisawa Hiroshi Takase Takahiko Sugihara Tetsuya Saito Yuko Iwasaki Koju Kamoi Tatsushi Kawaguchi Shinsuke Yasuda Kyoko Ohno-Matsui Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report Case Reports in Ophthalmology anti-neutrophil cytoplasmic antibody-associated vasculitis granulomatosis with polyangiitis retinal detachment rituximab |
| title | Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report |
| title_full | Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report |
| title_fullStr | Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report |
| title_full_unstemmed | Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report |
| title_short | Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report |
| title_sort | granulomatosis with polyangiitis complicated by severe exudative retinal detachment and orbital granuloma successfully controlled with rituximab a case report |
| topic | anti-neutrophil cytoplasmic antibody-associated vasculitis granulomatosis with polyangiitis retinal detachment rituximab |
| url | https://beta.karger.com/Article/FullText/530007 |
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