Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case
This case report discusses a case of granulomatosis with polyangiitis (GPA) initially presenting with lacrimal gland involvement and subsequently developing additional features. GPA is a disease known for inflammation in the respiratory tract and kidneys. A 63-year-old male patient presented with a...
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| Format: | Article |
| Language: | English |
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Galenos Yayinevi
2024-12-01
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| Series: | Türk Oftalmoloji Dergisi |
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| Online Access: | https://www.oftalmoloji.org/articles/management-of-orbital-granulomatous-polyangiitis-presenting-with-lacrimal-gland-involvement-treatment-of-subsequent-peripheral-ulcerative-keratitis-anterior-uveitis-and-exudative-retinal-detachment-in-a-challenging-case/doi/tjo.galenos.2024.72317 |
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| author | Murat Oklar Elif Sarı Hatice Selen Kanar Hikmet Süslü Tufan Çiftçi Mehmet Engin Tezcan |
| author_facet | Murat Oklar Elif Sarı Hatice Selen Kanar Hikmet Süslü Tufan Çiftçi Mehmet Engin Tezcan |
| author_sort | Murat Oklar |
| collection | DOAJ |
| description | This case report discusses a case of granulomatosis with polyangiitis (GPA) initially presenting with lacrimal gland involvement and subsequently developing additional features. GPA is a disease known for inflammation in the respiratory tract and kidneys. A 63-year-old male patient presented with a mass, swelling, and ptosis in the right upper eyelid. The patient was referred to us when his symptoms had persisted and worsened despite topical and oral antibiotic therapy administered at another center. Based on clinical and laboratory findings from evaluation and consultations, GPA was diagnosed and confirmed by biopsy. The patient initially presented with necrotizing scleritis and later developed peripheral ulcerative keratitis and anterior uveitis, which regressed under cyclophosphamide treatment but progressed to exudative retinal detachment due to orbital involvement. Clinical remission was achieved after adding rituximab therapy. Presentation with lacrimal gland involvement may serve as an initial manifestation of locally aggressive orbital and adnexal GPA, which can exhibit variable clinical features. Rapid diagnosis and aggressive treatment are critical for preserving vision and preventing complications in patients with GPA. |
| format | Article |
| id | doaj-art-8335d11800a943a68cddbec39fe3a64f |
| institution | Kabale University |
| issn | 1300-0659 2147-2661 |
| language | English |
| publishDate | 2024-12-01 |
| publisher | Galenos Yayinevi |
| record_format | Article |
| series | Türk Oftalmoloji Dergisi |
| spelling | doaj-art-8335d11800a943a68cddbec39fe3a64f2025-01-07T08:25:02ZengGalenos YayineviTürk Oftalmoloji Dergisi1300-06592147-26612024-12-0154635836310.4274/tjo.galenos.2024.72317Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging CaseMurat Oklar0https://orcid.org/0000-0001-5254-0613Elif Sarı1https://orcid.org/0009-0009-6303-5685Hatice Selen Kanar2https://orcid.org/0000-0001-6505-9714Hikmet Süslü3https://orcid.org/0000-0002-5370-3442Tufan Çiftçi4https://orcid.org/0009-0000-4056-4511Mehmet Engin Tezcan5https://orcid.org/0000-0002-1753-4936University of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Ophthalmology, İstanbul, TürkiyeUniversity of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Ophthalmology, İstanbul, TürkiyeUniversity of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Ophthalmology, İstanbul, TürkiyeUniversity of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Neurosurgery, İstanbul, TürkiyeUniversity of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Pathology, İstanbul, TürkiyeUniversity of Health Sciences Türkiye, Kartal Dr. Lütfi Kırdar City Hospital, Clinic of Rheumatology, İstanbul, TürkiyeThis case report discusses a case of granulomatosis with polyangiitis (GPA) initially presenting with lacrimal gland involvement and subsequently developing additional features. GPA is a disease known for inflammation in the respiratory tract and kidneys. A 63-year-old male patient presented with a mass, swelling, and ptosis in the right upper eyelid. The patient was referred to us when his symptoms had persisted and worsened despite topical and oral antibiotic therapy administered at another center. Based on clinical and laboratory findings from evaluation and consultations, GPA was diagnosed and confirmed by biopsy. The patient initially presented with necrotizing scleritis and later developed peripheral ulcerative keratitis and anterior uveitis, which regressed under cyclophosphamide treatment but progressed to exudative retinal detachment due to orbital involvement. Clinical remission was achieved after adding rituximab therapy. Presentation with lacrimal gland involvement may serve as an initial manifestation of locally aggressive orbital and adnexal GPA, which can exhibit variable clinical features. Rapid diagnosis and aggressive treatment are critical for preserving vision and preventing complications in patients with GPA.https://www.oftalmoloji.org/articles/management-of-orbital-granulomatous-polyangiitis-presenting-with-lacrimal-gland-involvement-treatment-of-subsequent-peripheral-ulcerative-keratitis-anterior-uveitis-and-exudative-retinal-detachment-in-a-challenging-case/doi/tjo.galenos.2024.72317granulomatosis with polyangiitislacrimal glandperipheral ulcerative keratitisretinal detachmentnecrotizing scleritisuveitis |
| spellingShingle | Murat Oklar Elif Sarı Hatice Selen Kanar Hikmet Süslü Tufan Çiftçi Mehmet Engin Tezcan Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case Türk Oftalmoloji Dergisi granulomatosis with polyangiitis lacrimal gland peripheral ulcerative keratitis retinal detachment necrotizing scleritis uveitis |
| title | Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case |
| title_full | Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case |
| title_fullStr | Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case |
| title_full_unstemmed | Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case |
| title_short | Management of Orbital Granulomatous Polyangiitis Presenting with Lacrimal Gland Involvement: Treatment of Subsequent Peripheral Ulcerative Keratitis, Anterior Uveitis, and Exudative Retinal Detachment in a Challenging Case |
| title_sort | management of orbital granulomatous polyangiitis presenting with lacrimal gland involvement treatment of subsequent peripheral ulcerative keratitis anterior uveitis and exudative retinal detachment in a challenging case |
| topic | granulomatosis with polyangiitis lacrimal gland peripheral ulcerative keratitis retinal detachment necrotizing scleritis uveitis |
| url | https://www.oftalmoloji.org/articles/management-of-orbital-granulomatous-polyangiitis-presenting-with-lacrimal-gland-involvement-treatment-of-subsequent-peripheral-ulcerative-keratitis-anterior-uveitis-and-exudative-retinal-detachment-in-a-challenging-case/doi/tjo.galenos.2024.72317 |
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