Long-term follow-up of patients with birdshot retinochoroidopathy treated with systemic immunosuppression

Matthias D. Becker; Michael S. Wertheim; Justine R. Smith; James T. Rosenbaum

doi:10.1080/09273940490912407

Long-term follow-up of patients with birdshot retinochoroidopathy treated with systemic immunosuppression

Matthias D. Becker
, Michael S. Wertheim
, Justine R. Smith
, James T. Rosenbaum

Ophthalmology

Research output: Contribution to journal › Article › peer-review

49 Scopus citations

Abstract

Purpose: Birdshot retinochoroidopathy (BRC) is a rare uveitis syndrome of presumed autoimmune etiology. Therapy with systemic and periocular corticosteroids is of inconsistent efficacy, attendant with numerous potential long-term side effects. Corticosteroid-sparing strategies with agents such as cyclosporine A or azathioprine have been suggested for this disease. Methods: We retrospectively reviewed the medical charts of patients with BRC who were evaluated consecutively at a tertiary-care, referral-based North American uveitis clinic over a 15-year period. Results: Eleven Caucasian patients (22 eyes) were diagnosed with BRC, representing approximately 1% of all cases seen at the uveitis clinic. HLA-A29 was positive in all 11 patients. We elected to treat five patients with azathioprine, methotrexate, cyclosporine A, mycophenolate mofetil, and/or IvIg, as well as systemic or periocular corticosteroid injections. The median period of follow-up for the five treated patients was six years (range: 8 months-13 years). Inflammation was reduced or stabilized in five of five patients. Conclusion: Although the definitive strategy for the management of BRC is unknown, control of intraocular inflammation and preservation of vision is possible with corticosteroid-sparing immunosuppressive agents.

Original language	English (US)
Pages (from-to)	289-293
Number of pages	5
Journal	Ocular Immunology and Inflammation
Volume	13
Issue number	4
DOIs	https://doi.org/10.1080/09273940490912407
State	Published - Aug 2005

Funding

Acknowledgements: The authors are grateful for the following grants: a research grant from Deutsche Forschungsgemeinschaft, Germany Be 1795/3-1 (MDB), NIH grants EY06484 and EY06477 (JTR), Research to Prevent Blindness Awards (JTR and the Casey Eye Institute), and National Health and Medical Research Council of Australia 997099 (JRS). Correspondence and reprint requests to: Matthias D. Becker, M.D., Ph.D., FEBO, Department of Ophthalmology, Interdisciplinary Uveitis Center, University of Heidelberg, Im Neuenheimer Feld 400, 69120 Heidelberg, Germany. e-mail: Matthias.Becker@urz. uni-heidelberg.de

Funders	Funder number
National Eye Institute and Casey Eye Institute
National Health and Medical Research Council, Australia	997099
Author National Institutes of Health National Institutes of Health National Institutes of Health National Institutes of Health The Bev Hartig Huntington's Disease Foundation National Institutes of Health	EY06477, EY06484
Research to Prevent Blindness
Deutsche Forschungsgemeinschaft	Be 1795/3-1

Keywords

Birdshot syndrome
Immunosuppression
Uveitis

ASJC Scopus subject areas

Immunology and Allergy
Ophthalmology

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10.1080/09273940490912407

Cite this

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title = "Long-term follow-up of patients with birdshot retinochoroidopathy treated with systemic immunosuppression",

abstract = "Purpose: Birdshot retinochoroidopathy (BRC) is a rare uveitis syndrome of presumed autoimmune etiology. Therapy with systemic and periocular corticosteroids is of inconsistent efficacy, attendant with numerous potential long-term side effects. Corticosteroid-sparing strategies with agents such as cyclosporine A or azathioprine have been suggested for this disease. Methods: We retrospectively reviewed the medical charts of patients with BRC who were evaluated consecutively at a tertiary-care, referral-based North American uveitis clinic over a 15-year period. Results: Eleven Caucasian patients (22 eyes) were diagnosed with BRC, representing approximately 1\% of all cases seen at the uveitis clinic. HLA-A29 was positive in all 11 patients. We elected to treat five patients with azathioprine, methotrexate, cyclosporine A, mycophenolate mofetil, and/or IvIg, as well as systemic or periocular corticosteroid injections. The median period of follow-up for the five treated patients was six years (range: 8 months-13 years). Inflammation was reduced or stabilized in five of five patients. Conclusion: Although the definitive strategy for the management of BRC is unknown, control of intraocular inflammation and preservation of vision is possible with corticosteroid-sparing immunosuppressive agents.",

keywords = "Birdshot syndrome, Immunosuppression, Uveitis",

author = "Becker, \{Matthias D.\} and Wertheim, \{Michael S.\} and Smith, \{Justine R.\} and Rosenbaum, \{James T.\}",

note = "Funding Information: Acknowledgements: The authors are grateful for the following grants: a research grant from Deutsche Forschungsgemeinschaft, Germany Be 1795/3-1 (MDB), NIH grants EY06484 and EY06477 (JTR), Research to Prevent Blindness Awards (JTR and the Casey Eye Institute), and National Health and Medical Research Council of Australia 997099 (JRS). Correspondence and reprint requests to: Matthias D. Becker, M.D., Ph.D., FEBO, Department of Ophthalmology, Interdisciplinary Uveitis Center, University of Heidelberg, Im Neuenheimer Feld 400, 69120 Heidelberg, Germany. e-mail: Matthias.Becker@urz. uni-heidelberg.de",

year = "2005",

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language = "English (US)",

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AU - Becker, Matthias D.

AU - Wertheim, Michael S.

AU - Smith, Justine R.

AU - Rosenbaum, James T.

N1 - Funding Information: Acknowledgements: The authors are grateful for the following grants: a research grant from Deutsche Forschungsgemeinschaft, Germany Be 1795/3-1 (MDB), NIH grants EY06484 and EY06477 (JTR), Research to Prevent Blindness Awards (JTR and the Casey Eye Institute), and National Health and Medical Research Council of Australia 997099 (JRS). Correspondence and reprint requests to: Matthias D. Becker, M.D., Ph.D., FEBO, Department of Ophthalmology, Interdisciplinary Uveitis Center, University of Heidelberg, Im Neuenheimer Feld 400, 69120 Heidelberg, Germany. e-mail: Matthias.Becker@urz. uni-heidelberg.de

PY - 2005/8

Y1 - 2005/8

N2 - Purpose: Birdshot retinochoroidopathy (BRC) is a rare uveitis syndrome of presumed autoimmune etiology. Therapy with systemic and periocular corticosteroids is of inconsistent efficacy, attendant with numerous potential long-term side effects. Corticosteroid-sparing strategies with agents such as cyclosporine A or azathioprine have been suggested for this disease. Methods: We retrospectively reviewed the medical charts of patients with BRC who were evaluated consecutively at a tertiary-care, referral-based North American uveitis clinic over a 15-year period. Results: Eleven Caucasian patients (22 eyes) were diagnosed with BRC, representing approximately 1% of all cases seen at the uveitis clinic. HLA-A29 was positive in all 11 patients. We elected to treat five patients with azathioprine, methotrexate, cyclosporine A, mycophenolate mofetil, and/or IvIg, as well as systemic or periocular corticosteroid injections. The median period of follow-up for the five treated patients was six years (range: 8 months-13 years). Inflammation was reduced or stabilized in five of five patients. Conclusion: Although the definitive strategy for the management of BRC is unknown, control of intraocular inflammation and preservation of vision is possible with corticosteroid-sparing immunosuppressive agents.

AB - Purpose: Birdshot retinochoroidopathy (BRC) is a rare uveitis syndrome of presumed autoimmune etiology. Therapy with systemic and periocular corticosteroids is of inconsistent efficacy, attendant with numerous potential long-term side effects. Corticosteroid-sparing strategies with agents such as cyclosporine A or azathioprine have been suggested for this disease. Methods: We retrospectively reviewed the medical charts of patients with BRC who were evaluated consecutively at a tertiary-care, referral-based North American uveitis clinic over a 15-year period. Results: Eleven Caucasian patients (22 eyes) were diagnosed with BRC, representing approximately 1% of all cases seen at the uveitis clinic. HLA-A29 was positive in all 11 patients. We elected to treat five patients with azathioprine, methotrexate, cyclosporine A, mycophenolate mofetil, and/or IvIg, as well as systemic or periocular corticosteroid injections. The median period of follow-up for the five treated patients was six years (range: 8 months-13 years). Inflammation was reduced or stabilized in five of five patients. Conclusion: Although the definitive strategy for the management of BRC is unknown, control of intraocular inflammation and preservation of vision is possible with corticosteroid-sparing immunosuppressive agents.

KW - Birdshot syndrome

KW - Immunosuppression

KW - Uveitis

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ER -

Long-term follow-up of patients with birdshot retinochoroidopathy treated with systemic immunosuppression

Abstract

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Keywords

ASJC Scopus subject areas

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