Abstract
Aim
To report three cases of presumed Vogt-Koyanagi-Harada (VKH) disease with unilateral ocular manifestations.
Methods
This retrospective study reviewed the long-term follow-up observations of three patients who attended the uveitis clinic at Tokyo Medical University Hospital. The patients were followed for 5–16 years with systemic clinical, ophthalmologic and laboratory examinations. Ophthalmoscopic findings, extraocular manifestations, visual acuity, and response to corticosteroid administration were evaluated.
Results
Three patients had characteristic clinical features of VKH involving only one eye, including diffuse choroiditis, serous retinal detachment, focal areas of delayed choroidal perfusion, multifocal areas of pinpoint leakage, macular oedema, and optic nerve staining. All patients received systemic corticosteroid therapy during the acute phase of the disease. During the follow-up period (5–16 years), all three patients developed sunset-glow fundus and nummular chorioretinal depigmented scars in the affected eye only, as well as systemic complications of deafness, vitiligo, and poliosis.
Conclusion
The clinical and laboratory features of all three patients were typical of VKH disease except for the unilateral involvement. It is important for ophthalmologists to recognize unilateral VKH disease, even though it is a rare clinical variant of the disease.
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References
Goto H, Mochizuki M, Yamaki K, Kotake S, Usui M, Ohno S (2007) Epidemiological survey of intraocular inflammation in Japan. Jpn J Ophthalmol 51:41–44, doi:10.1007/s10384-006-0383-4
Read RW, Holland GN, Rao NA, Tabbara KF, Ohno S, Arellanes-Garcia L, Pivetti-Pezzi P, Tessler HH, Usui M (2001) Revised diagnostic criteria for Vogt-Koyanagi-Harada disease: report of an international committee on nomenclature. Am J Ophthalmol 131:647–652, doi:10.1016/S0002-9394(01)00925-4
Forster DJ, Green RL, Rao NA (1991) Unilateral manifestation of the Vogt-Koyanagi-Harada syndrome in a 7-year-old child. Am J Ophthalmol 111:380–382
Kouda N, Sasaki H, Harada S, Yamada Y, Takahashi N, Sasaki K (2002) Early manifestation of Vogt-Koyanagi-Harada disease as unilateral posterior scleritis. Jpn J Ophthalmol 46:590–593, doi:10.1016/S0021-5155(02)00545-2
Cimino L, Auer C, Herbort CP (2000) Sensitivity of indocyanine green angiography for the follow-up of active inflammatory choriocapillaropathies. Ocul Immunol Inflamm 8:275–283, doi:10.1076/ocii.8.4.275.6462
Dozono K, Noda Y, Ariyama A, Takada Y, Kawano Y, Inomata H (2004) A case of suspected Vogt-Koyanagi-Harada disease initially manifested as unilateral symptoms of posterior scleritis. Folia Ophthalmol Jpn 55:403–408
Ohno S (1992) Immunogenetic and molecular genetic studies on ocular disease. Nippon Ganka Gakkai Zasshi 96:1158–1179
Acknowledgment
This study was supported in part by Grant-in-Aid 19791294 for Scientific Research from the Japan Society for the Promotion of Science.
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The authors have no proprietary interest in this study. The authors have full control of all primary data, and they agree to allow Graefe's Archive for Clinical and Experimental Ophthalmology to review their data upon request.
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Usui, Y., Goto, H., Sakai, Ji. et al. Presumed Vogt-Koyanagi-Harada disease with unilateral ocular involvement: report of three cases. Graefes Arch Clin Exp Ophthalmol 247, 1127–1132 (2009). https://doi.org/10.1007/s00417-009-1068-8
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DOI: https://doi.org/10.1007/s00417-009-1068-8