ABSTRACT
We report an unusual case of unilateral frosted branch angiitis associated with Behcet's disease, including a review of previously reported cases. A 39-year-old male with history of recurrent oral and genital ulcers presented with visual loss in his left eye. Fundus findings demonstrated occlusive retinal vasculitis resembling acute frosted branch angiitis. Laboratory examinations including viral markers revealed no abnormal findings except positive HLA-B51. The patient was treated with systemic steroid and cyclosporine. Six months after presentation, new oral ulcers and pseudofolliculitis appeared, and he was diagnosed with Behcet's disease following rheumatology consultation. During follow-up, there was no change in visual acuity of hand movement, and disc neovascularization developed even after complete panretinal photocoagulation. Ocular manifestations of Behcet's disease can present as unilateral frosted branch angiitis, and may consecutively involve in both eyes. Early immunosuppressive treatment is recommended.
Subject(s)
Adult , Humans , Male , Behcet Syndrome/complications , Diagnosis, Differential , Fluorescein Angiography , Fundus Oculi , Magnetic Resonance Imaging , Retinal Artery/pathology , Retinal Vasculitis/diagnosisABSTRACT
PURPOSE: To report a case of severe vaso-occlusive retinopathy with significant decrease of bilateral visual acuity as the first manifestation associated with systemic lupus erythematosus (SLE). CASE SUMMARY: A 23-year-old man was referred to our clinic with bilateral visual impairment of hand motion (HH). Fundus examination revealed severe retinal hemorrhage, cotton-wool patch, occlusive retinal vasculitis with vascular engorgement, and diffuse retinal edema in both eyes. Because of a malar rash on both cheeks, generalized edema was observed on initial examination with hypertension, azotemia, anemia, and thrombocytopenia, The patient was diagnosed with SLE, strongly positive to antinuclear antibody (ANA), and received an intravitreal injection of Bevacizumab (Avastin, Genentech Inc., San Francisco, CA, USA) in the left eye in addition to hemodialysis, transfusion, systemic corticosteroid and immunosuppressant treatment due to lupus nephritis. Eighteen months later, the retinal edema, cotton-wool patch and hemorrhage resolved, leaving epiretinal membrane without traction in his left eye and diffuse degeneration of the right eye. Final visual acuity was HM in the right eye and 20/100 in the left eye. CONCLUSIONS: Vaso-occlusive retinopathy in SLE can result in permanent visual impairment. In a patient with a high possibility of SLE retinopathy, a periodic fundus examination and intensive management of systemic disease should be considered.
Subject(s)
Humans , Anemia , Antibodies, Antinuclear , Antibodies, Monoclonal, Humanized , Azotemia , Cheek , Edema , Epiretinal Membrane , Exanthema , Eye , Hand , Hemorrhage , Hypertension , Intravitreal Injections , Lupus Erythematosus, Systemic , Lupus Nephritis , Papilledema , Renal Dialysis , Retinal Hemorrhage , Retinal Vasculitis , San Francisco , Thrombocytopenia , Traction , Vision Disorders , Visual Acuity , BevacizumabABSTRACT
PURPOSE: To report a case of occlusive retinal vasculitis in Churg-Strauss syndrome. CASE SUMMARY: A-48-year-old man visited our clinic complaining of suddenly decreased visual acuity in the left eye. Two months previously he had an operation for sinusitis, and he had been taking medications for asthma. In the initial examination, his best corrected visual acuity was 0.02 in the left eye. Fundus examination showed whitish, multiple, diffuse ischemic lesions around the posterior pole and slightly engorged retinal veins in the left eye. Fluorescein angiography showed delayed choroidal filling and delayed arteriovenous transit time in the left eye, but no apparently visible obstruction site. The eosinophil count was elevated to 65% in the white blood cell differentiated count, and perinuclear antineutrophil cytoplasmic antibodies (ANCA) with antimyeloperoxidase specificity (MPO-ANCA) was increased, but a chest X-ray was normal. Eosinophil infiltrations in the mucosa of the paranasal sinus were found, and peripheral neuropathy was found in a nerve conduction study. Hence, we diagnosed the patient with Churg-Strauss syndrome accompanied by occlusive retinal vasculitis, and started steroid therapy. Thirty days later after steroid therapy, the best corrected visual acuity of the left eye was 0.4. CONCLUSIONS: In patients with occlusive retinal vasculitis, we need to consider systemic diseases, such as Churg-Strauss syndrome.