Vogt&amp;ndash;Koyanagi&amp;ndash;Harada syndrome &amp;ndash; current perspectives

Baltmr, Abeir; Lightman, Sue; Tomkins-Netzer, Oren

doi:10.2147/opth.s94866

Cited by 63 publications

(63 citation statements)

References 165 publications

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“…Another clue for us to diagnose VKH has been, unexpectedly, MR imaging with signs of choroiditis and retinal detachments. Unspecific brain abnormalities have been reported in VKH too: Pachymeningeal enhancement, few scattered cerebral periventricular white matter lesions with no enhancement, or within the brainstem and peduncles [4,5].…”

Section: Discussionmentioning

confidence: 99%

Vogt-Koyanagi-Harada Syndrome: A Case of Bilateral Steroid-Responsive Loss of Vision

Vecchio¹,

Varrasi²,

Lucchetti³

et al. 2019

Arch Immunol

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We describe a 35-year-old Caucasian woman with a bilateral loss of vision progressed over 10 days, showing optic disk oedema at examination. Serological screening and Cerebrospinal Fluid (CSF) analysis excluded any infective, neoplastic, granulomatous or rheumatologic disorder. Axial brain magnetic resonance imaging showed a bilateral thickening of the choroid with retinal detachments, and few small cerebral periventricular white matter lesions. An ophthalmological review confirmed serous retinal detachments, and performed a retinal fluorescein angiography. These findings suggested an uncommon uveo meningitic disease, the Vogt-Koyanagi-Harada (VKH) syndrome. This rare autoimmune condition classically includes bilateral uveitic manifestations associated with neurological, auditory and skin involvement. When clinical presentation is incomplete, VKH syndrome diagnosis is supported by ocular and brain imaging to start an early treatment, as well as CSF analysis. High-dose steroids or immunosuppressant are the gold-standard treatments: Our patient improved quickly after intravenous methylprednisolone but worsened during tapering so we added Azathioprine, also as steroid-sparing agent, obtaining a complete and permanent recovery.

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Section: Discussionmentioning

confidence: 99%

Vogt-Koyanagi-Harada Syndrome: A Case of Bilateral Steroid-Responsive Loss of Vision

Vecchio¹,

Varrasi²,

Lucchetti³

et al. 2019

Arch Immunol

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“…In children, a shorter interval between symptoms and treatment is linked to better final outcomes of visual acuity . Oral steroid therapy should be gradually reduced over 2 years to prevent recurrence . Patients with steroid‐resistant disease or steroid intolerance can be treated with methotrexate or other antimetabolites as second‐line therapy.…”

Section: Discussionmentioning

confidence: 99%

White Eyelashes and Red Eyes in a 7‐Year‐Old Boy

Plumptre

Polubothu

Dhanes

et al. 2017

Pediatric Dermatology

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“…The absolute number of Treg cell in organ group is fewer than that in non-organ group (p<0.05), and the ratio of Th17/Treg in organ group was significantly higher than that in non-organ group. 4 The peripheral Th17 cell absolute number in patients with skeletal muscle inflammatory oedema was significantly higher than that of non inflammatory oedema patients (p<0.05); 6 The levels of Th17, Tregs and ratio of Th17/Treg did not correlated with pathological features of inflammatory infiltration (p>0.05).…”

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confidence: 95%

“…There are positive efficacy data of rituximab (RTX) for ocular surface inflammatory disease. [1][2][3][4] Objectives: To describe our experience with RTX as a therapy for severe ocular surface inflammatory diseases associated to rheumatic conditions. Methods: This is a retrospective observational study.…”

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confidence: 99%

AB1139 Rituximab for scleritis and peripheral ulcerative keratitis associated with rheumatologic disease

Pascual

Hurtado

Pérez

et al. 2018

Other Orphan Diseases

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BackgroundSome types of ocular surface inflammatory diseases are often related to rheumatic conditions: 37% of scleritis (especially diffuse and necrotizing forms) and 60% of peripheral ulcerative keratitis (PUK). Rheumatoid arthritis (RA) and ANCA-associated vasculitis are the most frequently related conditions. Significant loss of visual acuity can be observed if these ocular diseases are not properly treated. To date, no approved therapies are available. Consequently, the management of these ocular diseases are based on published evidence coming from clinical trials (scarce and often with a low sample size), observational studies and case reports. There are positive efficacy data of rituximab (RTX) for ocular surface inflammatory disease.1–4 ObjectivesTo describe our experience with RTX as a therapy for severe ocular surface inflammatory diseases associated to rheumatic conditions.MethodsThis is a retrospective observational study. It includes patients with severe scleritis or PUK associated to rheumatic diseases diagnosed and managed at our Multidisciplinary Uveitis Clinic between January 2006 and November 2017. We recorded demographic and clinical variables. As outcome variables we used the change in visual acuity and the presence of inflammatory activity by biomicroscopy.ResultsVA: visual acuity, AE: advers events, RA: rheumatoid arthritis, GPA: granulomatosis with polyangiitis, RP: relapsing polychondritis, LTI: latent tuberculous infection, DM: diabetes mellitus, CSC: central serous choroidopathy, MPLN: metilprednisolone, PDN: prednisone, DXM: dexametasone, MTX:methotrexate, CS-A: cyclosporine, LFN: leflunomide, IFX: infliximab, ETN: etanercept, AKR: anakinra TCZ: tocilizumab.ConclusionsAs previously described we consider rituximab as an effective therapy for severe ocular surface inflammatory diseases related to rheumatic conditions when other immunosuppressant drugs fail or are contraindicated.References[1] - Albert M, Beltrán E, Martínez-Costa L. Rituximab in rheumatoid arthritis-associated peripheral ulcerative keratitis. Arch Soc Esp Oftalmol. 2011; 86(4):118–20.[2] - Sims J. Scleritis: presentations, disease associations and management. Postgrad Med J. 2012;88(1046):713–8.[3] - Jabs DA, Mudun A, Dunn JP, Mardsh MJ. Episcleritis and scleritis: clinical features and treatment results. Am J Opthalmol2000;130:469.[4] - Suhler EB, Lim LL, Beardsley RM, Giles TR, Pasadhika S, Lee ST, et al. Rituximab therapy for refractory scleritis: results of a phase I/II dose-ranging, randomized, clinical trial. Ophthalmology. 2014;121(10):1885–91.Disclosure of InterestNone declaredAbstract AB1139 – Table 1RTX for scleritis and PUKAge (ys)GenderOcular diseaseRheumatic diseaseComorbiditiesPrevious treatmentRTX cyclesΔVAInflammatory activityAEFollow up (ms) 155FPUKRAAmiloidosisMTX, CS-A, IFX, MPLN ev1+0.1NoNo132258FScleritisGPAHBV chronic carrier, LTIMPLN ev, PDN1+0.1NoNo19359FScleritisRPDMMTX, CS-A, PDN, DXM io10Diffuse nodular scleritisNo17475MScleritisRPDM, Sweet’s syndrome, myelo displasic syndromeMTX, CS-A, ETN, PDN1...

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Vogt–Koyanagi–Harada syndrome – current perspectives

Cited by 63 publications

References 165 publications

Vogt-Koyanagi-Harada Syndrome: A Case of Bilateral Steroid-Responsive Loss of Vision

Vogt-Koyanagi-Harada Syndrome: A Case of Bilateral Steroid-Responsive Loss of Vision

White Eyelashes and Red Eyes in a 7‐Year‐Old Boy

AB1139 Rituximab for scleritis and peripheral ulcerative keratitis associated with rheumatologic disease

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