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2.
Immun Inflamm Dis ; 12(4): e1250, 2024 Apr.
Artículo en Inglés | MEDLINE | ID: mdl-38661242

RESUMEN

BACKGROUND: Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is responsible for coronavirus disease 2019 (COVID-19), a complex and multifaceted illness. COVID-19 is associated with various ocular manifestations including conjunctivitis, retinal vein occlusion and optic neuritis. However, the case of Vogt-Koyanagi-Harada (VKH) disease associated with SARS-CoV-2 is infrequent, and the specific association is still unclear. CASE PRESENTATION: In the present study, a 35-year-old female patient without any significant medical history presented with 1 week of bilateral blurred vision, occurring 2 weeks after a clinical course of COVID-19. Upon examination, both eyes exhibited bullous serous retinal detachments. She was diagnosed with incomplete VKH disease. Early diagnosis and treatment of VKH disease are essential for the visual prognosis of this aggressive disease. In this particular patient, ocular inflammatory signs and visual acuity improved via corticosteroid therapy. It is worth noting that the occurrence of VKH disease associated with SARS-CoV-2 is uncommon, and the specific connection between the two remains unknown. We review and summarize the clinical characteristics of VKH disease following SARS-CoV-2 infection, and discuss the potential mechanisms that may explain this phenomenon, based on similar studies previously reported. CONCLUSION: Despite the unclear causality, it is important for ophthalmologists and physicians to be recognizant of the possible association between VKH disease and COVID-19. SARS-CoV-2 may play a potential immunological triggering role in VKH disease. However, further in-depth research is necessary to investigate the clinical and epidemiological features, as well as the underlying mechanisms of this association.


Asunto(s)
COVID-19 , Síndrome Uveomeningoencefálico , Adulto , Femenino , Humanos , Corticoesteroides/uso terapéutico , COVID-19/complicaciones , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Agudeza Visual
3.
BMC Ophthalmol ; 24(1): 148, 2024 Apr 02.
Artículo en Inglés | MEDLINE | ID: mdl-38566041

RESUMEN

BACKGROUND: Bilateral retinal detachment and choroidal detachment in a patient are rare occurrences. The presence of bilateral diabetic retinopathy (DR) in such a case is even rarer and complicates the condition. CASE PRESENTATION: In this study, we document a case of unconventional VKH. Manifestations in this patient included intense peripheral retinal detachment and choroidal detachment, along with vitreous opacities akin to cotton wool spots, concurrent with DR. The diagnosis was considered as probable VKH with DR. Treatment according to VKH protocols, including high-dose corticosteroids, yielded positive results. CONCLUSIONS: VKH can co-occurrence with DR. VKH manifestations vary, and early, aggressive, and long-term treatment is essential. The complexity of treatment increases with concurrent DR, necessitating the use of immunosuppressants.


Asunto(s)
Efusiones Coroideas , Diabetes Mellitus , Retinopatía Diabética , Papiledema , Desprendimiento de Retina , Síndrome Uveomeningoencefálico , Humanos , Síndrome Uveomeningoencefálico/complicaciones , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Desprendimiento de Retina/etiología , Desprendimiento de Retina/complicaciones , Retinopatía Diabética/complicaciones , Retinopatía Diabética/diagnóstico , Papiledema/etiología
4.
BMC Ophthalmol ; 24(1): 115, 2024 Mar 13.
Artículo en Inglés | MEDLINE | ID: mdl-38481205

RESUMEN

BACKGROUND: Ocular manifestations are known for non-Hodgkin lymphoma, but are rare for Hodgkin lymphoma. We report a case of Vogt-Koyanagi-Harada (VKH) disease presenting as serous retinal detachment and uveitis in both eyes in a child undergoing chemotherapy for Hodgkin lymphoma. CASE PRESENTATION: The patient was a 7-year-old boy with stage IIB Hodgkin lymphoma (nodular lymphocyte predominant type) who was undergoing chemotherapy, including 2 cycles of the OEPA regimen and 1 cycle of the COPDAC regimen. Two days after the end of the COPDAC regimen, the patient complained of headache and of blurred and decreased vision in both eyes. On the basis of optic symptoms, such as uveitis and serous retinal detachment in both eyes, increased cell counts in cerebrospinal fluid, and positivity for human leukocyte antigen (HLA)-DR4 in peripheral blood cells, incomplete VKH disease was diagnosed. Intravenous treatment with high-dose prednisolone (60mg/m2/day) for 7 days improved both visual acuity and serous retinal detachment and enabled the remains of the COPDAC chemotherapy cycle to be administered. With prednisolone treatment, visual acuity improved from 20/500 to 20/20 in the right eye and from 20/63 to 20/25 in the left eye. Because multiple vitiligo lesions later appeared in the abdomen, complete VKH disease was finally diagnosed. CONCLUSION: The onset of VKH disease occurred during chemotherapy for Hodgkin lymphoma. The patient was HLA-DR4-positive and might have had a predisposition to develop autoimmune diseases, including VKH disease. However, the anticancer drugs administered to this patient have not been reported to cause uveitis. Whether Hodgkin lymphoma triggered the development of VKH remains unclear. Early diagnosis of VKH disease and prompt treatment with high-dose prednisone enabled the patient to maintain good visual function despite chemotherapy for Hodgkin lymphoma.


Asunto(s)
Enfermedad de Hodgkin , Desprendimiento de Retina , Síndrome Uveomeningoencefálico , Masculino , Niño , Humanos , Síndrome Uveomeningoencefálico/inducido químicamente , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Desprendimiento de Retina/tratamiento farmacológico , Enfermedad de Hodgkin/diagnóstico , Enfermedad de Hodgkin/tratamiento farmacológico , Glucocorticoides/uso terapéutico , Prednisolona/uso terapéutico
5.
Am J Ophthalmol ; 262: 25-33, 2024 Jun.
Artículo en Inglés | MEDLINE | ID: mdl-38369223

RESUMEN

PURPOSE: To develop a more tailored immunomodulatory treatment (IMT) strategy based on a novel 2-arm risk stratification system in Vogt-Koyanagi-Harada (VKH) patients. DESIGN: A retrospective clinical cohort study. METHODS: Seventy-nine VKH patients in the acute stage were stratified into low- (n = 58) and high-risk (n = 21) groups based on their exposure to risk factors. They were treated with oral glucocorticoids (GCs) plus as-needed (PRN) or first-line IMT. Best corrected visual acuity (BCVA), sunset glow fundus (SGF) occurrence, relapse rate, and systemic adverse events were evaluated during follow-up. RESULTS: Compared with the low-risk group, the high-risk group showed poorer BCVA at baseline (estimated difference 0.51, 95% CI 0.30-0.78; P < .001) and 6-month follow-up (estimated difference 0.08, 95% CI 0.00-0.08; P = .006), higher incidence of SGF at 12 months (52% vs 28%; RR 1.9, 95% CI 1.1-3.4; P = .040), and higher relapse rate at 6 months (24% vs 5%; RR 4.6, 95% CI 1.2-17.5; P = .028) and 12 months (52% vs 12%; RR 4.4, 95% CI 1.9-9.7; P < .001). In the low-risk cohort, no significant difference between the 2 IMT strategies was observed in primary outcomes. In the high-risk cohort, patients with the immediate IMT showed better BCVA (estimated difference -0.20, 95% CI -0.3 to -0.08; P = .007), lower incidence of SGF (27% vs 80%; RR 0.3, 95% CI 0.1-0.9; P = .030), and lower relapse rate (27% vs 80%; RR 0.3, 95% CI 0.1-0.9; P = .030) compared with the PRN regimen. Moreover, the immediate IMT regimen had a higher frequency of systemic adverse events than the PRN regimen (47% vs 7%; RR 7.1, 95% CI 2.5-20.4; P < .001). CONCLUSIONS: High-risk stratification at baseline was associated with poor prognosis. The immediate IMT regimen was only beneficial for high-risk VKH patients regarding visual outcome, SGF, and relapse rate. This study suggests a potential need for a customized IMT strategy for VKH patients.


Asunto(s)
Glucocorticoides , Síndrome Uveomeningoencefálico , Agudeza Visual , Humanos , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/fisiopatología , Estudios Retrospectivos , Masculino , Femenino , Agudeza Visual/fisiología , Adulto , Glucocorticoides/uso terapéutico , Glucocorticoides/administración & dosificación , Persona de Mediana Edad , Medición de Riesgo , Estudios de Seguimiento , Factores de Riesgo , Administración Oral , Recurrencia , Adulto Joven , Tomografía de Coherencia Óptica , Angiografía con Fluoresceína/métodos , Inmunosupresores/uso terapéutico
6.
Ocul Immunol Inflamm ; 32(4): 433-436, 2024 May.
Artículo en Inglés | MEDLINE | ID: mdl-38412247

RESUMEN

PURPOSE: To present a case of Vogt Koyanagi Harada (VKH) associated with autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy syndrome (APECED) in a two-year-old. CASE PRESENTATION: A two-year-old type 1 diabetic with hypothyroidism presented with impaired fixation. Ocular examination revealed right vitritis, choroiditis, a hyperemic disc, and an area of exudative detachment. At the same time, there was no fundus view in the left eye, and ultrasonographic assessment revealed vitritis and a thickened choroid. Patient developed sunset glow fundus with alopecia, poliosis and vitiligo and a diagnosis of complete VKH with APECED was made. CONCLUSION: APECED is a rare endocrine disorder and has been reported to be associated with VKH twice. Likewise, VKH is commonly present in much older patients; this is the first time ever to be diagnosed in a two-year-old child.


Asunto(s)
Poliendocrinopatías Autoinmunes , Síndrome Uveomeningoencefálico , Preescolar , Femenino , Humanos , Diabetes Mellitus Tipo 1/complicaciones , Diabetes Mellitus Tipo 1/diagnóstico , Angiografía con Fluoresceína/métodos , Fondo de Ojo , Poliendocrinopatías Autoinmunes/diagnóstico , Poliendocrinopatías Autoinmunes/complicaciones , Tomografía de Coherencia Óptica , Ultrasonografía , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/complicaciones , Síndrome Uveomeningoencefálico/tratamiento farmacológico
7.
Cesk Slov Oftalmol ; 80(Ahead of print): 1-5, 2024.
Artículo en Inglés | MEDLINE | ID: mdl-38413229

RESUMEN

AIMS: We present the clinical spectrum, the initial clinical presentation with management trends in treating 14 Vogt-Koyanagi-Harada (VKH) disease cases in a tertiary center in the Northern part of Malaysia. CASE SERIES: There were 14 cases of Vogt-Koyanagi-Harada (VKH) disease retrospectively reviewed over five years (from 2015 to 2020). The mean age at presentation was 37.7 years (range 21-64 years), with female predominance (85.7%). All cases presented with acute uveitic stage and bilateral eye involvement. Of them, 11 (78.6%) were probable VKH, and three (21.4%) were incomplete VKH. All patients attended with acute panuveitis at first presentation. The main posterior segment involvement was disc edema in 57.1% (16 out of 28 eyes) and exudative retinal detachment (ERD) in 35.7% (10 out of 28 eyes). Most of them presented with blindness (3/60 and worse) and moderate visual impair- ment (6/18-6/60); 35.71% each, followed by mild visual impairment (6/12-6/18), and severe visual impairment (6/60-3/60); 7.1% each. Ten patients (71.4%) required combination second-line immunomodulatory treatment during subsequent visits, and only four patients (28.6%) responded well to corticosteroid therapy. Most of the cases achieved no visual impairment (64.3%), followed by mild visual impairment (21.4%) and moderate visual impairment (14.3%), and none were severe or blind at the end of follow-up. CONCLUSION: VKH is a potentially blinding illness if there is inadequate control of the disease in the acute stage. Most of our patients achieved good visual outcomes with early immunomodulatory treatment and systemic corticosteroids.


Asunto(s)
Síndrome Uveomeningoencefálico , Humanos , Femenino , Adulto Joven , Adulto , Persona de Mediana Edad , Masculino , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Estudios Retrospectivos , Malasia/epidemiología , Glucocorticoides , Trastornos de la Visión , Ceguera/tratamiento farmacológico
8.
J Int Med Res ; 52(1): 3000605231221081, 2024 Jan.
Artículo en Inglés | MEDLINE | ID: mdl-38170955

RESUMEN

We report a case of Vogt-Koyanagi-Harada (VKH) disease that recurred 46 years after initial treatment. A 59-year-old woman presented with a 2-month history of bilateral vision blurring. She had received her third dose of coronavirus disease 2019 (COVID-19) vaccination 4 months before the onset of blurring. The best-corrected visual acuity (BCVA) was 1.0 in the right eye and 0.15 in the left eye at the initial visit. Iritis and synechia were observed between the lens and iris bilaterally. A sunset glow fundus was found in both eyes with no serous retinal detachments or disc hyperemia. The patient had a history of VKH disease and had been treated with whole-body corticosteroid administration at another hospital when she was 13 years old. The patient was diagnosed with VKH disease recurrence, and oral corticosteroid therapy and corticosteroid eyedrop treatment were initiated. The treatment response was good. At the time of this writing, recurrence had not been observed for 14 months, and the BCVA was 1.0 in both eyes. To our knowledge, this case represents the longest recorded interval of VKH disease recurrence in the literature to date. COVID-19 vaccination might be the cause of long-term well-controlled disease recurrence.


Asunto(s)
Vacunas contra la COVID-19 , COVID-19 , Síndrome Uveomeningoencefálico , Adolescente , Femenino , Humanos , Persona de Mediana Edad , Corticoesteroides/uso terapéutico , Vacunas contra la COVID-19/efectos adversos , Glucocorticoides/uso terapéutico , Síndrome Uveomeningoencefálico/inducido químicamente , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico
9.
Clin Immunol ; 256: 109800, 2023 11.
Artículo en Inglés | MEDLINE | ID: mdl-37821074

RESUMEN

The resistance development of the combination regimen of corticosteroids (CS) with cyclosporin A (CsA) leads to therapeutic failure of some patients with autoimmune diseases. In the male patients with Vogt-Koyanagi-Harada (VKH) disease, we have identified RPS4Y1 as an important resistance gene of the regimen and a functional mediator of chlorambucil (CLB). However, it remains unclear what is responsible for the resistance in female patients. In the present study, we performed RNA sequencing, tandem mass tag (TMT) proteomics, gain- and loss-of-function assays and rescue assays to screen and validate potential resistant mediators. The results showed that only Fc epsilon receptor Ig (FCER1G) exhibited significantly differential expression in CD4+ T cells among female CsA & CS resistant, sensitive and CLB & CsA & CS treated patients at transcription and protein levels. Inhibition of FCER1G was demonstrated to modulate CD4+ T cell resistance to CsA & CS in female patients. Importantly, the inhibition was mediated by elevated DNA methylation in the promoter region of the FCER1G gene. Moreover, we found that the salvage effect of CLB on CsA & CS resistance was mediated by an increased FCER1G expression via DNA demethylation in female patients. Taken together, the downregulation of FCER1G due to DNA hypermethylation is responsible for the resistance to CsA & CS and CLB reverses this resistance by inducing FCER1G expression via DNA demethylation in female patients. Modulation of FCER1G would be a promising sensitization strategy in female patients with resistance to CsA & CS.


Asunto(s)
Ciclosporina , Síndrome Uveomeningoencefálico , Femenino , Humanos , Masculino , Corticoesteroides/uso terapéutico , Ciclosporina/uso terapéutico , Regulación hacia Abajo , Linfocitos T/metabolismo , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Síndrome Uveomeningoencefálico/genética
10.
Retin Cases Brief Rep ; 17(5): 611-615, 2023 Sep 01.
Artículo en Inglés | MEDLINE | ID: mdl-37643052

RESUMEN

PURPOSE: To describe a case of bilateral Vogt-Koyanagi-Harada (VKH)-like uveitis during treatment with dabrafenib and trametinib and three months after discontinuation of nivolumab for malignant melanoma, and discuss the possible (synergistic) role(s) of mitogen-activated protein kinase (MAPK) inhibitors and immune checkpoint inhibitors in its pathophysiology. METHODS: Retrospective case report with fluorescein angiography and optical coherence tomography. RESULTS: A 55-year old patient with metastatic melanoma presented with a complaint of gradually worsening blurry vision in the right eye during treatment with dabrafenib and trametinib, three months after discontinuation of nivolumab. Based on the clinical examination, optical coherence tomography and fluorescein angiography findings, and a thorough laboratory work-up, he was diagnosed with a bilateral VKH-like uveitis without extraocular manifestations. The uveitis responded well to oral corticosteroids. CONCLUSION: Vogt-Koyanagi-Harada-like uveitis is a rare adverse effect of MAPK inhibitors and immune checkpoint inhibitors. Similar pathogenetic mechanisms including a drug-induced autoimmunity targeted against benign and malignant melanocytes may underlie MAPK inhibitor-induced and immune checkpoint inhibitors-induced VKH-like uveitis. In our report, the patient developed a VKH-like uveitis during MAPK inhibition therapy, four months after discontinuation of nivolumab. It is difficult to delineate whether MAPK inhibition alone was responsible for this adverse effect, or whether, on the contrary, potentiation occurred as a result of immune modulation by previous treatment with an immune checkpoint inhibitor. Further cases are needed to further clarify this latter hypothesis.


Asunto(s)
Efectos Colaterales y Reacciones Adversas Relacionados con Medicamentos , Melanoma , Uveítis , Síndrome Uveomeningoencefálico , Masculino , Humanos , Persona de Mediana Edad , Nivolumab/efectos adversos , Síndrome Uveomeningoencefálico/inducido químicamente , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Inhibidores de Puntos de Control Inmunológico , Estudios Retrospectivos , Melanoma/tratamiento farmacológico
11.
Korean J Ophthalmol ; 37(4): 299-306, 2023 08.
Artículo en Inglés | MEDLINE | ID: mdl-37400085

RESUMEN

PURPOSE: We report the estimated incidence of, and risk factors for, recurrent anterior uveitis in patients with initial acute-onset Vogt-Koyanagi-Harada (VKH) disease using survival analyses. METHODS: Patients who were diagnosed with initial acute-onset VKH disease during 2003-2022 at two university hospitals were included. Recurrent anterior uveitis was defined as the first occurrence of the granulomatous anterior uveitis with anterior chamber cells and flare of 2+ or more by the Standardization of Uveitis Nomenclature (SUN) Working Group grading scheme, after the disappearance of conspicuous uveitis and serous retinal detachment for at least 3 months, regardless of systemic or local treatment. The univariate log-rank test and multivariate Cox regression analyses were performed, including patients' demographic characteristics, underlying diseases, presence of prodromal symptoms, duration of visual symptoms, visual acuity, slit-lamp and fundus findings, and height of serous retinal detachment. The treatment method and response to treatment were also included. RESULTS: The estimated incidence was 39.3% at 10 years. Fifteen of 55 patients (27.3%) had recurrent anterior uveitis during the mean follow-up of 4.5 years. The presence of focal posterior synechiae at the diagnosis increased the risk of recurrent anterior uveitis 6.97-fold compared to the absence of synechiae (95% confidence interval [CI], 2.20-22.11; p < 0.001). Use of systemic high-dose steroid therapy more than 7 days after the development of visual symptoms resulted in a hazard ratio of 4.55 (95% CI, 1.27-16.40; p = 0.020). CONCLUSIONS: This study reports the estimated incidence and risk factors of recurrent anterior uveitis in VKH disease from survival analyses. However, because of the retrospective nature of this study, it is hard to confirm the consistency of the medical records regarding risk factors, thus, the presence of focal posterior synechiae can be inconclusive as a risk factor. Further studies are warranted.


Asunto(s)
Desprendimiento de Retina , Uveítis Anterior , Síndrome Uveomeningoencefálico , Humanos , Incidencia , Desprendimiento de Retina/diagnóstico , Desprendimiento de Retina/epidemiología , Desprendimiento de Retina/etiología , Estudios Retrospectivos , Factores de Riesgo , Uveítis , Uveítis Anterior/diagnóstico , Uveítis Anterior/epidemiología , Uveítis Anterior/tratamiento farmacológico , Síndrome Uveomeningoencefálico/complicaciones , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Recurrencia
12.
J Immunother ; 46(8): 295-298, 2023 10 01.
Artículo en Inglés | MEDLINE | ID: mdl-37315200

RESUMEN

Checkpoint inhibition targeting programmed cell-death protein 1 has demonstrated efficacy for a wide range of indications including cutaneous malignancy. However, immune-related adverse events (irAEs), including infrequent but visually impactful ocular irAEs, require careful consideration of treatment options, including medication withdrawal, local corticosteroids, or rarely immunomodulation. This case presents a 53-year-old woman who developed uveitis and mucous membrane ulcers after treatment for numerous cutaneous neoplasms, primarily squamous cell carcinoma, with the programmed cell-death protein 1 inhibitor cemiplimab. Ophthalmic examination revealed diffuse choroidal depigmentation consistent with a Vogt-Koyanagi-Harada-like syndrome. Topical and periocular steroids were used to treat the intraocular inflammation, and cemiplimab was discontinued. Because of ongoing severe uveitis, systemic corticosteroids and corticosteroid-sparing immunosuppression were initiated. Specifically, azathioprine and methotrexate were introduced, but each was stopped due to side effects, prompting the initiation of adalimumab (ADA) treatment. While ADA controlled intraocular inflammation, the squamous cell carcinomas were noted to progress, resulting in the discontinuation of ADA. However, a uveitis recurrence was observed. After a discussion of risks and benefits of biologic immunosuppressive therapy, including the risk of vision loss, ADA was restarted with successful disease quiescence at a 16-month follow-up. The cutaneous neoplasms were managed with topical and intralesional therapies, such as 5-fluorouracil. Recent dermatologic examinations suggested no new cutaneous lesions. This scenario presents the effective use of ADA in an ocular irAE that balances the management of sight-threatening ocular inflammation with the risk of promoting recurrent or de novo neoplastic disease.


Asunto(s)
Neoplasias Cutáneas , Uveítis , Síndrome Uveomeningoencefálico , Femenino , Humanos , Persona de Mediana Edad , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Uveítis/diagnóstico , Adalimumab/uso terapéutico , Inflamación , Neoplasias Cutáneas/tratamiento farmacológico , Corticoesteroides/uso terapéutico
13.
Nat Commun ; 14(1): 3768, 2023 06 24.
Artículo en Inglés | MEDLINE | ID: mdl-37355662

RESUMEN

Biologics are increasingly used to treat Vogt-Koyanagi-Harada disease, but head-to-head comparisons with conventional immunosuppressants are lacking. Here in this randomized trial (Chinese Clinical Trial Registry, ChiCTR2100043061), we assigned 110 patients (27 early-phase and 83 late-phase) to cyclosporine-based immunosuppressant strategy (N = 56) or adalimumab-based biologic strategy (N = 54), each combined with a modified corticosteroid regimen. The primary outcome is change from baseline in best-corrected visual acuity at week 26. The margin of non-inferiority for cyclosporine is -7 letters. The primary outcome is 11.2 letters (95% CI, 7.5 to 14.9) in the cyclosporine group and 6.3 letters (95% CI, 3.1 to 9.6) in the adalimumab group (difference, 4.9; 95% CI, 0.2 to 9.5; P < 0.001 for non-inferiority). The between-group difference is -0.8 letters (95% CI, -6.1 to 4.5) in early-phase disease and 5.7 letters (95% CI, 0.2 to 11.2) in late-phase. Serious adverse events are reported less frequently in the cyclosporine group than in the adalimumab group (0.70 vs. 1.21 events per patient-year). Here, we report that combined with a non-standard corticosteroid regimen, cyclosporine-based immunosuppressant strategy is non-inferior to adalimumab-based biologic strategy by 26 weeks for visual improvement in a cohort of patients with Vogt-Koyanagi-Harada disease, 75% of whom have a late-phase disease.


Asunto(s)
Productos Biológicos , Síndrome Uveomeningoencefálico , Humanos , Inmunosupresores/uso terapéutico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Adalimumab/uso terapéutico , Ciclosporina/uso terapéutico , Productos Biológicos/uso terapéutico
14.
BMC Ophthalmol ; 23(1): 199, 2023 May 05.
Artículo en Inglés | MEDLINE | ID: mdl-37147563

RESUMEN

BACKGROUNDS: To characterize the acute phase clinical manifestations and visual outcomes of the patients with Vogt-Koyanagi Harada (VKH) disease in southern China. METHODS: In total, 186 patients with acute-onset VKH disease were recruited. The demographic data, clinical signs, ophthalmic examinations, and visual outcomes were analyzed. RESULTS: Among the 186 VKH patients, 3 were diagnosed as complete VKH, 125 as incomplete VKH, and 58 as probable VKH. All patients visited the hospital within 3 months of onset and complained of decreased vision. For the extraocular manifestations, 121 patients (65%) referred neurological symptoms. Anterior chamber activity was negative in most eyes within an onset of 7 days, which increased slightly with onset beyond 1 week. Exudative retinal detachment (366 eyes, 98%) and optic disc hyperaemia (314 eyes, 84%) were commonly observed at presentation. A typical ancillary examination helped with the diagnosis of VKH. Systemic corticosteroid therapy was prescribed. The logMAR best-corrected visual acuity improved significantly from 0.74 ± 0.54 at baseline to 0.12 ± 0.24 at the 1-year follow-up visit. The recurrence rate was 18% in the follow-up visits. Erythrocyte sedimentation rate and C-reactive protein were significantly correlated to VKH recurrences. CONCLUSION: Posterior uveitis, followed by mild anterior uveitis, is the typical initial manifestation in the acute phase of Chinese VKH patients. Visual outcome improvement is promising in most patients receiving systemic corticosteroid therapy in the acute phase. Detection of the clinical features at the initial onset of VKH could facilitate early treatment and better vision improvement.


Asunto(s)
Desprendimiento de Retina , Uveítis Posterior , Síndrome Uveomeningoencefálico , Humanos , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Visión Ocular , Enfermedad Aguda , Corticoesteroides/uso terapéutico , Estudios Retrospectivos
16.
Photodiagnosis Photodyn Ther ; 42: 103634, 2023 Jun.
Artículo en Inglés | MEDLINE | ID: mdl-37244453

RESUMEN

PURPOSE: To investigate the diagnostic value of optical density ratio (ODR) in various diseases with subretinal fluid (SRF) due to different pathophysiologies. METHODS: Patients with acute central serous chorioretinopathy, CSCR (n = 49), Vogt Koyanagi Harada disease, VKH (n = 34), and choroidal hemangioma (n = 17) characterized with SRF were included. Spectral-domain optical coherence tomography (SD-OCT) images were analyzed using ImageJ by three independent readers. The ODRs were calculated using "region of interest (ROI)" and "entire region (TOTAL)" selection methods from the SRF to the vitreous, retinal nerve fiber layer (RNFL), and retinal pigment epithelium (RPE) reflectivity ratios. A correlation analysis between age, central macular thickness (CMT), SRF height, SRF width, and ODRs were obtained. RESULTS: Optical density (OD) measurement was highly reproducible (intraclass correlation coefficient> 0.9). Optical density of the SRF, vitreous, RNFL, and signal strength were comparable (p = 0.360, p = 0.247, p = 0.105, and 0.628, respectively). There was no difference in SRF OD measurements between the two methods (p = 0.401), while there was a significant difference in vitreous OD measurements (p = 0.016). ANOVA test of ODR(ROI), ODR(TOTAL), ODR-RPE (ROI) and ODR-RNFL (ROI) revealed no significant difference among acute CSCR, VKH disease and choroidal hemangioma groups (p > 0.05 for all). Correlation analysis revealed a significant negative correlation between SRF height (p < 0.05) and CMT (p < 0.01) with SRF ODR(ROI). CONCLUSION: ODR measurement appears to be a highly repeatable SD-OCT parameter for diseases characterized with SRF collection. Despite variations in their pathophysiology, the ODR was not statistically different in acute CSCR, VKH disease, and choroidal hemangioma.


Asunto(s)
Coriorretinopatía Serosa Central , Neoplasias de la Coroides , Hemangioma , Fotoquimioterapia , Síndrome Uveomeningoencefálico , Humanos , Coriorretinopatía Serosa Central/diagnóstico por imagen , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Estudios Transversales , Líquido Subretiniano/diagnóstico por imagen , Estudios Retrospectivos , Fotoquimioterapia/métodos , Fármacos Fotosensibilizantes/uso terapéutico , Enfermedad Aguda , Neoplasias de la Coroides/diagnóstico por imagen , Tomografía de Coherencia Óptica/métodos , Hemangioma/diagnóstico por imagen , Angiografía con Fluoresceína/métodos
17.
Retina ; 43(9): 1496-1505, 2023 09 01.
Artículo en Inglés | MEDLINE | ID: mdl-37224445

RESUMEN

PURPOSE: To report the clinical profile and outcomes of acute Vogt-Koyanagi-Harada disease with a strict immunosuppression regimen and investigate the risk factors for a prolonged disease course. METHODS: A total of 101 patients with acute Vogt-Koyanagi-Harada (202 eyes) with more than 24 months of follow-up were recruited from January 2011 to June 2020. They were divided into two groups according to the interval between the onset of Vogt-Koyanagi-Harada and treatment. Oral prednisone was gradually tapered off by a diminished dose according to a relatively strict protocol. Patient responses to the treatment regimen were classified as long-term drug-free remission or chronic recurrent. RESULTS: Ninety-six patients (95.0%) achieved long-term drug-free remission without recurrence, while 5 (5.0%) had chronic recurrence. Most patients achieved good best-corrected visual acuity (90.6% ≧20/25). A generalized estimation equation model demonstrated that time of visit, ocular complications, and cigarette smoking were independent risk factors for a longer disease course, and smokers required a higher drug dose and longer treatment course than nonsmokers. CONCLUSION: An immunosuppressive regimen with an appropriate tapering speed can lead to long-term drug-free remission in patients with acute Vogt-Koyanagi-Harada. Cigarette smoking significantly affects ocular inflammation.


Asunto(s)
Síndrome Uveomeningoencefálico , Humanos , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Glucocorticoides , Enfermedad Aguda , Inmunosupresores/uso terapéutico , Prednisona/uso terapéutico , Progresión de la Enfermedad , Estudios Retrospectivos
18.
Ocul Immunol Inflamm ; 31(6): 1305-1309, 2023 Aug.
Artículo en Inglés | MEDLINE | ID: mdl-37084286

RESUMEN

PURPOSE: The use of immunomodulatory therapy in the setting of coexistence of uveitis and coronavirus disease (COVID-19) remains controversial. We report a case of COVID-19 during systemic steroid therapy for Vogt-Koyanagi-Harada disease (VKH). CASE REPORT: A 43-year-old female was diagnosed with VKH and started on steroid pulse therapy (1,000 mg/day) followed by high-dose oral corticosteroids. Two weeks after discharge from the hospital, she was readmitted to the intensive care unit with severe acute respiratory syndrome due to SARS-CoV-2 infection confirmed by PCR test, and fortunately both VKH and COVID-19-induced respiratory disease improved. CONCLUSION: Given the absence of international agreement on how to manage COVID-19 patients with steroid-dependent VKH, existing clinical guidelines should be reviewed thoroughly to formulate useful strategies for managing VKH patients on steroid treatment who contract COVID-19. Furthermore, the outcomes of patients with steroid-dependent autoimmune uveitis including VKH who develop COVID-19 should be analyzed.


Asunto(s)
COVID-19 , Uveítis , Síndrome Uveomeningoencefálico , Femenino , Humanos , Adulto , Síndrome Uveomeningoencefálico/complicaciones , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , SARS-CoV-2 , Uveítis/diagnóstico , Uveítis/tratamiento farmacológico , Esteroides/uso terapéutico
19.
Int Ophthalmol ; 43(8): 2857-2866, 2023 Aug.
Artículo en Inglés | MEDLINE | ID: mdl-36929369

RESUMEN

PURPOSE: To investigate the incidence and pre/post-treatment risk factors of glaucoma in patients with Vogt-Koyanagi-Harada (VKH) disease. METHODS: Data regarding secondary glaucoma were collected from the medical records of patients with VKH disease who were followed up at the uveitis service at Hiroshima University for more than 6 months. We examined the incidence of glaucoma and pre/post-treatment risk factors for glaucoma in patients with VKH disease. RESULTS: Forty-nine patients with VKH disease were included in this study (31 women and 18 men). The mean age at onset was 50.4 ± 15.4 years and the mean length of follow-up was 40.7 ± 25.5 months. The most common initial treatment was pulse intravenous corticosteroid therapy (89.8%). Fifteen patients developed secondary glaucoma during follow-up. The median time of glaucoma onset from VKH development was 4.5 months (range 0-44 months). Disc swelling type as a pre-treatment factor (p = 0.089, hazard ratio = 7.268), worse final best corrected visual acuity (p = 0.099, odds ratio = 1.545), and cataract progression (p = 0.076, odds ratio = 7.886) as post-treatment factors showed trends for glaucoma development. The patients who progressed to the chronic recurrent stage had more complications including glaucoma. CONCLUSION: Secondary glaucoma occurred in more than 30% of patients with VKH disease. The factors that showed a trend toward glaucoma development may reflect an association with delayed treatment initiation and prolonged ocular inflammation.


Asunto(s)
Glaucoma , Síndrome Uveomeningoencefálico , Masculino , Humanos , Femenino , Síndrome Uveomeningoencefálico/complicaciones , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Incidencia , Agudeza Visual , Estudios Retrospectivos , Glaucoma/diagnóstico , Glaucoma/epidemiología , Glaucoma/etiología , Factores de Riesgo
20.
Ophthalmic Surg Lasers Imaging Retina ; 54(3): 183-187, 2023 03.
Artículo en Inglés | MEDLINE | ID: mdl-36944065

RESUMEN

The use of checkpoint inhibitors has been associated with multiple ocular and orbital complications including Vogt-Koyanagi-Harada disease. In the current case, a 55-year-old man presented with visual changes 3 months following discontinuation of nivolumab for metastatic renal cell carcinoma. This is the second report of delayed presentation following discontinuation of a checkpoint inhibitor and the only case not associated with an alternative targeted therapy at the time of presentation. This article highlights a unique presentation of delayed checkpoint inhibitor-associated Vogt-Koyanagi-Harada and summarizes the reported cases. [Ophthalmic Surg Lasers Imaging Retina 2023;54(3):183-187.].


Asunto(s)
Inhibidores de Puntos de Control Inmunológico , Nivolumab , Síndrome Uveomeningoencefálico , Humanos , Masculino , Persona de Mediana Edad , Carcinoma de Células Renales/tratamiento farmacológico , Carcinoma de Células Renales/complicaciones , Ojo , Neoplasias Renales/tratamiento farmacológico , Neoplasias Renales/complicaciones , Nivolumab/efectos adversos , Síndrome Uveomeningoencefálico/inducido químicamente , Síndrome Uveomeningoencefálico/diagnóstico , Síndrome Uveomeningoencefálico/tratamiento farmacológico , Inhibidores de Puntos de Control Inmunológico/efectos adversos
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