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1.
Retina ; 44(8): 1344-1350, 2024 Aug 01.
Article in English | MEDLINE | ID: mdl-39047128

ABSTRACT

PURPOSE: To evaluate changes in scleral thickness in Vogt-Koyanagi-Harada (VKH) disease. METHODS: This study included 34 eyes of 17 treatment-naïve patients with acute-phase VKH disease. Scleral thickness and the presence of ciliochoroidal effusion were examined using anterior segment optical coherence tomography at baseline and 1 week, 2 weeks, and 12 weeks after the start of corticosteroid treatment. Scleral thickness was measured 6 mm posterior to the scleral spur in four directions. RESULTS: Twenty-eight eyes (82.4%) initially had ciliochoroidal effusion, but this rapidly decreased to nine eyes (26.5%) after 1 week. The sclera with ciliochoroidal effusion became thinner from baseline to 1 week at the superior (400.2 ± 46.9-353.5 ± 47.9 µm), temporal (428.4 ± 53.6-387.8 ± 56.1 µm), inferior (451.5 ± 71.0-400.5 ± 50.5 µm), and nasal (452.4 ± 78.0-407.6 ± 62.9 µm) points (P < 0.01 for all), and no further changes were observed. The sclera without ciliochoroidal effusion remained unchanged. CONCLUSION: In VKH disease, eyes with ciliochoroidal effusion exhibited the maximum scleral thickness during the acute phase. This thickening responded rapidly to treatment and became thinner within 1 week. Inflammation in VKH disease may affect not only the choroid but also the sclera.


Subject(s)
Glucocorticoids , Sclera , Tomography, Optical Coherence , Uveomeningoencephalitic Syndrome , Humans , Uveomeningoencephalitic Syndrome/drug therapy , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/physiopathology , Tomography, Optical Coherence/methods , Sclera/pathology , Sclera/diagnostic imaging , Female , Male , Adult , Middle Aged , Acute Disease , Glucocorticoids/therapeutic use , Glucocorticoids/administration & dosage , Visual Acuity , Young Adult , Retrospective Studies , Aged , Choroid/pathology , Choroid/diagnostic imaging , Choroidal Effusions/diagnosis , Choroidal Effusions/drug therapy
2.
BMJ Case Rep ; 17(6)2024 Jun 27.
Article in English | MEDLINE | ID: mdl-38937264

ABSTRACT

Choroidal neovascular membrane (CNVM) in Vogt-Koyanagi-Harada disease (VKH) is a known entity, observed primarily during the chronic convalescent and chronic-recurrent phases of the disease. However, the peripapillary location of CNVM is a rare finding.We describe a case of chronic VKH with bilateral peripapillary CNVM detected using multimodal imaging and the associated differential diagnoses and treatment approach.A combination of anti-vascular endothelial growth factor injections, systemic steroids and immunosuppressants is often required to manage the aggressive course of this choroidal neovascularisation.


Subject(s)
Choroidal Neovascularization , Tomography, Optical Coherence , Uveomeningoencephalitic Syndrome , Humans , Uveomeningoencephalitic Syndrome/complications , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/drug therapy , Choroidal Neovascularization/drug therapy , Choroidal Neovascularization/etiology , Choroidal Neovascularization/diagnostic imaging , Choroidal Neovascularization/diagnosis , Fluorescein Angiography/methods , Diagnosis, Differential , Male , Female , Adult , Angiogenesis Inhibitors/therapeutic use , Angiogenesis Inhibitors/administration & dosage
3.
BMC Ophthalmol ; 24(1): 238, 2024 Jun 07.
Article in English | MEDLINE | ID: mdl-38849758

ABSTRACT

BACKGROUND: This study explores prognostic factors influencing Vogt-Koyanagi-Harada (VKH) disease and observes the efficacy and safety of Adalimumab (ADA) in treating recurrence in Vogt-Koyanagi-Harada (VKH) patients. METHODS: A retrospective study was conducted on all patients diagnosed with VKH disease at Beijing Tongren Hospital between 2020 and 2023. Clinical data included initial and final visual acuity, age, gender, ocular complications, treatment modalities, disease duration, and recurrence frequency. RESULTS: A total of 62 VKH patients were included, comprising 34 in the acute-resolved group and 28 in the chronic-recurrent group. The mean age of patients in the acute-resolved group was 38.29 ± 15.46 years, while the mean age of chronic-recurrent group had a 49.00 ± 16.43 years. Initial best-corrected visual acuity (BCVA) examination at the first visit showed an average BCVA of 0.64 ± 0.29 logMAR in the acute-resolved group and 1.38 ± 0.54 logMAR in the chronic-recurrent group (p = 0.002). During follow-up, ocular complications were observed in 29.4% of the acute-resolved group patients and 41.7% of the chronic-recurrent group patients (P = 0.006). "Sunset glow fundus" was observed in 23.5% of the acute-resolved group and 64.3% of the chronic-recurrent group patients (P = 0.001). Poor initial BCVA (P = 0.046) and the occurrence of "sunset glow fundus" (P = 0.040) were significantly associated with progression to the chronic recurrent phase. Logistic regression analysis revealed that older age at onset (P = 0.042) and the occurrence of "sunset glow fundus" (P = 0.037) were significant predictors for progression to the chronic recurrent phase. ADA significantly reduced anterior chamber inflammatory cells (P = 0.000) and vitreous cavity inflammatory cells (P = 0.001) in the chronic-recurrent group, and markedly decreased the recurrence rate in VKH patients (P = 0.009). CONCLUSION: In comparison to acute-resolved patients, chronic-recurrent patients exhibited poorer initial BCVA and a significantly increased incidence of "sunset glow fundus." Older age at onset and the occurrence of "sunset glow fundus" at diagnosis are crucial predictive factors for VKH patients progressing to the chronic recurrent phase. ADA effectively alleviates refractory VKH disease and is generally well-tolerated.


Subject(s)
Adalimumab , Recurrence , Uveomeningoencephalitic Syndrome , Visual Acuity , Humans , Uveomeningoencephalitic Syndrome/drug therapy , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/physiopathology , Adalimumab/therapeutic use , Female , Male , Retrospective Studies , Adult , Middle Aged , Visual Acuity/physiology , Chronic Disease , Young Adult , Anti-Inflammatory Agents/therapeutic use , Follow-Up Studies , Adolescent , Treatment Outcome , Aged , Prognosis
4.
BMC Ophthalmol ; 24(1): 240, 2024 Jun 07.
Article in English | MEDLINE | ID: mdl-38849786

ABSTRACT

BACKGROUND: Several immune checkpoint inhibitors (ICIs) have been linked to the occurrence of Vogt-Koyanagi-Harada disease (VKHD)-like uveitis. Among the ICIs, there has been no report of immune-related adverse events (irAEs) caused by a new programmed death protein-1(PD-1) monoclonal antibody (Toripalimab). CASE PRESENTATION: This paper presents a case of VKHD-like uveitis that arose following Toripalimab therapy for urothelial cancer of the bladder, and the patient experienced symptoms 10 days after the final dosage of 20 months of medication treatment. This patient with bladder uroepithelial carcinoma had severe binocular acute panuveitis with exudative retinal detachment after receiving Toripalimab therapy. Binocular VKHD-like uveitis was suggested as a diagnosis. Both eyes recovered after discontinuing immune checkpoint inhibitors and local and systemic corticosteroid treatment. CONCLUSIONS: This report suggests that VKHD-like uveitis can also occur in patients receiving novel PD-1 antibodies and the importance of paying attention to eye complications in patients receiving treatment over a long period.


Subject(s)
Immune Checkpoint Inhibitors , Uveomeningoencephalitic Syndrome , Humans , Uveomeningoencephalitic Syndrome/chemically induced , Uveomeningoencephalitic Syndrome/diagnosis , Immune Checkpoint Inhibitors/adverse effects , Male , Uveitis/chemically induced , Uveitis/diagnosis , Urinary Bladder Neoplasms/drug therapy , Programmed Cell Death 1 Receptor/antagonists & inhibitors , Antibodies, Monoclonal, Humanized/adverse effects , Antibodies, Monoclonal, Humanized/therapeutic use , Female , Middle Aged , Aged , Antineoplastic Agents, Immunological/adverse effects
6.
Immun Inflamm Dis ; 12(4): e1250, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38661242

ABSTRACT

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.


Subject(s)
COVID-19 , Uveomeningoencephalitic Syndrome , Adult , Female , Humans , Adrenal Cortex Hormones/therapeutic use , COVID-19/complications , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/drug therapy , Visual Acuity
8.
BMC Ophthalmol ; 24(1): 148, 2024 Apr 02.
Article in English | MEDLINE | ID: mdl-38566041

ABSTRACT

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.


Subject(s)
Choroidal Effusions , Diabetes Mellitus , Diabetic Retinopathy , Papilledema , Retinal Detachment , Uveomeningoencephalitic Syndrome , Humans , Uveomeningoencephalitic Syndrome/complications , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/drug therapy , Retinal Detachment/etiology , Retinal Detachment/complications , Diabetic Retinopathy/complications , Diabetic Retinopathy/diagnosis , Papilledema/etiology
9.
Eur J Ophthalmol ; 34(4): NP12-NP15, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38629147

ABSTRACT

A 45-year-old male with Vogt-Koyanagi-Harada (VKH) syndrome presented with vision loss in his right eye after discontinuing treatment during the COVID-19 pandemic. He was found to have bullous retinal detachment (RD) in the right eye and was started on subcutaneous adalimumab with oral corticosteroid following three doses of pulse corticosteroid. But when RD did not resolve after 4 months of treatment and ultrasound B scan showed bullous RD with retino-retinal adhesion, he was planned for surgical intervention. During surgery, there was retino-retinal adhesions due to long-standing "kissing exudative RD", causing non-settling exudative RD. Following surgery, the vision improved to 2/60, with attached retina. The patient has been under follow-up with us for the last one year now and developed no recurrence of RD till now. This case emphasises the significance of retinoretinal adhesion in long-standing bullous RD that does not respond to conventional aggressive medical therapy.


Subject(s)
Retinal Detachment , Tomography, Optical Coherence , Uveomeningoencephalitic Syndrome , Visual Acuity , Humans , Male , Retinal Detachment/etiology , Retinal Detachment/diagnosis , Retinal Detachment/surgery , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/complications , Uveomeningoencephalitic Syndrome/drug therapy , Middle Aged , Visual Acuity/physiology , COVID-19/complications , Glucocorticoids/therapeutic use , Glucocorticoids/administration & dosage , SARS-CoV-2 , Vitrectomy
10.
Photodiagnosis Photodyn Ther ; 46: 104061, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38521148

ABSTRACT

A 46-year-old male patient visited our clinic with a complaint of blurred vision in the right eye accompanied by headache and insomnia. The fundus examination showed three bullous retinal detachments in the right eye. Considering the prodromal symptoms and other fundus characteristics such as vitreous cells in the posterior pole and multifocal fluorescence leakages on fundus fluorescein angiography (FFA), initial diagnosis was considered as Vogt-Koyanagi-Harada (VKH). However, oral glucocorticoids didn't improve patient's vision. Further enhanced depth imaging (EDI)-optical coherence tomography (OCT) scan displayed hyper-reflective lesions at the choroidal layer. We proposed that hyper-reflective lesions at the choroidal layer on EDI-OCT may characterize the bullous variant of central serous chorioretinopathy (CSC). After fundus photocoagulation treatment, the patient's vision improved.


Subject(s)
Central Serous Chorioretinopathy , Fluorescein Angiography , Tomography, Optical Coherence , Uveomeningoencephalitic Syndrome , Humans , Uveomeningoencephalitic Syndrome/diagnosis , Male , Middle Aged , Tomography, Optical Coherence/methods , Diagnosis, Differential , Central Serous Chorioretinopathy/diagnosis , Fluorescein Angiography/methods , Retinal Detachment
11.
Acta Ophthalmol ; 102(6): 720-727, 2024 Sep.
Article in English | MEDLINE | ID: mdl-38470990

ABSTRACT

PURPOSE: To investigate the association between pretreatment blood flow velocity in the choroid and optic nerve head (ONH) and retinal oxygen metabolism in the acute uveitic phase and the development of 'sunset glow fundus' in Vogt-Koyanagi-Harada (VKH) disease. METHODS: Retrospective analysis of 41 patients (82 eyes). Laser speckle flowgraphy and retinal oximetry measurements were performed at the presentation. The main outcome measure was the development of 'sunset glow fundus'. RESULTS: Twenty patients (40 eyes) presented in the phase preceding anterior segment inflammation (early presentation), and 21 patients (42 eyes) presented with anterior segment inflammation (late presentation). In ONH, mean blur rate (MBR)-vessel, representing blood flow velocity in retinal vessels, was significantly lower in the late presentation group, while choroidal MBR was not significantly different. The late presentation group had significantly lower oxygen saturation in retinal venules, a higher arteriovenous oxygen saturation difference and a smaller calibre of retinal arterioles compared with the early presentation group. Eyes that subsequently developed 'sunset glow fundus' had significantly lower ONH MBR-vessels, lower oxygen saturation in retinal venules, a higher arteriovenous oxygen saturation difference and a smaller calibre of retinal arterioles compared with eyes without 'sunset glow fundus'. ONH MBR-vessel had a significant negative correlation with arteriovenous oxygen saturation difference and a significant positive correlation with calibre of retinal arterioles. CONCLUSIONS: In the acute uveitic phase of VKH disease, the development of 'sunset glow fundus' is associated with pretreatment reduced retinal blood flow velocity, calibre of retinal arterioles and oxygen saturation in retinal venules, as well as an increased arteriovenous oxygen saturation difference.


Subject(s)
Choroid , Laser-Doppler Flowmetry , Oxygen Consumption , Oxygen , Regional Blood Flow , Retinal Vessels , Uveomeningoencephalitic Syndrome , Humans , Uveomeningoencephalitic Syndrome/physiopathology , Uveomeningoencephalitic Syndrome/metabolism , Uveomeningoencephalitic Syndrome/diagnosis , Male , Retrospective Studies , Female , Adult , Regional Blood Flow/physiology , Oxygen/metabolism , Middle Aged , Acute Disease , Choroid/blood supply , Choroid/metabolism , Retinal Vessels/physiopathology , Retinal Vessels/metabolism , Blood Flow Velocity/physiology , Oxygen Consumption/physiology , Oximetry/methods , Optic Disk/blood supply , Optic Disk/metabolism , Fluorescein Angiography/methods , Oxygen Saturation/physiology , Visual Acuity , Young Adult , Follow-Up Studies , Fundus Oculi
12.
BMC Ophthalmol ; 24(1): 115, 2024 Mar 13.
Article in English | MEDLINE | ID: mdl-38481205

ABSTRACT

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.


Subject(s)
Hodgkin Disease , Retinal Detachment , Uveomeningoencephalitic Syndrome , Male , Child , Humans , Uveomeningoencephalitic Syndrome/chemically induced , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/drug therapy , Retinal Detachment/drug therapy , Hodgkin Disease/diagnosis , Hodgkin Disease/drug therapy , Glucocorticoids/therapeutic use , Prednisolone/therapeutic use
13.
Cesk Slov Oftalmol ; 80(3): 140-144, 2024.
Article in English | MEDLINE | ID: mdl-38413229

ABSTRACT

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.


Subject(s)
Uveomeningoencephalitic Syndrome , Humans , Female , Young Adult , Adult , Middle Aged , Male , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/drug therapy , Retrospective Studies , Malaysia/epidemiology , Glucocorticoids , Vision Disorders , Blindness/drug therapy
14.
Am J Ophthalmol ; 262: 25-33, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38369223

ABSTRACT

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.


Subject(s)
Glucocorticoids , Uveomeningoencephalitic Syndrome , Visual Acuity , Humans , Uveomeningoencephalitic Syndrome/drug therapy , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/physiopathology , Retrospective Studies , Male , Female , Visual Acuity/physiology , Adult , Glucocorticoids/therapeutic use , Glucocorticoids/administration & dosage , Middle Aged , Risk Assessment , Follow-Up Studies , Risk Factors , Administration, Oral , Recurrence , Young Adult , Tomography, Optical Coherence , Fluorescein Angiography/methods , Immunosuppressive Agents/therapeutic use
15.
Ocul Immunol Inflamm ; 32(4): 433-436, 2024 May.
Article in English | MEDLINE | ID: mdl-38412247

ABSTRACT

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.


Subject(s)
Polyendocrinopathies, Autoimmune , Uveomeningoencephalitic Syndrome , Child, Preschool , Female , Humans , Diabetes Mellitus, Type 1/complications , Diabetes Mellitus, Type 1/diagnosis , Fluorescein Angiography/methods , Fundus Oculi , Polyendocrinopathies, Autoimmune/diagnosis , Polyendocrinopathies, Autoimmune/complications , Tomography, Optical Coherence , Ultrasonography , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/complications , Uveomeningoencephalitic Syndrome/drug therapy
16.
Indian J Ophthalmol ; 72(Suppl 4): S580-S583, 2024 Jul 01.
Article in English | MEDLINE | ID: mdl-38189444

ABSTRACT

PURPOSE: To describe disease characteristics and outcomes of Vogt-Koyanagi-Harada (VKH) disease in elderly patients. METHODS: Retrospective analysis of patients older than 50 years with VKH disease at two referral centers in India. Demographics, extraocular and ocular involvement, treatment, complications, and visual acuity outcomes were noted. RESULTS: In total, 69 patients (mean age at presentation: 56.4 ± 4.7 years) were analyzed; 6/69 patients had diabetes mellitus at presentation, and 10/69 had hypertension. Clinical signs included anterior chamber cells >2+ (29%), granulomatous keratic precipitates (23%), disc hyperemia (26%), neurosensory retinal detachment (34.7%), and "sunset-glow" fundus (52.1%). Patients were classified as probable (n = 50, 72.4%), incomplete (n = 18, 26%), and complete VKH (n = 1, 1.4%). The mean follow-up period was 20.2 ± 19.4 months. Improvement in mean BCVA of (0.63 LogMAR, 6 Snellen lines) was noted on the last follow-up. Patients receiving systemic steroids with immunosuppressants ( P < 0.0001) had better visual outcomes at final follow-up compared to steroids alone ( P = 0.103). Eight patients (11.6%) had complications due to systemic immunosuppressants, and 17 patients (24.6%) developed diabetes mellitus or had worsening of diabetes while on systemic corticosteroids. CONCLUSION: Few patients presented with systemic manifestations in our cohort. Those treated with steroids and concurrent immunosuppressants had better outcomes. However, therapy with immunosuppressants was encountered with major dose-limiting complications in a significant number of elderly patients with VKH syndrome.


Subject(s)
Uveomeningoencephalitic Syndrome , Visual Acuity , Humans , Male , Retrospective Studies , Female , Middle Aged , Visual Acuity/physiology , Aged , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/drug therapy , Follow-Up Studies , India/epidemiology , Fundus Oculi , Fluorescein Angiography/methods , Glucocorticoids/therapeutic use
17.
J Int Med Res ; 52(1): 3000605231221081, 2024 Jan.
Article in English | MEDLINE | ID: mdl-38170955

ABSTRACT

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.


Subject(s)
COVID-19 Vaccines , COVID-19 , Uveomeningoencephalitic Syndrome , Adolescent , Female , Humans , Middle Aged , Adrenal Cortex Hormones/therapeutic use , COVID-19 Vaccines/adverse effects , Glucocorticoids/therapeutic use , Uveomeningoencephalitic Syndrome/chemically induced , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/drug therapy
18.
Ocul Immunol Inflamm ; 32(1): 98-104, 2024 Jan.
Article in English | MEDLINE | ID: mdl-36637986

ABSTRACT

PURPOSE: To characterize the ocular inflammatory side effects associated with immune checkpoint inhibitor (CPI) treatment in a Northern California population. DESIGN: Retrospective case series. PARTICIPANTS: Patients receiving CPI within an integrated healthcare delivery system. METHODS: All patients within Kaiser Permanente Northern California receiving CPI between January 1, 2012 and November 1, 2018 were identified. Medical records of those seen in the ophthalmology clinic at least once were retrospectively reviewed. MAIN OUTCOME MEASURES: Type and duration of ocular inflammation, indication for and exposure to CPI, time from exposure to diagnosis of ocular inflammation. RESULTS: 31 cases of ocular inflammation were identified in 5061 patients (0.61%) receiving CPI. Mean ± SD age was 67 ± 11.9 (range 38-89). Mean time from exposure to diagnosis was 6.8 ± 5.5 months (range 0.5-17). 87% of cases were bilateral, and 43% of cases were chronic. Average ophthalmology follow-up was 16 ± 18 months (range 0-71). 16/31 (52%) had anterior uveitis, 7/31 (23%) had serous retinal detachment or panuveitis resembling Vogt-Koyanagi-Harada syndrome, 4/31 (13%) had papillitis, and 6/31 (19%) had diplopia or ocular motility defect. There was one case each (3.2%) of melanoma associated retinopathy, corneal edema, granulomatous lacrimal gland enlargement, and choroidal neovascularization. CONCLUSIONS: Ocular inflammation is a rare immune associated side effect of CPI treatment, the most common manifestation of which is anterior uveitis.


Subject(s)
Uveitis, Anterior , Uveitis , Uveomeningoencephalitic Syndrome , Humans , Immune Checkpoint Inhibitors/adverse effects , Retrospective Studies , Uveomeningoencephalitic Syndrome/diagnosis , Vision Disorders/drug therapy , Uveitis, Anterior/drug therapy , Acute Disease , Inflammation/drug therapy , Uveitis/drug therapy
20.
J Dermatol ; 51(6): 854-857, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38111371

ABSTRACT

The combination of BRAF kinase inhibitors (BRAFis) and MEK kinase inhibitors (MEKis) is one of the most promising chemotherapy regimens in the treatment of BRAF-mutant melanoma. Although BRAFi plus MEKi combined therapy is widely used for the treatment of BRAFV600-mutated melanoma, the incidence of uveitis caused by BRAFi plus MEKi is limited. In this report, we described five cases (two men and three women) of Vogt-Koyanagi-Harada (VKH) disease-like uveitis in melanoma patients who received BRAFi plus MEKi combined therapy. Of note, all the patients had the HLA-DRB1*04 haplotype, which is frequently detected in VKH-like non-infectious uveitis. On the other hand, among BRAFi plus MEKi-treated patients who did not develop VKH disease-like uveitis, only one of five (20%) patients had the HLA-DRB1*04 haplotype. Collectively, BRAFi/MEKi might induce severe VKH disease-like uveitis in melanoma patients with the HLA-DRB1*04 haplotype.


Subject(s)
HLA-DRB1 Chains , Melanoma , Protein Kinase Inhibitors , Proto-Oncogene Proteins B-raf , Uveomeningoencephalitic Syndrome , Humans , HLA-DRB1 Chains/genetics , Melanoma/drug therapy , Melanoma/genetics , Male , Uveomeningoencephalitic Syndrome/chemically induced , Uveomeningoencephalitic Syndrome/diagnosis , Uveomeningoencephalitic Syndrome/genetics , Female , Proto-Oncogene Proteins B-raf/antagonists & inhibitors , Proto-Oncogene Proteins B-raf/genetics , Middle Aged , Protein Kinase Inhibitors/adverse effects , Skin Neoplasms/drug therapy , Skin Neoplasms/genetics , Aged , Antineoplastic Combined Chemotherapy Protocols/adverse effects , Adult , Sulfonamides/adverse effects , Sulfonamides/administration & dosage , Vemurafenib/adverse effects , Vemurafenib/administration & dosage , Uveitis/chemically induced , Uveitis/diagnosis , Uveitis/genetics , Haplotypes
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