{"title":"Type 2 Uveal Effusion Syndrome with Psoriasis Vulgaris: A Case Report.","authors":"Chen Zhou, Yunfeng Lu, Lingying Ge, Chenglang Xin, Xue Yin","doi":"10.1080/09273948.2025.2525493","DOIUrl":"https://doi.org/10.1080/09273948.2025.2525493","url":null,"abstract":"<p><strong>Purpose: </strong>To report the case of uveal effusion syndrome (UES) in a 35-year-old woman with psoriasis vulgaris, emphasizing scleral pathology and systemic inflammatory contributions mediated by proteomic alterations.</p><p><strong>Methods: </strong>Medical history, clinical findings and treatment were reviewed, along with histopathological and proteomic analysis.</p><p><strong>Results: </strong>A 35-year-old woman presented with retinal and choroidal detachments in the left eye and scleral thickening (3 mm vs. normal 1 mm). Concurrently, her psoriasis symptoms worsened. Histology revealed collagen disorganization and extracellular matrix remodeling in the sclera, accompanied by elevated inflammatory factors (e.g. MCP-4, RANTES, Fractalkine). Treatment included partial sclerotomy, leading to resolution of detachments and improved visual acuity.</p><p><strong>Conclusion: </strong>Systemic inflammation associated with psoriasis may contribute to UES pathogenesis. Dermatologists should refer psoriasis patients for ophthalmologic examinations, while ophthalmologists should be aware of underlying health conditions when treating UES.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-4"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144541654","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pichaporn Artornsombudh, Maxwell Pistilli, Craig W Newcomb, C Stephen Foster, Douglas A Jabs, Jennifer E Thorne, Nirali P Bhatt, James T Rosenbaum, Grace A Levy-Clarke, H Nida Sen, Eric B Suhler, Kurt A Dreger, Jeanine M Buchanich, Hosne Begum, Tonetta D Fitzgerald, Naira Khachatryan, Teresa L Liesegang, Gui-Shuang Ying, Sapna S Gangaputra, John H Kempen, Site Cohort Study Research Group
{"title":"Incidence and Outcomes of Cataract in Eyes with Ocular Cicatricial Pemphigoid.","authors":"Pichaporn Artornsombudh, Maxwell Pistilli, Craig W Newcomb, C Stephen Foster, Douglas A Jabs, Jennifer E Thorne, Nirali P Bhatt, James T Rosenbaum, Grace A Levy-Clarke, H Nida Sen, Eric B Suhler, Kurt A Dreger, Jeanine M Buchanich, Hosne Begum, Tonetta D Fitzgerald, Naira Khachatryan, Teresa L Liesegang, Gui-Shuang Ying, Sapna S Gangaputra, John H Kempen, Site Cohort Study Research Group","doi":"10.1080/09273948.2025.2450471","DOIUrl":"10.1080/09273948.2025.2450471","url":null,"abstract":"<p><strong>Purpose: </strong>To identify the incidence of cataract and the outcomes of cataract surgery in eyes with ocular cicatricial pemphigoid (OCP).</p><p><strong>Methods: </strong>Phakic eyes were identified from the Systemic Immunosuppressive Therapy for Eye Diseases Cohort Study and followed for the incidence of visually significant cataract defined as: newly reduced visual acuity 20/50 or worse attributed to cataract; and/or incident cataract surgery. Secondarily, all eyes with OCP that underwent cataract surgery and had a year of follow up thereafter, were included in an analysis of visual outcome.</p><p><strong>Results: </strong>Three hundred fifty-five phakic eyes (200 patients) with OCP were at risk. Eighty eyes developed visually significant cataract over 1064 eye years (incidence rate = 7.5%/eye-year, 95% confidence interval [CI] = 5.6 to 10.1). Higher age was associated with increased incidence of cataract (adjusted hazard ratio [aHR] = 4.47; 95% CI, 1.95-10.23 for age 60-75 inclusive and aHR = 8.37; 95% CI, 3.60-19.42 for age > 75, each compared with age <60 years). Seventy-nine eyes of 61 patients were monitored for > = 1 year following cataract surgery. Cataract surgery was associated with an improvement of vision around 4 lines, which was sustained through at least 48 months. Poorer pre-operative visual acuity was associated with poorer long-term visual outcome.</p><p><strong>Conclusions: </strong>The incidence of cataract was high in this older population. No factors predictive of cataract such as duration of OCP or use of corticosteroids were identified. Visual acuity improved after surgery by a median of 4 lines' gain at one year; poorer long-term outcome among those with initially poorer visual acuity may be secondary to corneal scarring.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"791-799"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12187555/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143256208","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Half a Century's Quest for Seasonal Hyperacute Panuveitis (SHAPU): From Academic Curiosity to Public Health Concern.","authors":"Yun Yao Goh, Ranju Kharel Sitaula, William Rojas-Carabali, Rupesh Agrawal, Pratap Karki, Haramaya Gurung, Eliya Shrestha, Pursottam Joshi, Anadi Khatri, Ananda Kumar Sharma, Sagun Narayan Joshi, Daya Ram Bhusal, Madan Prasad Upadhyay","doi":"10.1080/09273948.2024.2444510","DOIUrl":"10.1080/09273948.2024.2444510","url":null,"abstract":"<p><p>For the past 50 years, significant progress has been made in understanding Seasonal Hyperacute Panuveitis (SHAPU), a mysterious blinding disease first reported in Nepal in 1975. Predominantly affecting Nepalese children, SHAPU occurs cyclically every odd year from September to December. While initially misclassified as endophthalmitis, SHAPU is set apart by its lack of trauma or surgery, failure to grow organisms in most intraocular fluid cultures, and its hallmark presentation of a \"white pupil in red eye\" with an association with moth exposure. Recent findings have expanded SHAPU's geographical scope beyond Nepal, with cases reported in Bhutan. Moreover, sporadic summer outbreaks have emerged, showing worse clinical outcomes than the typical cases. Recent studies suggest that the <i>Gazalina</i> moth may play a role, although the exact cause of SHAPU remains undetermined. Despite the increased understanding of SHAPU's geographic distribution and cyclical patterns, it is possible that some cases remain misdiagnosed or underreported. Continued research is critical to further exploring the aetiology of SHAPU, which will lead to better diagnostic tools, targeted treatments, and improved outcomes. The inclusion of SHAPU under more general categories such as endophthalmitis underscores the need for a clearer distinction of this condition to prevent blindness in at-risk populations.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"819-826"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142971791","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Aniruddha Agarwal, Sahar Al Ali, Asia Mubashir, Mahdi Shkoukani, Matteo Belletti, Mohammed Saeed, Hassan Alnuaimat, Asim Malik
{"title":"Serpiginous-Like Choroiditis with ANCA-PR3 Vasculitis in a Patient from Endemic Country: A Complex Phenotypic, Immunological and Epidemiological Puzzle.","authors":"Aniruddha Agarwal, Sahar Al Ali, Asia Mubashir, Mahdi Shkoukani, Matteo Belletti, Mohammed Saeed, Hassan Alnuaimat, Asim Malik","doi":"10.1080/09273948.2024.2440566","DOIUrl":"10.1080/09273948.2024.2440566","url":null,"abstract":"<p><strong>Purpose: </strong>To report a complex case of serpiginous-like choroiditis (SLC) in a patient with anti-neutrophil cytoplasmic antibody (ANCA)-anti-proteinase 3 (PR3)-associated vasculitis with systemic involvement.</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 40-year-old male from a tuberculosis (TB)-endemic region presented with bilateral active SLC lesions. He was diagnosed with ANCA-PR3 vasculitis with unilateral otitis media, nasopharyngeal mass, lung abscess, pleurisy, and joint pain, all of which responded well to corticosteroids and rituximab. Extensive evaluations and biopsies ruled out Mycobacterium tuberculosis infection. Despite consensus criteria recommending anti-tubercular therapy (ATT), the SLC lesions were effectively managed with an intravitreal dexamethasone implant and systemic azathioprine, showing no recurrence at 18 months without ATT.</p><p><strong>Conclusions: </strong>The development of SLC lesions in the context of systemic autoimmune conditions like ANCA-PR3 vasculitis is uncommon. In these cases, the need for ATT should be carefully evaluated, with close attention to systemic disease manifestations and tailored management strategies.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"836-840"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142801821","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Fabio Daniel Padilla-Pantoja, Fernando Godin, Fernando Rojas-Rojas, Jennifer Camargo-González, David Ancona-Lezama
{"title":"Bilateral Choroidal Metastases from Primary Cutaneous Melanoma Following Nivolumab Discontinuation: A Case Report.","authors":"Fabio Daniel Padilla-Pantoja, Fernando Godin, Fernando Rojas-Rojas, Jennifer Camargo-González, David Ancona-Lezama","doi":"10.1080/09273948.2025.2456646","DOIUrl":"10.1080/09273948.2025.2456646","url":null,"abstract":"<p><strong>Purpose: </strong>To present the case of a young patient with BRAF V600E-mutant cutaneous melanoma who developed bilateral choroidal metastases complicated by neovascular glaucoma (NVG) in both eyes following the interruption of nivolumab therapy.</p><p><strong>Methods: </strong>A 28-year-old female with primary cutaneous melanoma of the left hand underwent surgical resection and adjuvant nivolumab. Immunotherapy was discontinued due to immune-related acute interstitial nephritis. Three months after cessation, she presented with bilateral decreased vision, pain, and redness.</p><p><strong>Results: </strong>Ophthalmic examination and imaging revealed bilateral choroidal thickening with focal nodular lesions consistent with metastatic disease, along with uveitis and NVG in both eyes. This condition required topical anti-inflammatory treatment and cyclophotocoagulation for intraocular pressure (IOP) control. Nivolumab was reinitiated in combination with systemic corticosteroids to manage immune-related toxicity. At the 12-month follow-up, the patient retained visual function in one eye, with no signs of new metastatic lesions.</p><p><strong>Conclusions: </strong>Bilateral choroidal metastases from cutaneous melanoma are rare and can be complicated by NVG. Interruption of immune checkpoint inhibitors (ICIs) like nivolumab may lead to metastatic recurrence. Prompt resumption of immunotherapy, coupled with appropriate ocular interventions, is crucial for controlling disease progression and preserving vision. Careful management of immune-related adverse effects (irAEs) is essential to prevent treatment discontinuation and subsequent disease relapse.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"864-867"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143040143","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Sunset Glow Fundus and Bilateral Choroidal Neovascularization in a Pediatric Patient Receiving Nivolumab: A Rare Pediatric Immune-Related Ocular Complication.","authors":"Izlem Ozturan, Sibel Kadayifcilar, Figen Bezci Aygun, Tezer Kutluk","doi":"10.1080/09273948.2025.2524584","DOIUrl":"https://doi.org/10.1080/09273948.2025.2524584","url":null,"abstract":"<p><strong>Purpose: </strong>To report a rare case of immune checkpoint inhibitor (ICI)- associated bilateral choroidal neovascularization (CNV) and sunset glow fundus appearance in a pediatric patient receiving long-term nivolumab therapy.</p><p><strong>Method: </strong>Observational case report.</p><p><strong>Case report: </strong>A 15-year-old male with metastatic myoepithelial carcinoma, on nivolumab for 2.5 years, presented with bilateral blurred vision. Fundus examination revealed a sunset glow fundus, punched-out lesions, and CNV in both eyes. Optical coherence tomography (OCT) confirmed inflammatory CNV with choroidal thickening. Nivolumab was discontinued, and the patient received systemic corticosteroids and intravitreal ranibizumab. Despite initial improvement, CNV recurred upon steroid tapering, requiring additional anti-VEGF injections. At four months, vision stabilized with no CNV reactivation.</p><p><strong>Conclusion: </strong>This case represents the first pediatric report of ICI- associated bilateral CNV and sunset glow fundus appearance. It underscores the importance of monitoring for late-onset immune-related ocular complications in patients on long-term nivolumab therapy.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-4"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144541651","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Laurie Mourozeau, Virginie Pichon, Aymane Bouzidi, Majida Charif, Marine Tessarech, Angélique Caignard, Patrizia Amati-Bonneau, Agnès Guichet, Christian Lavigne, Christophe Verny, Philippe Gohier, Guy Lenaers
{"title":"Two Cases of ROSAH-Like Syndrome Restricted to the Ophthalmologic Presentation.","authors":"Laurie Mourozeau, Virginie Pichon, Aymane Bouzidi, Majida Charif, Marine Tessarech, Angélique Caignard, Patrizia Amati-Bonneau, Agnès Guichet, Christian Lavigne, Christophe Verny, Philippe Gohier, Guy Lenaers","doi":"10.1080/09273948.2025.2453873","DOIUrl":"10.1080/09273948.2025.2453873","url":null,"abstract":"<p><strong>Purpose: </strong>To report the clinical presentation and follow-up, including the optical coherence tomography, angiography and electrophysiology of two individuals from the same family presenting with an isolated retinal dystrophy and optic nerve edema who were diagnosed with ROSAH-like syndrome.</p><p><strong>Method: </strong>Observational case report of a 55-year-old woman and her 36-year-old son with a genetic analysis of ROSAH, after a long-term follow-up.</p><p><strong>Results: </strong>Both the mother and her son displayed severe optic nerve infiltration and retinal pigment atrophy with intraocular inflammation, which were not improved by immunosuppressive treatment. Systemic investigations were not relevant of a syndromic presentation. Whole exome sequencing revealed the same <i>ALPK1</i> missense pathogenic variant c.710C>T; p.(Thr237Met) responsible for ROSAH syndrome.</p><p><strong>Conclusion: </strong><i>ALPK1</i> variant responsible for ROSAH syndrome may cause severe retinal dystrophy and an uveitis-like presentation resistant to conventional immunosuppressive drugs, without the systemic symptoms common to the ROSAH syndrome.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"849-853"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143024164","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Matteo Belletti, Zaid Zoumot, Asia Mubashir, Maria-Fernanda Bonilla, Aniruddha Agarwal
{"title":"Diagnostic and Management Challenges in Interferon-γ Release Assay-Positive Patient with Sarcoid Panuveitis from a Non-Endemic Tuberculosis Region.","authors":"Matteo Belletti, Zaid Zoumot, Asia Mubashir, Maria-Fernanda Bonilla, Aniruddha Agarwal","doi":"10.1080/09273948.2025.2450484","DOIUrl":"https://doi.org/10.1080/09273948.2025.2450484","url":null,"abstract":"<p><strong>Purpose: </strong>To report a case of biopsy-proven sarcoidosis in a patient with panuveitis and a positive interferon-gamma release assay (IGRA) from a non-endemic tuberculosis (TB) country.</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 26-year-old male from the United Arab Emirates (UAE) presented with granulomatous panuveitis characterized by mutton-fat keratic precipitates, anterior chamber and vitreous cells, and retinal vasculitis. Laboratory evaluation revealed positive IGRA, normal serum angiotensin-converting enzyme level, and chest imaging showing lung nodularity along with hilar lymphadenopathy. Endobronchial ultrasound-transbronchial needle aspiration (EBUS-TBNA) identified non-caseating granulomas and an elevated CD4+/CD8+ ratio. Both vitreous biopsy and EBUS-TBNA tested negative for TB polymerase chain reaction, leading to a definitive diagnosis of sarcoidosis. The patient was successfully managed with systemic immunosuppression and achieved sustained quiescence.</p><p><strong>Discussion: </strong>Patients from non-endemic TB countries can present with a positive IGRA in the context of sarcoidosis. Invasive biopsy evaluation is crucial for establishing an accurate diagnosis and initiating appropriate treatment.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":"33 5","pages":"845-848"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144485292","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Multicenter Analysis of Acute Retinal Necrosis: Clinical Characteristics, Viral Pathogens, and Diagnostic Predictive Factors.","authors":"Panuwat Soitong, Yaninsiri Ngathaweesuk, Noppakhun Panyayingyong, Atchara Amphornphruet, Variya Nganthavee, Boonsub Sakboonyarat, Sukhum Silpa-Archa, Narumon Keorochana","doi":"10.1080/09273948.2025.2456642","DOIUrl":"10.1080/09273948.2025.2456642","url":null,"abstract":"<p><strong>Purpose: </strong>This multicenter study aimed to investigate the clinical characteristics and factors associated with specific viral pathogens in patients with acute retinal necrosis (ARN).</p><p><strong>Methods: </strong>A retrospective multicenter cohort study included ARN patients who underwent aqueous or vitreous polymerase chain reaction (PCR) testing. Multivariable mixed-effects Poisson regression was used to identify factors associated with viral pathogens.</p><p><strong>Results: </strong>A total of 56 patients (65 eyes) with ARN were included, with a mean age was 39.9 ± 23.2 years. Anterior chamber cell showed significant inflammation in 89.2%, while vitreous haze was present in 100%. Multifocal retinitis was found in 92.3% of cases. Among these, 49.2% had diffuse or confluent patterns, 15.4% were circumferential, and 23.1% showed satellite lesions. Additionally, 18.5% were ameboid or scalloped shape, while 9.2% had wedge shapes. Varicella zoster virus (VZV) was the most common pathogen (30.8%), followed by Herpes simplex virus (HSV) (13.8%), Cytomegalovirus (CMV) (10.8%), Epstein-Barr virus (EBV) (9.2%), and human herpesvirus (HHV) (3.0%). Co-infections were observed in 20% of eyes. CMV-related ARN was significantly associated with wedge-shaped retinitis (aRR 4.19) and immunocompromised host (aRR 8.8). Younger age and optic disc edema (aRR 5.41) were correlated with HSV-related ARN.</p><p><strong>Conclusion: </strong>VZV was the predominant viral pathogen, with the increasing prevalence of CMV, EBV, and co-infection. Wedge-shaped retinitis and immunocompromised status were notably associated with CMV-related ARN, findings that may guide antiviral therapy decisions in settings where PCR diagnostics are either unavailable or delayed, ultimately improving visual outcomes.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"721-728"},"PeriodicalIF":2.6,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143024163","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}