Antônio Pedro de Britto Pereira Fortuna, Laura Gomes Nunes de Melo, Ricardo Lima de Almeida Neves, Raul Nunes Galvarro Vianna
{"title":"Active Juxtapapillary Toxoplasmic Retinochoroiditis Presenting with Henle Fiber Hemorrhages.","authors":"Antônio Pedro de Britto Pereira Fortuna, Laura Gomes Nunes de Melo, Ricardo Lima de Almeida Neves, Raul Nunes Galvarro Vianna","doi":"10.1080/09273948.2025.2493358","DOIUrl":"https://doi.org/10.1080/09273948.2025.2493358","url":null,"abstract":"<p><p>To report an atypical presentation of ocular toxoplasmosis manifesting as juxtapapillary toxoplasmic retinochoroiditis with Henle fiber layer (HFL) hemorrhages. Observational case report. A 56-year-old HIV-positive female from the southeast region of Brazil presented with unilateral juxtapapillary toxoplasmic retinochoroiditis and HFL hemorrhages at fundus examination. Multimodal imaging systems, such as optical coherence tomography (OCT), were used for diagnosis and monitoring. The patient had a satisfactory clinical response to medical treatment, with ocular toxoplasmosis being the main etiological diagnosis based on clinical data, imaging, and laboratory. HFL hemorrhages represent, as far as the author's knowledge goes, a novel finding in ocular toxoplasmosis, adding to the understanding of HFL hemorrhages, specifically in their pathophysiology and etiologies.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-4"},"PeriodicalIF":2.6,"publicationDate":"2025-04-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144064316","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}
Laura Daniela Rodríguez-Camelo, María Triana-Cavelier, Laura Inés Rojas-Serrano, Germán Mejía-Salgado, Camilo Andrés Gómez-Durán, Juanita Cardona-López, Claudia Eugenia Durán-Merino, Alejandra de-la-Torre
{"title":"Atypical Presentation of Frosted Branch Angiitis due to Ocular Toxoplasmosis: A Case Report.","authors":"Laura Daniela Rodríguez-Camelo, María Triana-Cavelier, Laura Inés Rojas-Serrano, Germán Mejía-Salgado, Camilo Andrés Gómez-Durán, Juanita Cardona-López, Claudia Eugenia Durán-Merino, Alejandra de-la-Torre","doi":"10.1080/09273948.2025.2493791","DOIUrl":"https://doi.org/10.1080/09273948.2025.2493791","url":null,"abstract":"<p><strong>Purpose: </strong>To report a case of Frosted Branch Angiitis (FBA) secondary to ocular toxoplasmosis in a patient with a retinochoroidal scar in the contralateral eye (OD) and a late active retinochoroiditis in the OS and to compare it with previous reports.</p><p><strong>Case description: </strong>A 25-year-old female experienced sudden vision loss and eye redness in the left eye (OS). During the initial examination, the best-corrected visual acuity was 20/20 in the right eye (OD) and counting fingers in the OS. Ophthalmological examination of the OD showed a 1-disc diameter scar in the lower nasal mid-periphery. At the same time, OS revealed 2+ cellularity in the anterior chamber and retinal vasculitis with perivascular sheathing, indicative of FBA. Initial treatment included antiviral and corticosteroid therapy (oral and topical), with partial improvement. Aqueous humor polymerase chain reaction confirmed toxoplasmosis as the etiology, necessitating a shift to specific anti-Toxoplasma therapy, which led to clinical improvement.</p><p><strong>Conclusion: </strong>This case highlights the importance of considering in the differential diagnosis of FBA, even in patients with retinochoroidal scars in the contralateral eye. Close follow-up is essential after FBA resolution, as new lesions may develop. Prompt anti- therapy and corticosteroid treatment are critical for preserving visual outcomes, as delays in diagnosis and treatment can lead to significant complications.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-7"},"PeriodicalIF":2.6,"publicationDate":"2025-04-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144036914","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}
Charlotte Lussier, Cynthia L Larche, Julie Vadboncoeur
{"title":"Successful Treatment of Ocular Post-Transplant Lymphoproliferative Disorder with Obinutuzumab in an 8-Year-Old Boy Following Kidney Transplant: A Case Report.","authors":"Charlotte Lussier, Cynthia L Larche, Julie Vadboncoeur","doi":"10.1080/09273948.2025.2495070","DOIUrl":"https://doi.org/10.1080/09273948.2025.2495070","url":null,"abstract":"<p><strong>Purpose: </strong>To report a case of an eight-year-old boy who developed an intraocular recurrence of lymphoproliferative disorder following a kidney transplant.</p><p><strong>Methods: </strong>Retrospective single case report.</p><p><strong>Results: </strong>The patient initially presented with systemic post-transplant lymphoproliferative disorder (PTLD), which later recurred as a masquerade syndrome with iris nodules and granulomatous uveitis. Initial treatment with systemic rituximab led to recurrence after 6 months. Complete recurrence resolution of both ocular and systemic disease was achieved with obinutuzumab.</p><p><strong>Conclusion: </strong>This case highlights the importance of early recognition and multidisciplinary management in PTLD. It also emphasizes the delicate balance between immunosuppression and antitumor therapy in transplant recipients, aiming to preserve graft function while effectively treating PTLD.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-6"},"PeriodicalIF":2.6,"publicationDate":"2025-04-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143991345","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}
Manu Sharma, Atul Arora, Shilpa Viswanath, Ankur Jindal, Amit Rawat, Deepti Suri, Surjit Singh, Vishali Gupta
{"title":"Retinal Vasculitis in Familial Blau Syndrome.","authors":"Manu Sharma, Atul Arora, Shilpa Viswanath, Ankur Jindal, Amit Rawat, Deepti Suri, Surjit Singh, Vishali Gupta","doi":"10.1080/09273948.2025.2495063","DOIUrl":"https://doi.org/10.1080/09273948.2025.2495063","url":null,"abstract":"<p><strong>Introduction: </strong>Blau syndrome is a rare autosomal dominant condition resulting from mutations in the Nucleotide Oligomerization Domain of 2 ;(NOD2) gene. The disease typically presents in early childhood with a triad of arthritis, dermatitis, and uveitis. In this report, we describe retinal vasculitis as a rare phenotype in a family affected by Blau syndrome.</p><p><strong>Methods: </strong>Retrospective case series of 3 family members with Blau Syndrome presenting with bilateral retinal vasculitis. The clinical presentation, systemic features, and imaging findings were reviewed.</p><p><strong>Results: </strong>Case I, a 3-year-old boy with childhood-onset of arthritis and uveitis. Ocular manifestations were anterior segment inflammation (2 + cells), keratic precipitates, posterior synechiae, complicated cataract, perivascular choroiditis scars, and diffuse small vascular leakage on fundus fluorescein angiography (FFA). Case II, cousin of Case I, was a 24-years-old female with childhood-onset arthritis and uveitis. Ocular features were bilateral panuveitis and small vessel leakage on FFA. Case III, sibling of case II had late onset of uveitis at 28 years of age. On examination, he had pigment on the anterior lens surface, vitritis, and small vessel vasculitis in both eyes. However, he did not have any history of arthritis or dermatitis. Bilateral small vessel vasculitis was a common feature observed in all the three cases. Case II and III were paternal cousins of Case I. All patients had a pathogenic variant in NOD2 gene (NOD2: g.18773C>T, c.C>T1000, p.R334W) and were diagnosed with familial Blau syndrome.</p><p><strong>Conclusions: </strong>We report retinal vasculitis as a rare phenotype of Blau syndrome.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-7"},"PeriodicalIF":2.6,"publicationDate":"2025-04-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144013030","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}
Ikhwanuliman Putera, Rina La Distia Nora, Josianne C E M Ten Berge, Nicole M A Nagtzaam, Sigrid M A Swagemakers, Saskia M Rombach, P Martin van Hagen, Willem A Dik
{"title":"Diagnostic Biomarkers for Uveitis: Serum BAFF and CXCL9 in Differentiating Ocular Sarcoidosis, Tuberculosis and Other Entities with Implication for QuantiFERON-Positive Uveitis.","authors":"Ikhwanuliman Putera, Rina La Distia Nora, Josianne C E M Ten Berge, Nicole M A Nagtzaam, Sigrid M A Swagemakers, Saskia M Rombach, P Martin van Hagen, Willem A Dik","doi":"10.1080/09273948.2025.2493357","DOIUrl":"https://doi.org/10.1080/09273948.2025.2493357","url":null,"abstract":"<p><strong>Purpose: </strong>To evaluate serum B cell activating factor (BAFF) and CXCL9 as diagnostic biomarkers for ocular sarcoidosis and ocular tuberculosis (TB). We also explore their role in the stratification of patients with QuantiFERON (QFT)-positive uveitis without another identifiable cause.</p><p><strong>Methods: </strong>Eighty uveitis patients with various entities were included: 10 with ocular sarcoidosis, 35 with TB-related uveitis (including 11 confirmed (ocular TB) and 24 QFT-positive uveitis of unknown cause) and 35 with other uveitis entities. Levels of serum BAFF and CXCL9 were measured.</p><p><strong>Results: </strong>Serum BAFF was higher in ocular sarcoidosis compared to other groups (area under the curve (AUC) = 0.74; 95% CI: 0.53-0.96, <i>p</i> = 0.017). At an optimal serum BAFF cut-off point of 772.3 pg/ml, the sensitivity was 70.0% (95% CI: 39.7-89.2) and the specificity was 89.1% (95% CI: 77.0-95.3). Serum CXCL9 was comparable between ocular sarcoidosis and confirmed ocular TB but was significantly higher in these groups compared to other uveitis entities (AUC = 0.71, 95% CI: 0.57-0.86, <i>p</i> = 0.011). To differentiate ocular sarcoidosis and TB from other uveitis entities, CXCL9 (cutoff: 105.5 pg/ml) showed a sensitivity of 88.9% (95% CI: 67.2-98.0) and a specificity of 54.3% (95% CI: 38.2-69.5). High CXCL9 TB-related uveitis patients demonstrated a higher proportion of uveitis resolution when fully treated with antitubercular treatment (ATT) (<i>p</i> = 0.027).</p><p><strong>Conclusions: </strong>Serum BAFF and CXCL9 serve as potential diagnostic biomarkers for differentiating ocular sarcoidosis, ocular TB and other uveitis entities. These might identify QFT-positive uveitis who are most in need of ATT.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-13"},"PeriodicalIF":2.6,"publicationDate":"2025-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143972982","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}
Sara Touhami, Yoo-Ri Chung, Francesc March, Victoria Konikowski, Bahram Bodaghi, Maria Vittoria Cicinelli
{"title":"Atypical Mid-Late Phase ICGA Hyperfluorescence in a Secondary MEWDS Case: A Distinct Disease or a Shift in Our Understanding of MEWDS?","authors":"Sara Touhami, Yoo-Ri Chung, Francesc March, Victoria Konikowski, Bahram Bodaghi, Maria Vittoria Cicinelli","doi":"10.1080/09273948.2025.2493363","DOIUrl":"https://doi.org/10.1080/09273948.2025.2493363","url":null,"abstract":"<p><strong>Purpose: </strong>To report a secondary multiple evanescent white dot syndrome (MEWDS) case with peculiar indocyanine green angiography (ICGA) findings.</p><p><strong>Method: </strong>Report of a patient with sickle cell disease (SCD) and a longstanding macular hole who developed an atypical form of secondary MEWDS. Analysis of multimodal imaging findings was performed.</p><p><strong>Results: </strong>A 33-year-old female with SCD presented with a longstanding full-thickness macular hole in the left eye, but then was lost to follow-up. Upon her return nine months later, fundus examination revealed new, asymptomatic, yellowish lesions at the level of the outer retina or retinal pigment epithelium (RPE). ICGA showed a peculiar hyperfluorescence in mid-to-late phases, while fundus autofluorescence (FAF) highlighted distinct temporal patterns of hyper-autofluorescence. Some of the ICGA hyperfluorescent lesions displayed a central hypofluorescent core. The multimodal imaging findings suggest a distinct disease or a sequential mechanism in MEWDS pathophysiology. The process may begin with an initial photoreceptoritis, marked by primary self-resolving hyper-autofluorescence on FAF, followed by an early RPE dysfunction with choroidal hyperpermeability evidenced by mid-late phase ICGA hyperfluorescence, then more marked RPE dysfunction shown by late-phase ICGA hypofluorescence and FAF hyper-autofluorescence.</p><p><strong>Conclusion: </strong>This case describes an unusual secondary MEWDS presentation with unique imaging findings. The peculiar ICGA behavior and evolving FAF patterns may suggest either a distinct disease or a shift in our understanding of MEWDS involving photoreceptoritis, early RPE dysfunction, followed by more advanced RPE damage. The findings underscore the need for a nuanced approach to interpreting white-dot syndromes.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-4"},"PeriodicalIF":2.6,"publicationDate":"2025-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144022802","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}
Ahmed M Abu El-Asrar, Nele Berghmans, Saleh A Al-Obeidan, Priscilla Gikandi, Ghislain Opdenakker, Jo Van Damme, Sofie Struyf
{"title":"Pentraxin 3 is a Novel Inflammatory Marker of HLA-B27-associated Uveitis.","authors":"Ahmed M Abu El-Asrar, Nele Berghmans, Saleh A Al-Obeidan, Priscilla Gikandi, Ghislain Opdenakker, Jo Van Damme, Sofie Struyf","doi":"10.1080/09273948.2025.2491565","DOIUrl":"https://doi.org/10.1080/09273948.2025.2491565","url":null,"abstract":"<p><strong>Purpose: </strong>Evidence exists that pentraxin 3 (PTX3) is involved in the pathogenesis of autoimmune diseases. The aim of this study was to analyze the levels of PTX3 in aqueous humor (AH) samples from patients with specific autoimmune uveitic entities. In addition, we correlated PTX3 levels with the levels of proinflammatory cytokines and chemokines and the clinical disease activity.</p><p><strong>Methods: </strong>AH samples from patients with active uveitis associated with human leukocyte antigen (HLA)-B27-related inflammation (<i>n</i> = 12), Behçet's disease (BD) (<i>n</i> = 13), Vogt-Koyanagi-Harada (VKH) disease (<i>n</i> = 12), sarcoidosis (<i>n</i> = 8) and control subjects (<i>n</i> = 9) were measured with the use of multiplex assays.</p><p><strong>Results: </strong>When considering all uveitis patients as one group (<i>n</i> = 45), PTX3 levels were significantly increased compared to controls (<i>p</i> = 0.002). When comparing the four individual disease groups to controls, PTX3 levels were significantly higher in only HLA-B27-associated uveitis (<i>p</i> < 0.001). PTX3 levels were significantly higher in patients with HLA-B27-associated uveitis than in patients with BD (<i>p</i> = 0.034) and VKH disease (<i>p</i> < 0.001). PTX3 levels had a significant correlation with clinical disease activity (<i>r</i> = 0.480; <i>p</i> = 0.001) and strong positive correlations with aqueous humor levels of interferon-γ (<i>r</i> = 0.749; <i>p</i> < 0.001), interleukin (IL)-1ß (<i>r</i> = 0.804; <i>p</i> < 0.001), IL-6 (<i>r</i> = 707; <i>p</i> < 0.001), CCL20 (<i>r</i> = 0.691; <i>p</i> < 0.001) and CXCL8 (<i>r</i> = 0.775; <i>p</i> < 0.001).</p><p><strong>Conclusions: </strong>PTX3 is an inflammatory marker and might exert a pathogenic role in HLA-B27-associated uveitis.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-7"},"PeriodicalIF":2.6,"publicationDate":"2025-04-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143974964","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}
Sapna S Gangaputra, Craig W Newcomb, Gui-Shuang Ying, Kurt A Dreger, Tonetta Fitzgerald, Pichaporn Artornsombudh, Srishti Kothari, Teresa L Liesegang, James T Rosenbaum, H Nida Sen, Eric B Suhler, Jennifer E Thorne, Nirali P Bhatt, C Stephen Foster, Douglas A Jabs, Grace A Levy-Clarke, Careen Y Lowder, Debra A Goldstein, Gary N Holland, Glenn J Jaffe, Janet L Davis, Nisha R Acharya, Paul A Latkany, Russell W Read, Sunir J Garg, Yannek I Liederman, Jeanine M Buchanich, John H Kempen, For The Systemic Immunosuppressive Therapy For Eye Diseases Site Cohort Study Research Group
{"title":"Effectiveness of Frequently Used TNF Inhibitors vs. Conventional Immunosuppressive Therapies for Noninfectious Uveitis.","authors":"Sapna S Gangaputra, Craig W Newcomb, Gui-Shuang Ying, Kurt A Dreger, Tonetta Fitzgerald, Pichaporn Artornsombudh, Srishti Kothari, Teresa L Liesegang, James T Rosenbaum, H Nida Sen, Eric B Suhler, Jennifer E Thorne, Nirali P Bhatt, C Stephen Foster, Douglas A Jabs, Grace A Levy-Clarke, Careen Y Lowder, Debra A Goldstein, Gary N Holland, Glenn J Jaffe, Janet L Davis, Nisha R Acharya, Paul A Latkany, Russell W Read, Sunir J Garg, Yannek I Liederman, Jeanine M Buchanich, John H Kempen, For The Systemic Immunosuppressive Therapy For Eye Diseases Site Cohort Study Research Group","doi":"10.1080/09273948.2025.2479802","DOIUrl":"https://doi.org/10.1080/09273948.2025.2479802","url":null,"abstract":"<p><strong>Purpose: </strong>To compare the corticosteroid sparing efficacy of frequently used antimetabolites to tumor necrosis factor (TNF) inhibitors in the management of noninfectious ocular inflammation.</p><p><strong>Methods: </strong>Retrospective analysis of patients with noninfectious uveitis on conventional antimetabolite (methotrexate, mycophenolate mofetil, azathioprine,or leflunomide, \"CONV\") or a TNF inhibitor (adalimumab or infliximab, \"TNFi\") with active inflammation or more than 7.5 mg daily prednisone. Eyes were assessed in three groups: CONV only, TNFi only and combination of both (COMB). Cox regression models compared treatment success, adjusted for age, race, smoking, anatomic location of uveitis, duration of uveitis and visual acuity. Corticosteroid sparing success was defined as: inactive or slightly active uveitis on <=7.5 mg daily oral prednisone and <=2 drops of prednisolone acetate 1%.</p><p><strong>Results: </strong>There were 1475 eligible patients in the analysis. By 6 and 12 months, respectively, the Cox model-predicted, percentage success was 27.6% and 44.9% for the CONV group; 34.2% and 53.9% in the TNFi group and 39.9% and 61.1% for the COMB group. COMB was more likely than CONV to achieve success (adjusted HR 1.58 (95% confidence interval (CI), 1.28, 1.95, <i>p</i> < 0.0001). Factors associated with lower success were age under 18 years, smoking, visual acuity worse than 20/50 at cohort entry, over 4-year duration of uveitis and daily baseline prednisone 7.5 mg or higher (all <i>p</i> < 0.05).</p><p><strong>Conclusion: </strong>Our results suggest COMB is more effective than CONV at achieving disease quiescence and corticosteroid sparing success among patients with active noninfectious uveitis. More research is needed to determine if TNFi alone is superior to CONV for uveitic corticosteroid-sparing.</p>","PeriodicalId":19406,"journal":{"name":"Ocular Immunology and Inflammation","volume":" ","pages":"1-9"},"PeriodicalIF":2.6,"publicationDate":"2025-04-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144015128","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}