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International Ophthalmology Feb 2024To present the long-term clinical outcomes of patients with sympathetic ophthalmia (SO).
PURPOSE
To present the long-term clinical outcomes of patients with sympathetic ophthalmia (SO).
METHODS
Retrospective review of patients' medical files between 2002 and 2022.
RESULTS
Included were seven patients (four males). The mean ± SD age at presentation was 37.9 ± 22.5 years. Four patients had co-morbidities: three had diabetes mellitus type 2 and one had Turner Syndrome. Trauma was the inciting event in six patients and postoperative endophthalmitis in one patient. Decreased visual acuity (VA) was the leading symptom in the sympathizing eye and all of the patients presented with panuveitis. The mean ± SD interval between the triggering incident and the onset of SO in six cases was 4.3 ± 4.2 months. One case presented 30 years following the eye injury. Five patients underwent enucleation/evisceration of the exciting eye. The mean ± SD presenting LogMAR BCVA in the sympathizing eye was 0.57 ± 0.82, and the final LogMAR BCVA was 0.61 ± 0.95. Inflammation was completely controlled in 5 patients at a mean ± SD of 8.55 ± 9.21 months following the institution of immunomodulatory therapy, and it was partially controlled in 2 patients. VA deteriorated in all 3 diabetic patients and improved or remained stable in the 4 young and healthy patients. The mean ± SD follow-up period after achieving drug-free remission was 28 ± 22.8 months. The mean ± SD follow-up time was 6.8 ± 5.6 years.
CONCLUSIONS
SO is one of the most sight-threatening conditions, affecting the healthy eye. In this cohort, the favorable visual outcome was especially seen in young and healthy individuals. Visual prognosis is directly related to prompt diagnosis and treatment.
Topics: Male; Humans; Adolescent; Young Adult; Adult; Middle Aged; Ophthalmia, Sympathetic; Inflammation; Panuveitis; Diabetes Mellitus, Type 2; Endophthalmitis
PubMed: 38324101
DOI: 10.1007/s10792-024-03007-x -
Indian Journal of Ophthalmology Jun 2024To study cases of Fuchs' uveitis (FU) diagnosed as uveitis other than FU at initial visits. (Observational Study)
Observational Study
PURPOSE
To study cases of Fuchs' uveitis (FU) diagnosed as uveitis other than FU at initial visits.
METHODS
A retrospective, observational chart review. Details of clinical presentation, initial diagnosis, treatment details, and follow-up before and after the diagnosis of FU was studied.
RESULT
Twenty eyes of 19 patients finally diagnosed with FU. Mean total follow-up was 39 months (range: 3-120 months, median: 30 months) and follow-up after diagnosis of FU was 18 months (range: 0-84 months, median: 11 months). Most of the cases were diagnosed with anterior plus intermediate uveitis at the presentation (n = 8, 42.1%) and received steroid treatment. Keratic precipitates (KPs) typical of FU developed after mean 5.4 months of presentation (range: 1-18 months, median: 2 months). Altered iris pattern was noted after 17 months of presentation (range: 2-70 months, median: 8.5). Vitritis of grades 1-2 was present in all eyes. Overlap of uveitis of other etiology (retinal vasculitis, presumed tubercular intermediate uveitis) was found in three patients. Systemic immune disease (systemic lupus erythematosus, pulmonary sarcoidosis) was present in two patients. No treatment for the ocular inflammation was given after the diagnosis of FU in any patient and no change in the pattern of FU was observed during further follow-up.
CONCLUSION
Evolving FU may lead to misdiagnosis and mismanagement. We suggest a conservative approach, withholding steroid treatment, allowing FU to evolve, in otherwise asymptomatic patients with white eye, mild to moderate anterior chamber (AC) and vitreous inflammation, absent posterior synechiae and normal fundus examination or fundus examination suggestive of resolved uveitis.
Topics: Humans; Retrospective Studies; Male; Female; Adult; Middle Aged; Follow-Up Studies; Diagnosis, Differential; Aged; Young Adult; Visual Acuity; Adolescent; Uveitis, Anterior; Child; Fundus Oculi; Fluorescein Angiography
PubMed: 38317311
DOI: 10.4103/IJO.IJO_1151_23 -
Journal of Cardiothoracic Surgery Feb 2024Aortic involvement in patients with Behcet's disease (BD) is rare, but it is one of the most severe manifestations. Open surgical repair of aortic aneurysm is...
BACKGROUND
Aortic involvement in patients with Behcet's disease (BD) is rare, but it is one of the most severe manifestations. Open surgical repair of aortic aneurysm is challenging considering the high risk of postoperative recurrent anastomotic pseudoaneurysms and is associated with a much higher mortality rate. Recently, endovascular treatment has proven to be a feasible, less invasive alternative to surgery for these patients.
CASE PRESENTATION
We report a total endovascular repair of a paravisceral abdominal aortic pseudoaneurysm in a 25-year-old male patient with BD. The pseudoaneurysm was successfully excluded, and the blood supply of visceral arteries was preserved with a physician-modified three-fenestration endograft under 3D image fusion guidance. Immunosuppressive therapy was continued for 1 year postoperatively. At 18 months, the patient was asymptomatic without abdominal pain. Computed tomography angiography demonstrated the absence of pseudoaneurysm recurrence, good patency of visceral vessels.
DISCUSSION AND CONCLUSIONS
Endovascular repair using physician-modified fenestrated endografts is a relatively safe and effective approach for treating paravisceral aortic pseudoaneurysm in BD patients. This technique enables the preservation of the visceral arteries and prevents aneurysm recurrence at the proximal and distal landing zones, which are common complications of open surgical repair in these patients. Furthermore, we emphasize the importance of adequate immunosuppressive therapy before and after surgical repair in BD patients, which is a major risk factor for recurrence and poor prognosis.
Topics: Adult; Humans; Male; Aneurysm, False; Aortic Aneurysm, Abdominal; Behcet Syndrome; Endovascular Procedures; Stents; Treatment Outcome
PubMed: 38311787
DOI: 10.1186/s13019-024-02523-2 -
Clinical Therapeutics Mar 2024Behçet's disease (BD) is a complex disorder affecting multiple systems and organs, and gastrointestinal BD is poorly understood. We aimed to identify factors...
PURPOSE
Behçet's disease (BD) is a complex disorder affecting multiple systems and organs, and gastrointestinal BD is poorly understood. We aimed to identify factors influencing the long-term outcomes of patients with gastrointestinal BD.
METHODS
Consecutive patients with gastrointestinal BD were analyzed retrospectively. Data on the following clinical characteristics were collected: sex, age at diagnosis, symptoms, endoscopic findings, medical treatments, and surgery. Mucosal healing and surgical rates at 1, 2, and 5 years were evaluated. Log-rank test and Cox proportional hazards regression models were used to evaluate the factors affecting long-term outcomes.
FINDINGS
Baseline data of 175 patients with gastrointestinal BD were included. The mean (SD) age at diagnosis was 38.3 (12.9) years. The typical clinical symptoms were oral ulcer (72.6%), abdominal pain (71.4%), and weight loss (41.1%). The most commonly involved location was the ileocecum; isolated oval ulcer was the most common ulcer type. Seventeen patients (9.7%) underwent 18 surgeries after inclusion. The cumulative surgical rates were 8.6% (n/N = 15/175), 8.6% (n/N = 15/175), and 9.1% (n/N = 16/175) in 1, 2, and 5 years, respectively. Data from 101 patients who underwent at least 2 endoscopies were included in the analysis for mucosal healing. Kaplan-Meier curve showed that the cumulative mucosal healing rates at 1, 2, and 5 years were 34.7% (n/N = 35/101), 41.6% (n/N = 42/101), and 61.4% (n/N = 62/101), respectively. We compared cumulative mucosal healing rates between 4 treatment groups, including 5-aminosalicylic acid (3% [n/N = 3/101]), mono-immunosuppressant (31.7% [n/N = 32/101]), combined therapy (36.6% [n/N = 37/101]), and escalation therapy (28.7% [n/N = 29/101]), and found that mono-immunosuppressant achieved earlier mucosal healing than combined therapy (P = 0.0008) and escalation therapy (P = 0.0008). The univariate analysis showed that moderate to severe disease activity (P = 0.013, P = 0.004), diameter of the maximal ulcer >4 cm (P = 0.002), and nonsimple esophageal involvement (P < 0.001) were risk factors, and number of ulcers between 2 and 5 was the protective factor of mucosal healing (P = 0.001). Multivariate regression analysis indicated that nonsimple esophageal involvement (P < 0.001) and the maximal ulcer >4 cm (P = 0.041) were independent risk factors of mucosal healing.
IMPLICATIONS
Most patients with gastrointestinal BD need long-term treatment to achieve mucosal healing. The location and size of ulcers have a significant impact on the mucosal healing of gastrointestinal BD.
Topics: Humans; Adult; Ulcer; Retrospective Studies; Behcet Syndrome; Gastrointestinal Diseases; China
PubMed: 38296718
DOI: 10.1016/j.clinthera.2023.12.012 -
Frontiers in Immunology 2023Ocular abnormalities have been reported in association with viral infections, including Long COVID, a debilitating illness caused by the Severe Acute Respiratory...
Ocular abnormalities have been reported in association with viral infections, including Long COVID, a debilitating illness caused by the Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV-2). This report presents a case of a female patient diagnosed with Acute Macular Neuroretinopathy (AMN) following an Influenza A virus infection during Long COVID who experienced severe inflammation symptoms and ocular complications. We hypothesize that the rare occurrence of AMN in this patient could be associated with the immune storm secondary to the viral infection during Long COVID.
Topics: Humans; Female; SARS-CoV-2; COVID-19; Post-Acute COVID-19 Syndrome; Influenza A virus; Virus Diseases; White Dot Syndromes
PubMed: 38288123
DOI: 10.3389/fimmu.2023.1302504 -
Biomedicines Dec 2023The perception of circulating granulocytes as cells with a predetermined immune response mainly triggered by pathogens is evolving, recognizing their functional...
The perception of circulating granulocytes as cells with a predetermined immune response mainly triggered by pathogens is evolving, recognizing their functional heterogeneity and adaptability, particularly within the neutrophil subset. The involvement of these cells in the pathophysiology of autoimmune uveitis has become increasingly clear, yet their exact role remains elusive. We used an equine model for autoimmune-mediated recurrent pan-uveitis to investigate early responses of granulocytes in different inflammatory environments. For this purpose, we performed differential proteomics on granulocytes from healthy and diseased horses stimulated with IL8, LPS, or PMA. Compared to healthy horses, granulocytes from the recurrent uveitis model significantly changed the cellular abundance of 384 proteins, with a considerable number of specific changes for each stimulant. To gain more insight into the functional impact of these stimulant-specific proteome changes in ERU pathogenesis, we used Ingenuity Pathway Analysis for pathway enrichment. This resulted in specific reaction patterns for each stimulant, with IL8 predominantly promoting Class I MHC-mediated antigen processing and presentation, LPS enhancing processes in phospholipid biosynthesis, and PMA, clearly inducing neutrophil degranulation. These findings shed light on the remarkably differentiated responses of neutrophils, offering valuable insights into their functional heterogeneity in a T-cell-driven disease. Raw data are available via ProteomeXchange with identifier PXD013648.
PubMed: 38275380
DOI: 10.3390/biomedicines12010019 -
Frontiers in Immunology 2023Observational studies found associations between ankylosing spondylitis (AS) and iridocyclitis (IC), but the causality remained unconfirmed.
BACKGROUND
Observational studies found associations between ankylosing spondylitis (AS) and iridocyclitis (IC), but the causality remained unconfirmed.
METHODS
We employed two-sample Mendelian randomization (MR) to investigate the bidirectional causal relationships between AS and IC. Single-nucleotide polymorphisms (SNPs) were chosen from the FinnGen database's genome-wide association studies (GWAS) following a rigorous evaluation of the studies' quality. Sensitivity analysis was performed to assess the potential influence of pleiotropy and heterogeneity on the MR findings.
RESULTS
Elevated genetic risk for AS showed positive causal effects on IC and its subtypes (IC, OR = 1.094, 95% CI = 1.035-1.157, = 0.00156; Acute/Subacute IC, OR = 1.327, 95% CI = 1.266-1.392, = 8.73×10; Chronic IC, OR = 1.454, 95% CI = 1.308-1.618, = 5.19×10). Significant causal association was specifically observed between Acute/Subacute IC and AS (OR = 1.944, 95% CI = 1.316-2.873, = 8.38×10). Sensitivity analysis suggested that horizontal pleiotropy was unlikely to influence the causality, and the leave-one-out analysis confirmed that a single SNP did not drive the observed associations.
CONCLUSION
Our findings provide new proof of a positive causal relationship between AS and IC in the European population. Notably, it is Acute/Subacute IC, rather than IC as a whole or Chronic IC, that is associated with an elevated risk of AS. These results emphasize the significance of considering AS characteristics in the diagnosis of Acute/Subacute IC.
Topics: Humans; Iridocyclitis; Genome-Wide Association Study; Mendelian Randomization Analysis; Spondylitis, Ankylosing; Uveitis, Anterior
PubMed: 38274819
DOI: 10.3389/fimmu.2023.1295118 -
Indian Journal of Ophthalmology Feb 2024
Topics: Humans; Retinal Pigment Epithelium; Antibodies, Monoclonal, Humanized; Uveitis, Posterior; Macular Degeneration; Intravitreal Injections; Angiogenesis Inhibitors; Wet Macular Degeneration
PubMed: 38273689
DOI: 10.4103/IJO.IJO_712_23 -
Scientific Reports Jan 2024This study investigated the efficacy of golimumab in the management of refractory non-infectious panuveitis. Nineteen patients (38 eyes; mean age, 31 years) were...
This study investigated the efficacy of golimumab in the management of refractory non-infectious panuveitis. Nineteen patients (38 eyes; mean age, 31 years) were retrospectively reviewed between June 2016 and June 2022. All patients had bilateral eye involvement and Behçet's disease was the most common diagnosis (57.9%). Compared to the period before golimumab treatment, the rate of uveitis relapses after golimumab treatment significantly decreased from 1.73 to 0.62 events per person-years (incidence ratio 0.33, 95% confidence interval 0.19-0.57, P < 0.001). After golimumab therapy, 12 patients (63.2%) were able to reduce the number or dosage of immunosuppressive drugs, and the median dosage of systemic corticosteroids was reduced from 15.0 to 7.5 mg/d (P = 0.013) compared to baseline. The median logMAR visual acuity improved from 0.9 at baseline to 0.6 at the last visit (P = 0.006). Golimumab demonstrated efficacy against refractory non-infectious panuveitis in terms of a corticosteroid-sparing effect and reduced the rate of uveitis relapses to approximately one-third.
Topics: Humans; Adult; Retrospective Studies; Uveitis; Behcet Syndrome; Adrenal Cortex Hormones; Recurrence; Treatment Outcome; Antibodies, Monoclonal
PubMed: 38273077
DOI: 10.1038/s41598-024-52526-1 -
Retina (Philadelphia, Pa.) May 2024To evaluate features of infectious panuveitis associated with multiple pathogens detected by ocular fluid sampling.
PURPOSE
To evaluate features of infectious panuveitis associated with multiple pathogens detected by ocular fluid sampling.
METHODS
Single-center, retrospective, consecutive case series of patients with aqueous/vitreous polymerase chain reaction testing with >1 positive result in a single sample from 2001 to 2021.
RESULTS
Of 1,588 polymerase chain reaction samples, 28 (1.76%) were positive for two pathogens. Most common pathogens were cytomegalovirus (n = 16, 57.1%) and Epstein-Barr virus (n = 13, 46.4%), followed by varicella zoster virus (n = 8, 28.6%), Toxoplasma gondii (n = 6, 21.4%), herpes simplex virus 2 (n = 6, 21.4%), herpes simplex virus 1 (n = 6, 21.4%), and Toxocara (n = 1, 3.6%). Mean initial and final visual acuity (logarithm of the minimum angle of resolution) were 1.3 ± 0.9 (Snellen ∼20/400) and 1.3 ± 1.1 (Snellen ∼20/400), respectively. Cytomegalovirus-positive eyes (n = 16, 61.5%) had a mean final visual acuity of 0.94 ± 1.1 (Snellen ∼20/175), whereas cytomegalovirus-negative eyes (n = 10, 38%) had a final visual acuity of 1.82 ± 1.0 (Snellen ∼20/1,320) ( P < 0.05). Main clinical features included intraocular inflammation (100%), retinal whitening (84.6%), immunosuppression (65.4%), retinal hemorrhage (38.5%), and retinal detachment (34.6%).
CONCLUSION
Cytomegalovirus or Epstein-Barr virus were common unique pathogens identified in multi-PCR-positive samples. Most patients with co-infection were immunosuppressed with a high rate of retinal detachment and poor final visual acuity. Cytomegalovirus-positive eyes had better visual outcomes compared with cytomegalovirus-negative eyes.
Topics: Humans; Retrospective Studies; Male; Female; Panuveitis; Middle Aged; Aqueous Humor; Eye Infections, Viral; Polymerase Chain Reaction; Visual Acuity; Adult; Aged; DNA, Viral; Vitreous Body; Cytomegalovirus; Young Adult; Toxoplasma
PubMed: 38271688
DOI: 10.1097/IAE.0000000000004037