Ocular Immunology and Inflammation最新文献

Purpose: To present the clinical manifestations, ocular complications and treatment results of pediatric pars planitis (PPP).

Methods: This retrospective clinical study was conducted at a tertiary care center and included 28 children (51 eyes) aged 16 years or younger. Visual acuity (VA), anterior chamber cell (ACC) grade, vitritis grade, central macular thickness (CMT), fundus fluorescein angiography (FFA) score, treatment approach, complications, and relapse rate were recorded. All data were analyzed for baseline, third month, sixth month, ninth month, and twelfth month.

Results: The mean or median follow-up duration was 12 months. A total of twenty patients (71%) were administered Methotrexate (MTX) and the mean MTX dosage was 8.6 ± 1.1 mg. Ten patients (35%) were administered corticosteroid, and the mean dosage was 29.6 ± 4.2 mg. Adalimumab was administered to 16 patients (57%). There was a significant improvement in VA (-0.068, p = 0.025) during the first 3 months of follow-up. There were no significant changes in the VA during the subsequent follow-up periods. ACC grade decreased significantly in the third month (-0.765, p < 0.001), while vitritis grade decreased significantly in the sixth month (-0.502, p < 0.001). There was no significant change in CMT in all study eyes during the follow-up periods. However, there was macular edema in 14 eyes (27%) in baseline examination. After treatment, CMT was lower than 250 µm in 8 eyes (57%) in the last follow-up.

Conclusion: Immunosuppressive therapy is effective in suppressing intraocular inflammation. Treatment should be aimed at controlling inflammation, macular edema, and papillitis. Early diagnosis and aggressive treatment are important for a good visual prognosis.

Purpose: To estimate the incidence and predictive factors for cataract in eyes with episcleritis and scleritis, and to evaluate the outcome of cataract surgery in those eyes.

Design: Retrospective cohort study at uveitis subspecialty centers.

Methods: One thousand three hundred eighty-four eyes with non-infectious scleritis and episcleritis at risk of cataract were included. Predictive factors for cataract development were assessed by multivariable Cox regression. The main outcomes were development of cataract, defined as the first reduction of presenting visual acuity <20/40 attributed to cataract or else occurrence of cataract surgery itself. A second cohort of eyes with episcleritis and scleritis that underwent cataract surgery was evaluated for postoperative outcomes. Logistic regression was utilized to assess variables associated with visual acuity 20/40 or better one year after cataract surgery.

Results: Seventy-six eyes developed cataract (incidence = 0.025/eye-year, 95% confidence interval: 0.019-0.031). Age ≥65 years, elevated intraocular pressure ≥30 mmHg, use of oral corticosteroids at the preceding visit, and anterior chamber inflammatory activity were associated with increased cataract incidence. Race/ethnicity, type of scleritis, and bilaterality were unassociated with cataract risk after adjustment. Among 79 cataractous eyes that underwent cataract surgery, median presenting visual acuity improved by 6 ETDRS lines. Pre-operative factors including duration of inflammation, immunotherapy use, and corticosteroid use were not significantly associated with odds of post-operative visual acuity 20/40 or better.

Conclusions: Under subspecialty management, the incidence of cataract was low in eyes with episcleritis and scleritis. Cataract surgery was associated with large and sustained improvements in visual acuity.

Purpose: To report an unusual case of progressive photoreceptor disruption following a herpetic labial infection.

Method: Case report of a patient with severe idiopathic photoreceptoritis.

Results: A 46-year-old female patient presented with blurred vision and inferior photopsia in her right eye one week after a herpetic labial eruption. Initial examination revealed mild vitreous inflammation and focal photoreceptor alterations in the superior periphery of the right retina. Despite oral valaciclovir, the disease progressed rapidly, prompting referral to a tertiary care center. Autofluorescence and optical coherence tomography showed diffuse photoreceptor damage with impending foveal involvement. Extensive workup, including viral markers, was inconclusive. Given the temporal association with the herpetic infection and the sight-threatening progression, treatment was escalated to intravenous acyclovir and subsequently foscarnet for 2 weeks, which slowed down disease progression. The frontal margin of retinal foci stabilized with no further progression, while ellipsoid zone restoration was achieved following systemic corticosteroids and interferon alpha-2a.

Conclusion: The term "photoreceptoritis" describes conditions involving diffuse photoreceptor damage, predominantly unilateral, without primary choriocapillaritis. This case highlights a severe form of photoreceptoritis with diffuse photoreceptors loss, likely triggered by a herpetic labial infection. The disease was stabilized with antiviral therapy, while structural recovery of the photoreceptors was achieved through anti-inflammatory and immunosuppressive treatments. This report underscores the importance of a multidisciplinary approach to managing atypical retinal inflammatory disorders.

Purpose: To present a unique case of recurrent panuveitis in a young patient associated with a novel pathogenic variant in the PIK3CD gene, expanding the clinical spectrum of Activated PIK3 delta syndrome (APDS), a primary immunodeficiency that predisposes individuals to infections, autoimmunity, and malignancies.

Methods: We evaluated a 15-year-old boy with refractory panuveitis, recurrent infections, and lymphadenopathy with Hodgkin lymphoma. Ophthalmic assessment and ultra-wide field fundus fluorescein angiography were conducted. Whole exome sequencing identified a novel heterozygous pathogenic variant in the PIK3CD gene. Immunological and histopathological evaluations further supported the diagnosis of APDS.

Results: The patient experienced progressive vision loss despite immunosuppressive therapy with prednisolone, methotrexate, and mycophenolate mofetil. Genetic testing revealed a novel PIK3CD pathogenic variant (c.1002C>G;p.Asn334Lys), confirmed via Sanger sequencing and predicted by in-silico tools to be pathogenic. Initial improvement was observed with steroids, but frequent relapses upon tapering underscored the chronicity of his inflammatory condition.

Conclusion: This case underscores the importance of genetic testing in diagnosing complex, treatment-resistant uveitis in children. The novel PIK3CD pathogenic variant expands the genetic landscape of APDS, indicating the need to consider underlying genetic causes in cases with recurrent systemic inflammation and infections. Managing APDS requires a careful balance of immunosuppressive treatment and monitoring for potential malignancies, emphasizing a multidisciplinary approach to optimize patient outcomes.

Purpose: The presentation of ocular sarcoidosis may involve different parts of the eye and/or adnexal tissues. Uveitis is the most common manifestation, and when it involves the posterior segment, it often presents as peripheral multifocal choroiditis. The occurrence of symptomatic occlusive retinal vasculitis (ORV) associated with ocular sarcoidosis is not well described in the literature. The objective of this article is to describe well characterized cases of biopsy-proven ocular sarcoidosis presenting with symptomatic ORV.

Methods: We conducted a retrospective study using the ophthalmological database, to include patients with symptomatic ORV and biopsy-proven sarcoidosis, who were followed at a tertiary ophthalmology and internal medicine center. We described analysis of ocular and systemic manifestations at baseline and response to local and systemic treatments.

Results: We identified five cases, all male, including four from sub-Saharan Africa and one from France. Uveitis affected both eyes in all cases, and none showed intermediate uveitis, panuveitis, or peripheral multifocal choroiditis. Ocular manifestations were consistently associated with constitutional symptoms and pulmonary involvement, with a marked increase in acute phase reactants in all cases and lymphocytic meningitis in 2/5. All patients responded well to local and systemic glucocorticoids (GCs), with two patients showing GC dependence requiring the addition of an immunosuppressant.

Conclusion: We provide a comprehensive analysis of the clinical findings and management strategies of newly-diagnosed sarcoidosis presenting with symptomatic ORV. Although such cases are unusual, the diagnosis of sarcoidosis should be considered when ORV is encountered.

Purpose: To evaluate the rate of ocular complications for pediatric uveitic patients, treated with anti-tumor necrosis factor-alpha (TNF-α) agents.

Methods: Retrospective study of pediatric patients with non-infectious chronic anterior uveitis, treated with anti-TNF-α at two tertiary uveitis centers. The primary outcome was the effect of therapy on ocular complications rate. Secondary outcomes included disease activity, achievement of steroid sparing effect and visual acuity.

Results: The cohort included 59 pediatric patients (115 eyes) with mean age of 6.6 ± 3.9 years. Most were females (50, 85%), Caucasians (49, 83%) with bilateral uveitis (56, 95%). The leading etiology was juvenile idiopathic arthritis-associated uveitis (36, 61%). Adalimumab was the most used medication (56 patients, 95%), with active uveitis being the most common indication for treatment (50 patients, 85%). The median interval between uveitis diagnosis and treatment initiation was 16.4 months (range 0.2-162). Anterior chamber cells and flare decrease was noted as early as 6 months after treatment initiation and remained stable throughout the follow-up period. Mean number of steroids drops per day decreased from 2.31 ± 2.4 at baseline to 0.62 ± 0.9, 0.60 ± 0.8, 0.55 ± 0.8, and 0.33 ± 0.6 at 1, 2, 3, and 5 years, respectively. In a multivariate analysis, longer interval between uveitis diagnosis and anti-TNF-α initiation was associated with an increased risk of developing ocular complications including cataract and glaucoma (HR = 1.02, p = 0.0004; HR = 1.04, p = 0.0002, per month, respectively).

Conclusions: Timely employment of anti-TNF-α agents to treat pediatric non-infectious anterior uveitis may improve outcomes, maintain inflammatory control, and reduce rate of complications.

Purpose: To evaluate the retinal vascular changes related to early Rheumatoid Arthritis (RA) and to correlate these changes with disease activity.

Methods: 108 eyes of 54 early RA patients and 66 eyes of 33 age and sex-matched healthy participants underwent Optical Coherence Tomography Angiography (OCTA) examination to evaluate superficial (SCP), deep retinal capillary plexus (DCP), and radial peripapillary capillaries (RPC), we collected Values of the vessel density (VD) of SCP and DCP, central foveal thickness (CFT), foveal avascular zone (FAZ) along with a full rheumatological examination and disease activity score by (DAS28).

Results: RPC values were significantly lower among RA patients in all subfields apart from the temporal one. There was a significant difference between RA patients and healthy controls in foveal and all perifoveal measurements, except the nasal perifoveal region. Also, the FAZ value was significantly higher in RA patients. We performed Spearman's correlation between OCTA and RA disease parameters and detected a negative correlation between the duration of disease-related symptoms and RPC and parafoveal measurements and between DAS-28 and foveal measurements. A significant positive correlation between anti-CCP titer and FAZ measurements was detected among RA patients.

Conclusion: OCTA can detect early retinal microcirculation affection in RA patients in the form of a reduction in the vascular density in most of the subfields of the peripapillary area as well as the fovea, perifoveal, and parafoveal areas. The results of the current study can provide new insight into the applications of OCTA as a diagnostic tool in RA patients' assessment and follow-up.

Purpose: To identify the incidence of cataract and the outcomes of cataract surgery in eyes with ocular cicatricial pemphigoid (OCP).

Methods: 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.

Results: 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.

Conclusions: 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.

Purpose: We sought to re-evaluate the utility of inpatient ocular Candidiasis screening, subsequent interventions, and outcomes, to ultimately guide alignment between American Academy of Ophthalmology (AAO) and Infectious Diseases Society of America (IDSA) guidelines.

Methods: A retrospective cohort study was conducted from 2017-2024 at two tertiary care centers in the New York metropolitan area. Inclusion criteria included positive Candidal cultures, a pertinent International Classification of Diseases, 10^th revision (ICD-10) code, or both. Culture results, mental status, visual acuity, ocular symptoms, systemic antifungal use, dilated fundus exam (DFE) findings, immunocompromised status, and interventions performed were recorded.

Results: Of 897 patient encounters meeting inclusion criteria, 285 (31.8%) underwent consultation to rule-out ocular Candidiasis. Among these patients, 212 (74.4%) had an unremarkable DFE, 35 (12.3%) had DFE findings attributable to other comorbidities, 24 (8.4%) had nonspecific DFE findings requiring follow-up, nine (3.2%) were diagnosed with Candida retinitis, and one (0.4%) had Candida endophthalmitis. There were no statistically significant differences in the odds of DFE positivity between the immunocompromised versus immunocompetent group (χ2 = 0.881, p = 0.348) or the symptomatic versus asymptomatic group (χ2 = 0.297, p = 0.586). During the entire study period, no ophthalmology-directed interventions were made.

Conclusion: Candida endophthalmitis and Candida retinitis are rare in the inpatient setting, even in patients with confirmed Candidemia. Patients are universally started on empiric intravenous antifungal treatment by primary or infectious disease teams prior to ophthalmology consultation, limiting the yield of these examinations. Further studies should be undertaken to create a protocol for these patients such that serious sequelae of disease are prevented while resources are also utilized appropriately.

Purpose: To describe an atypical case of bilateral uveal effusion, presumably triggered by amoxicillin, in a 10-year-old child with pneumonia.

Methods: A comprehensive ophthalmological evaluation and imaging studies, including anterior segment and macular optical coherence tomography, ultrasound biomicroscopy, and B-mode ultrasonography, were performed to assess anatomical changes and confirm the diagnosis.

Results: A 10-year-old boy presented with an acute, painless loss of visual acuity in both eyes, primarily affecting distance vision. He had a recent history of pneumonia, which had been treated with amoxicillin. Ophthalmic examination revealed bilateral acute myopia, significant refractive error, and shallow anterior chambers in both eyes. Ultrasound biomicroscopy and anterior segment optical coherence tomography revealed fluid in the supraciliary space and anterior rotation of the ciliary processes, suggestive of uveal effusion. Based on these findings, uveal effusion was suspected, prompting the discontinuation of amoxicillin. The patient was treated with topical dexamethasone, cyclopentolate, and oral azithromycin. After stopping amoxicillin and initiating treatment, the patient's myopia resolved, anterior chamber depth normalized, and the uveal effusion was resolved within three days. The patient remained asymptomatic during a 6-month follow-up period.

Conclusion: This case suggests a possible link between amoxicillin use and bilateral uveal effusion, a relationship not previously reported. Healthcare professionals should consider uveal effusion in the differential diagnosis of patients presenting with bilateral acute myopia, particularly when associated with recent medication use. Further research is needed to explore the potential role of amoxicillin in inducing uveal effusion.