Ocular Immunology and Inflammation最新文献

Purpose: We report a rare and challenging case of bilateral necrotizing scleritis in primary Sjögren's syndrome (pSS).

Methods: Retrospective case report.

Results: A 72-year-old diabetic, hypertensive female patient presented with sudden onset of painful red left eye and was noted to have a corneal ulcer with severe thinning. She was managed with topical fortified antibiotics and tissue glue and bandage contact lens. During subsequent follow-ups, she developed necrotizing scleral melts in both eyes. On investigations, antinuclear antibodies were positive in a dilution of 1:160 with 2+ speckled pattern, with antinuclear antibody line immunoassay showing anti SS-A/ Ro52 positive. In view of rapidly developing scleral thinning and impending perforation, she was started on intravenous methylprednisolone 1 g/day for 3 days, along with steroid-sparing immunomodulatory therapy (mycophenolate mofetil 500 mg twice a day). She showed a rapid response to therapy and is currently stable on tapering oral steroids and mycophenolate mofetil.

Conclusion: This case underscores the unique presentation of pSS, characterized by bilateral necrotizing scleritis. The favorable outcome was attained through prompt immunosuppressive intervention and a collaborative, multidisciplinary approach. Further, this case report addresses a gap in the existing literature concerning pSS-related scleritis. It also emphasizes the crucial role of a rheumatologist in the comprehensive management of this condition.

Purpose: To assess the clinical relevance of pathophysiology-based biomarkers, specifically serum C1q and whole blood interferon gene signature score (IGSS), in ocular tuberculosis (OTB) diagnosis by conducting an integrative analysis of clinical presentations and treatment response.

Methods: This retrospective cohort study analysed data from 70 patients with suspected OTB at a tertiary care uveitis practice in Indonesia. Serum C1q levels and whole blood IGSS were quantified. Patients were categorized into four quadrants based on their biomarker profiles: quadrant 1 (high C1q & low IGSS), quadrant 2 (high C1q & high IGSS), quadrant 3 (low C1q & high IGSS), and quadrant 4 (low C1q & low IGSS). Characteristics of clinical presentations, work-up results, and treatment outcomes were explored according to the predefined quadrants.

Results: We identified that the majority of OTB patients diagnosed with concurrent active pulmonary TB were in quadrant 1, 2, or 3 (20/23, 87.0%). Twenty-seven patients (27/47, 57.4%) with clinically undifferentiated uveitis were in quadrant 4 (p < 0.001). Among patients in quadrants 1, 2, and 3, completion of a full course of antitubercular treatment (ATT) was associated with a lower number of patients showing persistence or recurrence of ocular inflammation compared to those who were not fully treated with ATT (14.3% vs 85.7%, p = 0.001).

Conclusions: Based on the analysis of clinical features and treatment outcomes, patients with elevated levels of either or both serum C1q and whole blood IGSS may reflect active TB disease in the eye, necessitating full ATT management.

Purpose: To investigate the diversity of ocular manifestations in patients with positive perinuclear antineutrophil cytoplasmic antibody (p-ANCA).

Methods: The clinical records of five patients exhibiting ocular manifestations and testing positive for serum MPO-ANCA were retrospectively reviewed.

Results: Three out of five patients were female. The youngest patient was aged 26, whereas the eldest was 83 years old. 80% (n = 4) of them had purely anterior segment involvement, with the commonest manifestation being peripheral ulcerative keratitis and scleritis. Only one patient had posterior segment involvement, specifically posterior ischemic optic neuropathy. Two patients demonstrated unilateral involvement. There was only one patient diagnosed with systemic involvement, presenting as mixed nephrotic and nephritic syndrome, who required a course of intravenous methylprednisolone during the active stage. Three patients required second-line immunosuppressants throughout the course due to frequent relapse.

Conclusion: Anterior segment ocular manifestations are not uncommon and can be the initial presentation of p-ANCA-associated vasculitis (AAV). Therefore, its evaluation should be considered in establishing the diagnosis of AAV, a life-threatening disease.

Purpose: To provide a comprehensive overview of the diagnostic and therapeutic journey of a pediatric patient with persistent sarcoid-associated panuveitis over a 10-year period, who ultimately developed bilateral macular subretinal fibrosis and visual loss.

Methods: Retrospective case report.

Results: The patient was diagnosed with sarcoidosis after undergoing a transbronchial biopsy. She was followed up because of granulomatous panuveitis, multifocal choroiditis, and papillitis bilaterally. She maintained a stable condition, and visual acuity was 0.3 RE and 0.5 LE. Immunomodulatory therapy included prednisone, methotrexate, and adalimumab. The patient was lost to follow-up for 20 months because of the COVID-19 pandemic. She was represented with active uveitis and was not responding to TNF-ɑ inhibitors (adalimumab and infliximab). Ultimately, the patient's intraocular inflammation was successfully controlled by using intravitreal steroids (Triamcinolone and Fluocinolone acetonide implant). However, the visual outcome was guarded because of bilateral subretinal fibrosis.

Conclusion: 10% of patients with sarcoidosis-associated uveitis risk blindness in one eye. The index case progressed to sight-robbing bilateral subretinal fibrosis, a rare complication of ocular sarcoidosis despite a combination of conventional and biologic anti-inflammatory therapies. There is a pressing need to develop new treatment agents for refractory non-infectious uveitis.

Background: Increased reactive oxygen species (ROS) are involved in the pathological process of dry eye disease. Our previous results suggested that norepinephrine (NE) has a protective effect on dry eye.

Purpose: This study explored the potential therapeutic role and underlying mechanisms of NE in benzalkonium chloride (BAC)-induced dry eye disease.

Methods: BAC-pretreated human corneal epithelial cells (HCEpiC) were cultured with various concentrations of NE. A BAC-induced dry eye mice model was established to explore the role of NE. Alterations in mice corneal tissues, ROS levels, mitochondrial function, and mitophagy levels were analyzed.

Results: In vitro, our results revealed that BAC-exposed HCEpiC led to mitochondrial malfunction, which involved excessive ROS production, decreased mitochondrial membrane potential (MMP), and promoted mitochondrial fragmentation through increased DRP1 and fission protein 1 (Fis1) expression and reduced mitofusin 2 (Mfn2) expression. Moreover, topical BAC application induced excessive mitophagy. These effects were reversed by NE. Additionally, the increased expression of LC3B, SQSTM1/p62, PINK1, and Parkin, which control mitophagy, in BAC-exposed HCEpiC was suppressed by NE. In BAC-induced C57BL/6J mice, NE resulted in lower fluorescein staining scores, decreased TUNEL-positive cells, and decreased mitochondrial fragmentation.

Conclusions: In conclusion, our findings showed that NE therapy prevented HCEpiC following BAC application by regulating mitochondrial quality control, which is controlled by PINK1/Parkin-dependent mitophagy. Our research suggests a potential targeted treatment for dry eye disease.

Purpose: We describe a rare complication of macular hole formation in rickettsia post-fever retinitis.

Patients and methods: Retrospective observational case report of a patient who presented with post-fever retinitis and cystoid macular edema that later progressed to a macular hole. Clinical record and multimodal imaging including fundus photography, fundus fluorescein angiography (FFA), and spectral domain optical coherence tomography (SD-OCT) were analyzed.

Results: Initial examination revealed multifocal retinitis lesions with vitritis associated with disc and macular edema, consistent with a diagnosis of post-fever retinitis. During the course of her treatment, she developed a full-thickness macular hole, complicating the management and requiring surgical intervention.

Conclusion: Continued monitoring and management are essential to address potential sequelae and ensure minimised long-term ocular morbidity in affected patients. Further reports are needed to precise and characterize this clinical relationship and its management.

Purpose: To report a case of exudative retinal detachment (ERD) in a child with Acute retinal necrosis (ARN).

Method: Retrospective Chart Review.

Result: A six-year-old boy presented with anterior uveitis with hypopyon and exudative retinal detachment with peripheral confluent patches of retinitis in the left eye. Examination of the right eye was unremarkable. B scan ultrasonography of the left eye revealed moderate vitreous echoes along with a membrane in the inferior quadrant persisting on low gains indicating localised inferior retinal detachment with the presence of shifting fluid. Fundus examination under general anaesthesia failed to locate any break in the retina. Polymerase chain reaction from the aqueous aspirate was positive for Herpes Simplex Virus (HSV)-I. The child was treated with intravenous acyclovir three times a day, intravitreal injection of ganciclovir and subsequently started on oral steroids (0.5 mg/kg/day). While continuing antiviral treatment, dose of corticosteroid was gradually increased to 1 mg/kg/day. Intravenous injection of acyclovir for 28 days and six intravitreal injections with corticosteroid led to complete resolution of ERD. Six months after initial presentation, the patient underwent lens aspiration with intraocular lens (IOL) implantation under general anaesthesia, under antiviral coverage. Following surgery, his BCVA in the left eye improved to 6/12.

Conclusion: ERD is a rare complication of ARN, typically associated with HSV infection. Meticulous monitoring and combined systemic and local antiviral therapy, complemented by judicious corticosteroid use, are essential for managing such cases effectively.

Purpose: To report a treatment approach of a rare presentation of herpes zoster ophthalmicus (HZO) with optic neuropathy and oculomotor nerve palsy.

Methods: Report of one case.

Results: Multiple lesions involving the optic nerve and cavernous sinus were demonstrated on magnetic resonance imaging in a patient with a characteristic herpes zoster rash in the V1 dermatomal distribution. Following treatment with systemic ganciclovir and intravenous corticosteroids, the patient experienced dramatic improvement in visual acuity, from hand motion to 20/25.

Conclusion: This report demonstrates a rare case of HZO optic neuropathy with cavernous sinus involvement that was successfully treated with an unconventional medication regimen.

Purpose: To characterize the spectrum of uveitis in patients visiting three tertiary hospitals in Hanoi, Vietnam.

Methods: This study collected prospective and multicenter data from patients diagnosed with uveitis at three tertiary hospitals in Hanoi City, Vietnam, between January 2022 and January 2024. Data on age, sex, clinical and laboratory findings, and etiology were collected.

Results: Of 410 patients included, 54.6% were women. The mean age of patients was 39.9 years. Most cases were unilateral and chronic. Anterior uveitis was the most common case (40%), followed by panuveitis (30%), posterior uveitis (26.1%), and intermediate uveitis (3.9%). Undifferentiated uveitis, accounting for 32%, was the most prevalent form across all anatomical groups. The leading etiologies for anterior uveitis included Posner-Schlossman syndrome (18.5%), cytomegalovirus (CMV)-induced uveitis (11%), and Herpes simplex virus-induced uveitis (8.5%). For posterior uveitis, the primary causes were Vogt-Koyanagi-Harada (VKH) syndrome (17.8%), toxocariasis (10.3%), and toxoplasmosis (6.5%). The identified causes of panuveitis included VKH syndrome (24.4%), Behcet's disease (15.4%), and CMV-induced panuveitis (5.7%). We observed six cases of uveitis associated with Haemophilus influenzae (1.5%) without any concomitant systemic symptoms. In our patient population, the most common complication was cataract (11.2%), followed by uveitic macular edema (11%).

Conclusions: Various uveitis patterns were observed among Vietnamese patients, with non-infectious uveitis being predominant.

Purpose: To determine the patterns of uveitis evident in patients presenting at the Uveitis Department of a tertiary care eye hospital located in Karachi, Pakistan.

Study design: Prospective cross-sectional study.

Location and duration of study: This study was conducted at Layton Rahmatullah Benevolent Trust (LRBT) Tertiary Teaching Eye Hospital-Korangi, Karachi, Pakistan from August 2020 to July 2023.

Methodology: This prospective study was conducted at the Uveitis Department of LRBT Tertiary Teaching Eye Hospital, Karachi, Pakistan, from August 2020 to July 2023, following ethical review board approval. Patients underwent detailed history-taking, clinical examination by a uveitis specialist. Informed written consent was obtained. Demographic data were recorded, excluding post-opendophthalmitis cases. Laboratory tests and ocular investigations were performed when necessary.

Result: In this study, 5791 patients were analysed, with a predominance of females (55.4%) and a mean age of 38.22 ± 13.06 years. Bilateral eye involvement was found in 60.4% of cases, and the most common uveitis types were anterior (35.1%) and pan-uveitis (28.9%). Non-infectious uveitis (72.14%) was more prevalent, with idiopathic uveitis being the most common cause (33.2%). Significant associations were found between anatomical locations of uveitis and various clinical characteristics (age, gender, eye involvement and infection status). For instance, anterior uveitis was more commonly associated with unilateral eye involvement, while bilateral eye involvement was more frequent in cases of pan-uveitis and posterior uveitis. Additionally, infectious aetiology was more prevalent in pan-uveitis and posterior uveitis (p < 0.001).

Conclusion: The primary aim of this study was to determine the patterns of uveitis cases observed at a tertiary eye care hospital in Karachi, Pakistan. The predominant aetiology identified was idiopathic uveitis. Nevertheless, it is essential to recognize that the determined prevalence may be subject to the inherent limitations of this study, notably pertaining to financial barriers and limited availability of diagnostic modalities.