Journal of Ophthalmic Inflammation and Infection最新文献

Purpose: To report a case of two monozygotic twins presenting with simultaneous onset of bilateral uveitis of variable phenotypic presentations, one of whom was pathologically confirmed to have sarcoidosis.

Observation: Two 21-year-old monozygotic male twins (Twin A and Twin B) presented with bilateral red eyes and photophobia of six weeks' duration. Their past medical history included learning difficulties and low IQ since early childhood. Twin A was operated for Celiac Artery Compression Syndrome. Examination and work-up revealed that Twin A had bilateral anterior granulomatous uveitis, and retinal phlebitis in addition to hilar and mediastinal lymphadenopathy. A biopsy was taken, and histopathological examination showed non-caseating granuloma. Twin B had bilateral non-granulomatous anterior uveitis, chorioretinal lesions, and peripheral retinal vasculitis. Genetic testing in the form of a Whole Exome Sequencing was done, and no causal variant was detected for uveitis or sarcoidosis, however, a homozygous likely pathogenic duplication in SYNGAP1 was detected. This mutation is associated with autosomal dominant intellectual developmental disorder.

Conclusion: This is the first-reported case of concurrent bilateral uveitis in monozygotic twins, with confirmed sarcoidosis in one. This presentation highlights the role of genetic predisposition and shared environmental factors in disease onset and clinical manifestations. Further research into the genetic-environmental interplay is needed to elucidate the mechanisms underlying simultaneous disease onset and guide personalized monitoring strategies for at-risk families.

Purpose: The purpose of this study was to describe the case of a pediatric patient diagnosed with mucous membrane pemphigoid (MMP) with exclusive ocular involvement presenting at diagnosis with peripheral ulcerative keratitis and provide a review of the literature.

Methods: A 12-year-old girl presented with cicatricial conjunctivitis and peripheral ulcerative keratitis (PUK). A conjunctival biopsy and direct immunofluorescence revealed linear deposits of IgG, IgM, and C3 at the basement membrane zone, confirming a diagnosis of ocular MMP.

Results: The patient was initially treated with dapsone 25 mg/day and prednisone 40 mg/day along with topical treatment including cyclosporine 0.05%, loteprednol etabonate 0.05%, and sodium hyaluronate 0.15% and trehalose 3%. Inflammation persisted as an increase in the extension of symblepharon was documented. Subsequently, dapsone was switched to oral methotrexate 15 mg/week and prednisone was successfully tapered to 5 mg/day. After three years of follow-up, disease activity remained quiescent.

Conclusions: Pediatric mucous membrane pemphigoid with ocular involvement is a rare condition of which few reports have been published, resulting in scarce information regarding its clinical course and response to treatment. We report the first case observed in a Hispanic patient, opening with peripheral ulcerative keratitis, and responding successfully to methotrexate.

Filamentous fungi are among the emerging causes of infections, although corneal infections caused by these fungi are rare, they can lead to severe clinical outcomes. In this report, we present the first documented case of keratitis caused by Stemphylium lycopersici, a filamentous hemipteran fungus of the Pleosporaceae family. A 66-year-old man presented conjunctival redness, irritation, and visual deterioration in his left eye, following a stone chip injury that occurred five months earlier. Despite multiple treatments, the causative pathogen remained unidentified, leading to worsening symptoms and significant vision loss. This deterioration led the patient to seek care at our hospital. An in vivo confocal microscopy (IVCM) examination suggested a fungal infection. Consequently, antifungal medications were administered, but the condition did not improve. Metagenomic next-generation sequencing (mNGS) examination of corneal scrapings revealed a mixed infection with S. lycopersici and human alphaherpesvirus 1. This definitive diagnosis facilitated the implementation of targeted therapy, leading to progressive symptomatic improvement. Early and rapid pathogen identification using mNGS analysis of corneal scrapings enables accurate management of infectious keratitis, contributing to visual recovery and reducing the risk of resistance to corneal pathogenic microbes.

Background: To report a case series of microbial keratitis showing atypical yeast-like structures in direct microscopy which were culture-negative but subsequently identified as yeast cells by PCR and DNA sequencing.

Main text: This is a retrospective, non-comparative case series of eight patients with infectious keratitis, where smear examination (potassium hydroxide + calcofluor white) showed spore like structures resembling yeast. There was no growth in any solid culture media. Routine PCR assay was performed using pan fungal primers followed by Sanger sequencing and nucleotide sequences were analysed using NCBI-BLAST software. Medical treatment in all patients were initiated based on clinical suspicion and presumptive microbiology report. Therapeutic penetrating keratoplasty was performed for patients not responding to medical antifungal therapy. Demographic, clinical data were collected for each patient from electronic medical records of the patients and outcome analysed. Amplification of fungal DNA was seen in the PCR assay of all samples. Nucleotide sequences of the amplicons obtained after Sanger sequencing and NCBI-BLAST analysis were found identical to Candida albicans (n = 7) and Citeromyces matriensis (n = 1). Patients were treated with antifungal drugs such as topical natamycin 5% or amphotericin B 0.15%. Ulcer resolved with scarring in 5 patients (62.5%), one patient had failed graft after therapeutic penetrating keratoplasty (12.5%), one (12.5%) eye became phthisical and one patient (12.5%) was lost to follow up.

Conclusion: Atypical structures resembling yeast-like cells detected in direct smear examination in the absence of growth in culture media should raise the suspicion of a fungal etiology and warrant further investigations to establish the diagnosis.

Topic: Vogt-Koyanagi-Harada (VKH)-like uveitis is uniquely reported with immune checkpoint inhibitors (ICI) and BRAF/MEK inhibitors. This article aims to provide a comprehensive portrait of the comorbidities, ocular presentations, treatments, and visual outcomes of patients with VKH-like uveitis following ICI therapy.

Clinical relevance: ICIs are increasingly used in cancer therapy, but poorly understood ocular immune-related adverse events (irAEs) can lead to suspension of treatment and be vision-threatening.

Methods: We conducted a systematic review (PROSPERO #CRD42024558269) according to PRISMA guidelines. MEDLINE, Embase, CENTRAL, and Web of Science were searched for English articles published up to June 28, 2024. All study designs reporting on incident VKH-like uveitis following ICI were included. Risk of Bias was assessed using a tool modified from Murad et al. (2018).

Results: Of 865 articles, we included 42 articles (4 observational studies, 28 case reports, 6 case series, 3 letters, and 1 editorial) from 12 countries, comprising 52 patients. The mean age was 60.0 ± 11.9 years, and 32 (61.5%) were females. Thirty-six (69.2%) had melanoma, and most were undergoing treatment with a PD-1 inhibitor alone (n = 33, 63.5%) or in combination with a CTLA-4 inhibitor (n = 10, 19.2%). The mean duration of ICI treatment before VKH-like uveitis symptoms was 22.2 ± 29.6 weeks, and the mean duration of ocular symptoms was 16.7 ± 18.6 weeks, with wide variation. Overall, 43 patients (73.1%) had imaging or exams suggesting bilateral involvement and 21 cases (40.4%) suggesting panuveitis. Only 31 cases (59.6%) met the acute initial-onset uveitis criteria, and 15 (28.8%) met the chronic phase criteria. Most (n = 47, 90.4%) required systemic or intravitreal steroids, termination of ICI (n = 31, 59.6%), and experienced full resolution or remission of visual symptoms (n = 43, 82.7%). Most articles (n = 40, 95.2%) were judged to be at medium risk of bias.

Conclusion: This descriptive systematic review consisted mostly of case reports, but it confirmed that a high proportion of VKH-like uveitis occur with PD-1 inhibitors and melanoma patients. VKH-like uveitis can lead to suspension of treatment. Further collaboration between oncologists and ophthalmologists is needed in the continuum of cancer care.

Introduction: Paediatric uveitis treatment recommendations suggest a step-up treatment approach starting with topical treatment, followed by antimetabolites and thereafter biologics. With this study, we are investigating the safety and efficacy of the current treatment approach in a large cohort.

Material and methods: Single center retrospective study. Patients with non-infectious uveitis under the age of 18 years at first presentation, between January 2012 and June 2022, were eligible for inclusion. Data extracted from the electronic health records included age at first presentation, sex, involved eye segment, visual acuity (VA), complications, associated systemic disease, treatments, and number of consultations. Cases were grouped according to their final treatment regime (topical only, methotrexate, TNF alpha inhibitor, other). VA outcome, treatment response, adverse events, and frequency of consultations were evaluated. The study was approved by the local ethics committee.

Results: 64 non-infectious paediatric uveitis cases were included. Age at first diagnosis ranged from 2 to 17 years, with a two-peak distribution, 52% were male. Anterior uveitis was the most common presentation, followed by intermediate uveitis, posterior uveitis, and panuveitis. Topical treatment achieved remission in 23%, anti-metabolites in 12%, and escalation to TNF alpha inhibitors in 30%. Alternative treatments or observation only were documented in 16% and 17%, respectively. Median duration from first presentation to the start of anti-metabolite or TNF alpha inhibitor were 115 days and 269 days, respectively. There was a median of eight consultations during the first year of follow up. Frequency of consultations during the first year increased with every treatment escalation. VA outcome did not differ between the different treatment groups.

Conclusion: The step-up treatment approach shows a safe profile in regards to VA outcome. Methotrexate presents a high rate of treatment failure and adverse effects. Adalimumab and infliximab are effective and safe. Timely treatment escalation might lower treatment burden for affected children, their families, and health care providers.

Objective: To describe two patients with fulminant endophthalmitis after penetrating ocular injuries by cat claw and review the literature regarding animal-related endophthalmitis.

Design: Case series.

Participants: In the study period, 298 patients were identified with a diagnosis of endophthalmitis, of which two were identified in association with open globe injury by cat claw.

Methods: All patients with endophthalmitis after cat claw open globe injury in an academic center in a 20-year period are reported. Clinical and laboratory presentations, medical and surgical treatment, and outcomes are described. A literature review is summarized.

Exposure: Open globe injury by cat claw.

Main outcome measures: Interventions and ocular anatomical and functional outcomes.

Results: Case 1: A 27-year-old female sustained a penetrating injury of the left eye by a cat claw. The laceration was repaired the next day, and intravitreal antibiotics injections were given. She developed acute fulminant endophthalmitis the following day and underwent pars plana vitrectomy, anterior chamber washout, and intravitreal antibiotics injection. Cultures isolated Propionibacterium acnes. A retinal detachment was noted after 48 days, requiring a second pars plana vitrectomy and tamponade with sulfur hexafluoride gas. The retina remained attached. Visual acuity at 14 months follow-up was 20/200. Case 2: A 42-year-old male developed endophthalmitis two days after a penetrating injury of the right eye by a cat claw. Pars plana vitrectomy and intravitreal antibiotics injections were performed the same day. Cultures identified Pasteurella multocida. The patient progressed to panophthalmitis in 24 h and received intravenous antibiotics. He developed proliferative vitreoretinopathy with recurrent retinal detachments requiring multiple vitrectomies. His visual acuity was hand motions at 7 months follow-up.

Conclusions and relevance: Open globe injuries caused by cat claw may result in hyperacute and acute endophthalmitis. Propionibacterium acnes and Pasteurella multocida were isolated from the two cases reported here. Despite immediate interventions, both patients developed retinal detachment and had poor final visual acuity. Our report reveals that endophthalmitis caused by animal trauma is rare with potentially devastating outcomes, thereby requiring timely diagnosis and treatment.

Background: Pediatric uveitis is often asymptomatic, which may expose to sight-threatening ocular complications. The impact of modern medication on frequency of long-term ocular complications in pediatric patients with anterior idiopathic uveitis (idio-U) or juvenile idiopathic arthritis associated uveitis (JIA-U) is not fully understood. We aimed to evaluate the occurrence of ocular complications during the era of modern treatment on the population-based cohort of children with idio-U or JIA-U.

Methods: A longitudinal, population-based cohort study of children with idio-U or JIA-U in 2008-2020. Variables assessed included age, gender, age at diagnosis, laterality, chronicity, vision, and ocular complications.

Results: 107 pediatric patients and 172 eyes with either idio-U (19 patients) or JIA-U (88 patients) were included. The mean age at uveitis onset was 10.0 ± 3.7 and 5.4 ± 3.2 years in idio-U and JIA-U, respectively (p < 0.001). Uveitis was chronic in 58% in idio-U and 74% in JIA-U patients, respectively. Uveitis was complicated with glaucoma in 45% of idio-U and 18% of JIA-U patients (p = 0.019). Cataract was developed in 31% of idio-U and 22% of JIA-U eyes (p = 0.28), and posterior synechiae in 21% and 9% of the eyes with idio-U and JIA-U, respectively. None of the eyes were hypotonic. Female gender was overrepresented in ocular complications. Glaucoma surgery was accomplished in 25 (15%) and cataract surgery in 19 (11%) eyes. Bilateral visual acuity remained > 0.5 in all patients.

Conclusions: Glaucoma, ocular hypertension, and cataract were the most typical complications of uveitis. Complications occurred mostly in girls and in idio-U patients. JIA-U patients with severe uveitis, young age at uveitis onset and female gender were predisposing factors for surgical management.

Objective: To report a case series of non-infectious anterior scleritis resistant to multiple lines of conventional therapies which were eventually successfully treated with off-label subconjunctival dexamethasone implant (Ozurdex^®) injection (SDI).

Methods: A retrospective case series of 4 patients (6 eyes).

Results: In the index case series, the patients had a mean age of 57.2 years (range 36 to 82 years, SD 19.2 years) with 50% being female. Two patients had underlying autoimmune diseases: rheumatoid arthritis (n = 1), and granulomatosis with polyangiitis (GPA) (n = 1). The other patients were diagnosed with idiopathic anterior scleritis after extensive systemic investigations (n = 2). The mean follow-up duration and the mean number of concomitant therapies prior to SDI was 27 (SD 17.7) months and 2 (SD 0.81), respectively. In all patients, symptom resolution and significant improvement in disease activity were achieved after SDI, persisting for an extended period following the resorption of the implant. No scleral melt, infection or ocular hypertension were noted following SDI.

Conclusion: SDI may be a safe and effective therapeutic option for resistant non-infectious anterior scleritis.