Ocular Immunology and Inflammation最新文献

Purpose: To identify all possible ophthalmic presentations in children with Presumed trematode-induced uveitis, and determine their incidence in Beni Suef. Additionally, it explores potential correlations between ocular signs and demographic variables.

Methods: The study is a retrospective, case series which was conducted in three phases. The first phase involved subject recruitment and demographic data collection. The second phase included a thorough general examination and laboratory workup. In the third phase, ophthalmological examination was conducted.

Results: A total of 83 patients (99 eyes) were included. 80 of them were males. The mean age was 11.1 ± 2.7 years. Unilateral ocular involvement was seen in 66 patients. Anterior chamber granuloma was the most frequent presentation (71.7%), followed by ciliary body granuloma (15.2%) and vitritis (13.1%). Corneal granuloma occurred in 10.1%, macular edema in 9.1%, episcleral involvement in 6.1%, and iris involvement in 5.1%. Complications included cataracts (9 cases) and phthisis bulbi (1 case). One case showed hemorrhagic AC granuloma. Anterior chamber granuloma was more common in older children (mean age 11.5 years, p = 0.010), while vitritis was more common in younger ones (mean age 9.1 years, p = 0.029). No significant correlation was found between ocular signs and residence.

Conclusion: In Beni Suef, children with presumed trematode-induced uveitis have diverse ocular manifestations, primarily anterior chamber and ciliary body granulomas with vitritis. Complications such as cataract and phthisis bulbi may occur. Atypical cases, including hemorrhage within a granuloma, may develop. The study suggests age-related variations in ocular signs but no significant correlation with residence.

Purpose: To report a rare case of unilateral dacryoadenitis and contralateral solitary choroidal granuloma as the initial presentation of systemic sarcoidosis, highlighting diagnostic challenges and treatment complications.

Methods: A 45-year-old male presenting with right-sided dacryoadenitis and a contralateral choroidal granuloma was evaluated. The diagnostic work-up included clinical examination, orbital and chest CT, SPECT-CT with gallium-67, biopsies of the lacrimal gland and mediastinal lymph nodes, serum ACE and lysozyme levels, and fundus imaging with EDI-OCT. The diagnosis of sarcoidosis was established based on WASOG and IWOS criteria. Treatment included corticosteroids, azathioprine, and adalimumab. Anti-tubercular therapy was subsequently added after a later diagnosis of tuberculosis during sarcoidosis treatment.

Results: At presentation, visual acuity was 20/200 in the right eye (OD) and 20/25 in the left eye (OS), with right-sided proptosis and photophobia. Orbital CT showed lacrimal gland enlargement; biopsy revealed lymphoplasmacytic infiltration without granulomas. EDI-OCT confirmed a solitary choroidal granuloma in the OS. Elevated ACE and lysozyme, bilateral hilar lymphadenopathy, and gallium-67 uptake supported the sarcoidosis diagnosis. Initial immunosuppressive therapy improved symptoms, but disseminated tuberculosis developed, requiring 14 months of anti-tubercular therapy.

Conclusion: This case highlights the diagnostic complexity of ocular sarcoidosis with inconclusive histopathology and underscores the diagnostic value of solitary choroidal granuloma. In tuberculosis-endemic regions, thorough infectious screening before immunosuppression and long-term multidisciplinary follow-up are essential to prevent serious complications.

Purpose: Diagnosis of giant cell arteritis (GCA) can sometimes be challenging, especially in the setting of a negative CRP.

Methods: We report a challenging case of GCA.

Results: A 73-year-old male patient presented with unilateral paracentral acute middle maculopathy (PAMM). Because the patient demonstrated homolateral internal carotid artery stenosis, and two negative CRP tests, the diagnosis of GCA could have been overlooked if diffuse aortitis had not been observed on supraortic CT-scan. The diagnosis was further confirmed with PET scan, demonstrating femoral arteritis in addition to aortitis.

Conclusion: If PAMM and negative CRP are frequently encountered in "cranial GCA," "aortoarteritic GCA" is usually associated with inflammatory signs, which was not the case in this patient, reminding us that a high degree of suspicion should always be maintained in case of PAMM in elderly patients.

Purpose: To assess appeal outcomes and factors influencing those outcomes in insurance-mandated non-medical switching (NMS) from adalimumab (Humira®) to biosimilars in patients with uveitis and ocular inflammatory diseases.

Methods: This retrospective observational study reviewed the electronic medical records of patients receiving Humira® for non-infectious uveitis and ocular inflammatory diseases prior to pharmacy benefit manager (PBM) formulary changes effective April 1, 2024, at the University of Texas Southwestern Medical Center (UTSW) and University of Texas Health Austin (UTHA). Data collected included PBM assignment, treatment duration, remission status, incidence of NMS mandates, appeal outcomes, and final treatment status. Appeals at UTSW cited Texas Senate Bill 680, which allows exemptions from new step-therapy protocols. Logistic regression assessed associations between patient characteristics, PBM, and NMS outcomes.

Results: Of the 75 patients, 30 (40%) received NMS mandates. CVS Caremark patients were more likely to receive mandates for NMS (86.4% vs 20.8%; p < 0.01) and had a lower appeal success rate (14.3% vs 90.9%; p < 0.01) compared to non-CVS Caremark patients. Ultimately, 18 (24%) patients (94% CVS Caremark patients, p < 0.01) were forcibly switched. Median appeal time was 11 days (range 1-35), and processing biosimilar transitions added considerable administrative burden, even in the absence of appeals.

Conclusion: PBM-driven formulary changes disrupted treatment continuity for patients with non-infectious ocular inflammation. Enforcement of NMS mandates varied by PBM and often disregarded clinical stability and physician input, underscoring the urgent need for policy reform and uveitis-specific biosimilar outcome data.

Purpose: To present two rare pediatric cases of bilateral acute occlusive retinal vasculitis (ORV) associated with respiratory viral infections.

Methods: This retrospective review of two cases included a comprehensive clinical evaluation and detailed examinations, including wide-field RetCam color fundus photography, fundus fluorescein angiography, B-ultrasound imaging, aqueous humor cytokine analysis, and laboratory testing for viral infections. Treatments consisted of antivirals, corticosteroids, and anti-VEGF therapy.

Results: Both patients presented with severe vision loss, retinal hemorrhages, retinal vascular occlusion, and exudative detachment. Elevated intraocular VEGF and IL-6 suggested an immune-mediated response. Despite treatment, both had poor visual recovery with optic atrophy and retinal vessel attenuation.

Conclusion: Respiratory viral infections may trigger severe pediatric ORV via immune-mediated inflammation. In these two cases, early diagnosis, viral screening, and prompt treatment were applied, but the prognosis remains poor. Further research is needed to refine management strategies.

Purpose: To describe a rare case of pediatric granulomatosis with polyangiitis (GPA) initially presenting as peripheral ulcerative keratitis (PUK), followed by orbital inflammation, highlighting the diagnostic challenges and importance of early recognition.

Methods: A 12-year-old boy with a prior history of allergic conjunctivitis presented with unilateral peripheral corneal opacities. The condition was initially misdiagnosed and treated as infectious keratitis and allergic conjunctivitis without improvement. Comprehensive clinical, serologic, and imaging evaluations were performed, including ANCA testing, chest and orbital CT scans, and infectious serologies.

Results: Laboratory workup revealed elevated inflammatory markers, positive p-ANCA, and high anti-myeloperoxidase (MPO) antibody titers. Chest CT showed pulmonary nodules consistent with vasculitic involvement. The patient subsequently developed orbital pseudotumor and recurrent epistaxis, consolidating the diagnosis of GPA despite MPO-ANCA positivity, which is more commonly linked to microscopic polyangiitis. Immunosuppressive therapy with systemic corticosteroids and methotrexate led to complete remission of ocular inflammation, resolution of orbital involvement, and preservation of 20/20 visual acuity bilaterally after one year of follow-up.

Conclusion: This case represents an exceptional pediatric presentation of GPA, with PUK as the sole initial manifestation preceding systemic disease. It underscores the need for heightened clinical suspicion in children with atypical peripheral keratitis unresponsive to standard therapy. Early recognition and multidisciplinary management are crucial to prevent irreversible visual loss and systemic complications.

Purpose: To report the first documented case of endophthalmitis caused by Letendraea helminthicola following a penetrating globe injury from a cat scratch.

Methods: A 32-year-old male presented with post-traumatic endophthalmitis after a penetrating ocular injury caused by a cat scratch. Clinical evaluation, serial ophthalmic imaging, microbiological investigations, and polymerase chain reaction (PCR) analysis of conjunctival tissue were performed.

Results: Despite initial improvement with empiric broad-spectrum antibiotics, the patient developed progressive vitritis requiring multiple pars plana vitrectomies. Conventional microbiological cultures remained negative; however, PCR identified L. helminthicola from conjunctival tissue. Targeted antifungal therapy with systemic and intravitreal voriconazole led to infection control, although visual recovery was limited due to macular involvement.

Conclusion: L. helminthicola can cause post-traumatic fungal endophthalmitis following ocular injuries involving animal scratches. Molecular diagnostics are critical for identifying rare pathogens, and prompt targeted antifungal therapy is essential for optimizing outcomes.

Background: Sjögren's syndrome (SS) is a chronic autoimmune disorder primarily affecting exocrine glands, although it may also involve multiple organs, including the eye. The choroid, the most vascularized ocular structure, is affected by systemic diseases. The choroidal vascularity index (CVI), a parameter that reflects vascular damage, decreases as vascular injury increases.

Method: A total of 40 SS patients, aged 18-75 years, and 40 healthy controls were enrolled. Disease activity was assessed using the Sjögren's Syndrome Disease Activity Index (SSDAI); patients with an SSDAI score ≥5 were classified as having active disease, while those with scores <5 were considered in remission. Enhanced depth imaging optical coherence tomography (EDI-OCT) images were analyzed using ImageJ software to measure the luminal area (LA) and total choroidal area (TCA), with CVI calculated as the ratio LA/TCA.

Results: SS patients exhibited significantly lower CVI values (mean: 0.62 ± 0.04) compared to controls (mean: 0.69 ± 0.02; p < 0.001). No significant correlation was found between CVI and SS disease activity as measured by the SSDAI. Additionally, CVI did not significantly differ with respect to gender, age, disease duration, anti-SS-A, anti-SS-B, or rheumatoid factor (RF) positivity. However, CVI was significantly lower in patients with positive antinuclear antibodies. In multivariable analysis, ANA positivity and CRP ≥5 mg/L were independently associated with lower CVI, whereas ESR showed no association with CVI.

Conclusion: Our findings indicate that SS patients exhibit reduced CVI, reflecting ocular vascular involvement, which was not associated with disease activity. As a non-invasive method, CVI measurement may serve as a useful tool for assessing microvascular damage induced by inflammatory processes.