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SAGE Open Medical Case Reports 2024Rosai-Dorfman disease is a rare non-Langerhans cell histiocytosis disorder, also known as sinus histiocytosis, with massive lymphadenopathy. Rosai-Dorfman disease is...
Rosai-Dorfman disease is a rare non-Langerhans cell histiocytosis disorder, also known as sinus histiocytosis, with massive lymphadenopathy. Rosai-Dorfman disease is classified into nodal and extranodal diseases. Ocular involvement is one of the common extranodal manifestations; however, uveitis manifestation is infrequent. We reported an interesting Rosai-Dorfman disease patient who presented with chronic bilateral granulomatous anterior uveitis with disc hyperemia and cystoid macular edema. She was a 60-year-old Thai female who had abnormal skin nodules on her back after multiple recurrent chronic anterior uveitis. The diagnosis has been made based on a constellation of the patient's information, physical examination, and histopathological results. We successfully treated the patient with topical steroids, posterior subtenon triamcinolone 20-mg injection, and methotrexate.
PubMed: 38505579
DOI: 10.1177/2050313X241239528 -
Ophthalmology and Therapy May 2024This study aimed to assess the efficacy and safety of adalimumab in pediatric patients with chronic non-infectious posterior uveitis and panuveitis (not associated with...
INTRODUCTION
This study aimed to assess the efficacy and safety of adalimumab in pediatric patients with chronic non-infectious posterior uveitis and panuveitis (not associated with juvenile idiopathic arthritis).
METHODS
The medical records of children (< 18 years old) with chronic non-infectious posterior uveitis and panuveitis were collected and analyzed in this retrospective cohort study. Children were allocated to a conventional adalimumab-free treatment (CT) or adalimumab (ADA) group based on whether they additionally received adalimumab.
RESULTS
In total, 69 children (138 eyes) were included, with 21 (42 eyes) and 48 (96 eyes) in the CT and ADA groups, respectively. During the average follow-up period of 24 months, the improvement in all ocular parameters (best-corrected visual acuity, intraocular inflammation, fluorescein angiography score) was better in the ADA group than in the CT group, except for changes in central macular thickness, which did not significantly differ between the groups. The mean time of first alleviation, which was after 1.03 ± 0.12 months of therapy, was earlier in the ADA group than in the CT group (2.30 ± 0.46 months). In the ADA group, 90.6% of children had remission within 3 months, and 47.9% had no relapse during follow-up. Cough and cold were the most common adverse events in the ADA group; however, the number of adverse events was similar between both the groups.
CONCLUSIONS
Adalimumab was effective in the treatment of chronic noninfectious posterior uveitis and panuveitis in pediatric patients, and disease inactivity was accomplished in the majority of the patients, thereby improving visual outcomes and maintaining disease stability. Adverse events were limited and tolerable.
PubMed: 38498278
DOI: 10.1007/s40123-024-00884-4 -
Eye (London, England) Jun 2024To evaluate the utility of ultrawide-field fluorescein angiography (UWFFA) in patients with anterior uveitis by investigating the detection of retinal vascular leakage...
PURPOSE
To evaluate the utility of ultrawide-field fluorescein angiography (UWFFA) in patients with anterior uveitis by investigating the detection of retinal vascular leakage (RVL) and the subsequent implications on disease diagnosis and management.
STUDY DESIGN/MATERIALS AND METHODS
Patients, who were referred to the National Eye Institute (NEI) for evaluation of anterior uveitis and underwent UWFFA imaging at the initial visit, were included in this study. The electronic medical records of eligible patients were reviewed. The UWFFA images were assessed for severity of retinal vascular leakage, presence of macular leakage, and optic disc leakage by a two-grader system, and intergrader agreement was calculated using the κ-value. Patients with altered diagnoses and management attributable to UWFFA results were noted.
RESULTS
A total of 93 eyes of 63 patients were included in the study. Of 93 eyes, 31 (33.3%) eyes had RVL on UWFFA, with 26 (28.0%) eyes and 5 (5.4%) eyes showing mild and moderate-severe RVL, respectively. Twenty-five (26.9%) eyes showed macular leakage, and 7 (7.5%) eyes showed optic disc leakage. The κ-values ranged from 0.85 - 0.87 indicating excellent intergrader agreement. Of the 31 eyes with RVL, the diagnosis was changed to anterior/intermediate uveitis for 9 (29.0%) eyes and to panuveitis for 4 (12.9%) eyes. Systemic treatment was escalated in 5 patients.
CONCLUSION
Our results suggest that UWFFA imaging is useful in detecting subclinical posterior involvement in patients with anterior uveitis. Moreover, UWFFA results in altered diagnosis and treatment approaches in a portion of patients.
Topics: Humans; Fluorescein Angiography; Female; Male; Middle Aged; Adult; Uveitis, Anterior; Retrospective Studies; Aged; Retinal Vessels; Young Adult; Capillary Permeability; Adolescent; Retinal Diseases
PubMed: 38472380
DOI: 10.1038/s41433-024-03012-5 -
Indian Journal of Ophthalmology Jul 2024To evaluate the spectrum of uveitis occurring after 60 years of age in elderly patients who presented to a tertiary care eye center in India.
PURPOSE
To evaluate the spectrum of uveitis occurring after 60 years of age in elderly patients who presented to a tertiary care eye center in India.
METHODS
Retrospective study of patients who visited a tertiary eye care institute between January 2010 and July 2020.
RESULT
Eighty-seven patients developed uveitis after 60 years, with only 44.8% having sufficient follow-up documentation and were included in the final analysis. The median age of these patients was 64 (IQR: 62-70) years, and 69% of them were male. Among the identifiable causes of uveitis, infectious uveitis (36%) was the most common and noninfectious uveitis was noted in 23% of patients. The most common subtype of uveitis was anterior uveitis (52%), followed by intermediate uveitis (32%), panuveitis (11%), and posterior uveitis (7%). Tuberculosis (28%) was the most common cause in our cohort, followed by HLA B27 (10%), sarcoid (8%), and Vogt-Koyanagi-Harada disease (5%). In 41% of patients, a definitive diagnosis of uveitis could not be achieved, and the anterior uveitis group had the highest number of undifferentiated uveitis cases. There were no undifferentiated cases of uveitis in the posterior and panuveitis category. The median follow-up period of these patients was 52 (15-91) months and 66% of eyes had recurrence. A statistically significant improvement in vision was seen in anterior uveitis and panuveitis groups, whereas the median visual acuity of the intermediate uveitis group remained stable throughout the follow-up period.
CONCLUSION
Uveitis in the elderly can have a higher recurrence rate; however, the lack of follow-up in these groups of patients is a major challenge.
Topics: Humans; Male; India; Retrospective Studies; Female; Aged; Middle Aged; Uveitis; Follow-Up Studies; Visual Acuity; Incidence
PubMed: 38454869
DOI: 10.4103/IJO.IJO_2788_23 -
Indian Journal of Ophthalmology Jul 2024Acute Zonal Occult Outer Retinopathy (AZOOR) is characterized by sudden visual impairment, often presenting with scotoma and photopsia in young to middle-aged adults,... (Review)
Review
Acute Zonal Occult Outer Retinopathy (AZOOR) is characterized by sudden visual impairment, often presenting with scotoma and photopsia in young to middle-aged adults, showing a female predominance. This condition, distinct from genetic disorders like retinitis pigmentosa, lacks a hereditary basis and exhibits unique fundus changes and imaging features indicative of outer retinal dysfunction. Recent advancements have broadened our understanding, identifying variants like Acute Annular Outer Retinopathy (AAOR) and Multizonal outer retinopathy and retinal pigment epitheliopathy (MORR), each with specific clinical presentations and imaging characteristics. The diagnosis of AZOOR and its variants primarily relies on excluding other conditions through comprehensive evaluation, including imaging and serological testing. Treatment approaches, including the use of corticosteroids and immunosuppressives, remain debated, with some evidence suggesting benefits in the early stages. The prognosis of AZOOR varies, with most patients experiencing stabilization, although complications like choroidal neovascularization may occur, requiring targeted therapy. This manuscript elucidates the complexity of AZOOR, emphasizing the necessity of high clinical suspicion and the role of advanced imaging in diagnosis and management.
Topics: Humans; White Dot Syndromes; Scotoma; Tomography, Optical Coherence; Fluorescein Angiography; Fundus Oculi; Visual Acuity; Visual Fields; Electroretinography
PubMed: 38454854
DOI: 10.4103/IJO.IJO_3228_23 -
Indian Journal of Ophthalmology Jul 2024To compare various ocular thermography parameters in posterior scleritis (PS), choroiditis (choroidal granuloma [CG], Vogt-Koyanagi-Harada [VKH] syndrome), central... (Comparative Study)
Comparative Study Observational Study
PURPOSE
To compare various ocular thermography parameters in posterior scleritis (PS), choroiditis (choroidal granuloma [CG], Vogt-Koyanagi-Harada [VKH] syndrome), central serous chorioretinopathy (CSCR), and healthy controls.
METHODS
This retrospective, observational, comparative study evaluated cases undergoing ocular thermography between April 2017 and October 2023. The study groups included cases of PS, CG, and VKH, while the control group comprised CSCR cases and healthy individuals. Various thermography parameters were assessed, which were as follows: Ocular surface temperature (OST), central corneal temperature (CCT), average scleral temperature (ST), nasal scleral temperature (nST), temperature difference between both the eyes (∆t), and difference between scleral and corneal temperatures (ST - CCT, nST - CCT).
RESULTS
It was found that ∆t was significantly higher in the PS group compared to the CG ( P = 0.005), CSCR ( P = 0.0001), and control groups (dilated control: P =0.006, undilated control: P = 0.0001). ST - CCT and nST - CCT were significantly higher in the undilated control and CSCR groups and significantly lower in the PS group. ST - CCT and nST - CCT showed less difference in the affected eyes compared to contralateral healthy eyes of PS and CG cases. OST, CCT, ST, and nST displayed statistically insignificant differences across all groups.
CONCLUSION
It is advisable to focus on temperature differences between the affected and healthy eyes, or the difference between the central corneal and scleral temperature of the affected eye, utilizing parameters such as ∆t, ST - CCT, and nST - CCT. These composite parameters offer a more effective approach than individual measurements like OST, CCT, ST, and nST. Thermography can serve as a screening tool to suspect and differentiate PS.
Topics: Humans; Scleritis; Retrospective Studies; Thermography; Female; Male; Adult; Middle Aged; Sclera; Choroiditis; Body Temperature
PubMed: 38454839
DOI: 10.4103/IJO.IJO_2830_23 -
BMC Ophthalmology Mar 2024To report a case of unusual presentation of retinochoroiditis caused by Rickettsia typhi in a patient without prior uveitis.
BACKGROUND
To report a case of unusual presentation of retinochoroiditis caused by Rickettsia typhi in a patient without prior uveitis.
CASE PRESENTATION
In this case, we describe a 24-year-old male soldier with no previous eye disease, who was referred to our ophthalmology department due to bilateral retinochoroiditis and vitritis. The patient initially presented with a paracentral scotoma in his right eye persisting for 7 days and scattered dark spots in his left eye for 2 days in June 2023. Preceding these ocular symptoms, he experienced a two-week episode of fever, headaches, night sweats, and rapid weight loss of 10 kg. A transient rash covered his body briefly. His mother had a history of recurrent eye inflammation. Physical examination revealed bilateral keratic precipitates on the lower corneal periphery, 1 + anterior vitreous cells, small retinal lesions and mild optic discs elevation. Fluorescein angiography indicated mild discs hyperfluorescence, and the clinically visible round punctate lesions on OCT showed inner retinal hyper-reflective lesion with a depth till outer plexiform layer possibly suggestive of a retinitis lesion. Laboratory tests were normal except thrombocytosis, elevated ESR, liver enzymes and ACE levels, with positive Rickettsia typhi serology tests. Rheumatology and infectious disease consultations ruled out autoimmune diseases, confirming Rickettsia typhi infection. Treatment included systemic doxycycline and prednisone, with improvement of visual acuity, ocular symptoms, OCT abnormalities and resolution of inflammation. Prednisone was discontinued, and after two months, additional improvement was seen clinically, with preserved retinal structures on OCT.
CONCLUSION
This study explores retinochoroiditis as a rare ocular presentation of Rickettsia typhi, an unusual infection in the Middle East. Previously reported ocular manifestations include conjunctivitis, vitritis, post infectious optic neuropathy and a few cases of uveitis. Ocular symptoms followed systemic illness, highlighting the need for awareness among clinicians. Diagnosis relies on seroconversion, with fluorescein angiography and OCT aiding in assessment. Empiric doxycycline and systemic corticosteroid therapy is recommended. Ocular symptoms resolved in two months. Awareness of these ocular manifestations is essential for timely diagnosis and management. Further research is needed to fully understand this aspect of murine typhus.
Topics: Humans; Male; Young Adult; Chorioretinitis; Doxycycline; Inflammation; Prednisone; Typhus, Endemic Flea-Borne
PubMed: 38454387
DOI: 10.1186/s12886-024-03329-5 -
Middle East African Journal of... 2023The purpose was to study the anatomical and functional outcome following single low-dose suprachoroidal triamcinolone acetonide (LD-SCTA) (2 mg) injection in...
PURPOSE
The purpose was to study the anatomical and functional outcome following single low-dose suprachoroidal triamcinolone acetonide (LD-SCTA) (2 mg) injection in noninfectious posterior uveitis.
METHODS
Eleven patients with macular edema (ME) more than 280 μ secondary to noninfectious uveitis were included in the study. A single LD-SCTA (0.5 ml) injection was performed in the study eye with the help of a novel suprachoroidal microneedle (Pricon, Iscon Surgicals, Jodhpur, Rajasthan, India). The study parameters were noted at 4 and 12 weeks post LD-SCTA injection.
RESULTS
Ten of 11 patients had a significant decrease in central macular thickness (CMT). The mean CMT measurement at baseline was 513.6 ± 191.73 μm for the 10 patients who responded to the treatment, which reduced significantly to 265.1 ± 34.72 μm ( < 0.003) and 260.6 ± 34.72 μm ( < 0.002) at 4 and 12 weeks, respectively. The mean best-corrected visual acuity (BCVA) at baseline was 0.84 ± 0.41 logMAR unit which improved to 0.52 ± 0.33 ( < 0.001) and 0.25 ± 0.22 ( < 0.000) at weeks 4 and 12, respectively. The mean intraocular pressure at baseline recorded was 16.36 ± 2.97 mmHg, 19.45 ± 4.80 mmHg ( = 0.06) at 4 weeks, and 17.27 ± 2.53 mmHg ( = 0.35) at 12 weeks. One eye which did not respond to LD-SCTA was a case of recurrent Vogt-Koyanagi-Harada disease.
CONCLUSION
Single LD-SCTA injection is efficacious in reducing CMT in ME, improving BCVA, and controlling the inflammation in noninfectious posterior uveitis. LD-SCTA can be used as a first-line therapy in noninfectious uveitis over other routes of steroid administration with a favorable outcome and safety profile.
Topics: Humans; Triamcinolone Acetonide; Macular Edema; India; Uveitis, Posterior; Uveomeningoencephalitic Syndrome
PubMed: 38435103
DOI: 10.4103/meajo.meajo_78_21 -
Turkish Journal of Ophthalmology Feb 2024To report the ocular findings, laboratory results, and management of patients with tubulointerstitial nephritis and uveitis syndrome (TINU), whose numbers increased...
OBJECTIVES
To report the ocular findings, laboratory results, and management of patients with tubulointerstitial nephritis and uveitis syndrome (TINU), whose numbers increased during the 2019 coronavirus disease (COVID-19) pandemic.
MATERIALS AND METHODS
Demographic characteristics, ophthalmic examination findings, laboratory results including polymerase chain reaction (PCR) test for severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), serum SARS-CoV-2 immunoglobulin G (IgG) antibody, and treatment of patients diagnosed with TINU between March 2020 and March 2022 were evaluated retrospectively.
RESULTS
The study included 19 eyes of 10 patients (6 female/4 male). The mean age was 13.5±2.4 years (range: 8-16 years). The mean follow-up duration was 13.5±6.1 months (range: 6-24 months). All patients presented with anterior uveitis. Anterior uveitis was bilateral in 9 patients (90%) and unilateral in 1 patient (10%). Posterior segment findings were normal in 8 patients (80%), and bilateral optic disc edema was observed in only 2 patients (20%). None of the patients had a previous SARS-CoV-2 infection and/or vaccination history. The SARS-CoV-2 PCR test was negative in all patients at presentation. The SARS-CoV-2 IgG antibody test was reactive in 7 patients (70%). Recurrent uveitis developed in 8 patients (80%) during follow-up. Systemic immunomodulatory therapy was required for the control of ocular inflammation in 7 patients (70%) with severe uveitis flare-ups.
CONCLUSION
TINU is a multisystemic autoimmune disease, especially in response to environmental triggering factors such as viral infections. Although TINU is a rare disease, the number of cases increased during the COVID-19 pandemic. SARS-CoV-2 antibodies were detected at a significant rate of 70% in these patients, who did not have a history of SARS-CoV-2 infection and vaccination. Previous asymptomatic SARS-CoV-2 infection in children may be a triggering factor in the development of TINU.
Topics: Child; Humans; Male; Female; Adolescent; Pandemics; Retrospective Studies; COVID-19; SARS-CoV-2; Uveitis; Uveitis, Anterior; Nephritis, Interstitial
PubMed: 38385308
DOI: 10.4274/tjo.galenos.2023.24280