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CATASTROPHIC ANTIPHOSPHOLIPID SYNDROME AND POSTERIOR OCULAR INVOLVEMENT: Case Series of 11 Patients and Literature Review.
Morel, N, Bonnet, C, Mehawej, H, Le Guern, V, Pérard, L, Roumier, M, Brezin, A, Godeau, B, Haroche, J, Benhamou, Y, et al
Retina (Philadelphia, Pa.). 2021;(11):2332-2341
Abstract
PURPOSE To describe the posterior ophthalmic manifestations of catastrophic antiphospholipid syndrome. METHODS Retrospective case series of patients presenting with catastrophic antiphospholipid syndrome and posterior segment ocular manifestations. The main outcomes were the type of posterior segment manifestations at catastrophic antiphospholipid syndrome diagnosis, specifically retinal vascular occlusion, vasculitis, or choroidopathy, and the final best-corrected visual acuity. RESULTS This study included 23 patients (11 cases treated by the authors and 12 published case reports); 21 (91%) of them female. Their median age at diagnosis was 28 years (range, 16-79 years). Ophthalmologic manifestations were usually bilateral (n = 19, 83%) and involved vascular occlusive retinopathy (n = 17, 74%), choroidopathy (n = 11, 48%), or retinal vasculitis (n = 1, 4%). Final best-corrected visual acuity was not significantly worse than the best-corrected visual acuity at diagnosis (P = 0.16). Retinal vascular occlusions were associated with poorer final visual acuity than choroidopathy (P = 0.002). After a median follow-up of 14 months (range, 2-132 months), nearly half the patients (n = 11, 48%) had permanent vision loss including best-corrected visual acuity of <20/400 for 4 patients. CONCLUSION Posterior ophthalmic manifestations of catastrophic antiphospholipid syndrome were mainly bilateral retinal vascular occlusion, which had the worst visual prognosis, followed by choroidopathy and retinal vasculitis. Permanent visual loss was common.
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Unilateral retinitis pigmentosa in children.
Mercado, CL, Pham, BH, Beres, S, Marmor, MF, Lambert, SR
Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus. 2018;(6):457-461.e4
Abstract
BACKGROUND Retinitis pigmentosa (RP) is a group of rare inherited retinal disorders characterized by diffuse progressive degeneration of the retina that typically presents bilaterally. Unilateral RP has not often been reported in children. We present a series of cases that illustrate discrimination between unilateral and asymmetric disease and between dystrophy and acquired degeneration. METHODS Four patients (9-15 years of age; 3 females) were referred to our institution for possible unilateral RP based on fundus appearance and unilateral symptoms. All underwent full-field electroretinography (ERG), spectral domain optical coherence tomography (SD-OCT), widefield and color fundus photography, and fundus autofluorescence (FAF) imaging. Genetic testing and a vitamin and essential fatty acids panel were also conducted in 1 patient. RESULTS Unilateral retinal degeneration was confirmed in 2 patients, whose fellow eyes showed no abnormalities on ERG or imaging. The other 2 patients were found to have highly asymmetric retinal degeneration based on ERG, wide-angle images, and repeated examinations (range, 0.3-9.8 years). Genetic testing and blood testing in 1 unilateral case were negative. CONCLUSIONS Childhood-onset "unilateral RP" remains a difficult and uncertain diagnosis. ERG testing and longitudinal and widefield fundus examination are necessary to exclude asymmetrical disease. Although unilateral degeneration may exist in some children, its inherited or acquired etiology remains poorly understood.
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CASES IN REFINING MANAGEMENT OF DIABETIC MACULAR EDEMA.
Retina (Philadelphia, Pa.). 2018;:1-12
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Use of Handheld Intraoperative Spectral-Domain Optical Coherence Tomography in a Variety of Vitreoretinal Diseases.
Branchini, LA, Gurley, K, Duker, JS, Reichel, E
Ophthalmic surgery, lasers & imaging retina. 2016;(1):49-54
Abstract
BACKGROUND AND OBJECTIVE The use of intraoperative optical coherence tomography (iOCT) has been described with a variety of imaging devices and techniques. The purpose of this investigation is to examine the role of iVue (Optovue, Fremont, CA), a commercially available, handheld spectral-domain iOCT system, in vitreoretinal surgery. PATIENTS AND METHODS For this retrospective, observational case series, patients who underwent a vitreoretinal surgical procedure and were imaged with the iVue were identified. Images were qualitatively assessed. RESULTS Five cases were identified, including an examination under anesthesia, epiretinal membrane, macular hole, retinal detachment, and non-clearing vitreous hemorrhage in the setting of proliferative diabetic retinopathy. CONCLUSION Clinically useful images were obtained in all cases, though it was difficult to center the scan on the area of interest in the retina. Further work is necessary to improve system design and investigate the ways in which iOCT can aid in vitreoretinal surgery.
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Swept-Source Optical Coherence Tomography Angiography of Paediatric Macular Diseases.
Stanga, PE, Papayannis, A, Tsamis, E, Chwiejczak, K, Stringa, F, Jalil, A, Cole, T, Biswas, S
Developments in ophthalmology. 2016;:166-73
Abstract
PURPOSE To describe the optical coherence tomography (OCT) angiography (OCTA) features of paediatric macular pathologies. METHODS Retrospective serial case reports of patients who underwent routine clinical examination and OCTA of the posterior pole using both a DRI OCT Atlantis prototype and Triton Swept-Source OCT. When considered necessary, imaging was performed using Optos wide-field imaging or another non-invasive imaging system. The findings were compared with the current literature. RESULTS Three cases with X-linked retinoschisis, 2 with epiretinal membrane, 1 with Best disease and 2 with Coats disease are fully illustrated. CONCLUSION OCTA is an effective, non-invasive imaging technique that can offer additional information regarding the morphologies and vascular characteristics of macular lesions in paediatric ophthalmology. Because of the rarity and characteristics of many paediatric macular pathologies, further multi-centric research is required with regard to the utilisation and features of OCTA imaging.
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Enhanced depth imaging optical coherence tomography features in a young case of primary hyperoxaluria Type 1.
Özişik, GG, Asena, L, Bulam, B, Güngör, SG
Retinal cases & brief reports. 2015;(1):92-4
Abstract
PURPOSE To describe the enhanced depth imaging optical coherence tomography findings in a very young case of Type 1 primary hyperoxaluria. METHODS Observational case report of a young patient who underwent clinical examination and enhanced depth imaging optical coherence tomography evaluation. RESULTS A 4-year-old boy with a history of Type 1 primary hyperoxaluria and resulting chronic renal failure was referred to us for ophthalmologic examination. There were no ocular symptoms when he was referred to us. Fundus examination showed deposition of calcium oxalate crystals at the posterior pole located symmetrically in both eyes. Enhanced depth imaging optical coherence tomography evaluation revealed hyperreflective structures, localized under the photoreceptor inner segment/outer segment junction, and over the retinal pigment epithelium, consistent with deposition of oxalate crystals. There were no oxalate crystals in the superficial retinal layers, and we observed no evidence of deposition of oxalate crystals in choroid with enhanced depth imaging optical coherence tomography. CONCLUSION We could not demonstrate any oxalate deposits in the choroid with enhanced depth imaging optical coherence tomography in this young case of primary hyperoxaluria Type 1. This may be related to the young age of our patient, and the amounts of the crystalline deposition may increase in the years ahead.