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1.
Graefes Arch Clin Exp Ophthalmol ; 260(5): 1543-1550, 2022 May.
Artículo en Inglés | MEDLINE | ID: mdl-35001204

RESUMEN

PURPOSE: To investigate real-world safety and efficacy of voretigene neparvovec gene therapy administration in pediatric patients with biallelic RPE65 disease-causing variants. METHODS: A retrospective study of 27 eyes of 14 patients with RPE65-associated Leber congenital amaurosis examined postoperative complications and longitudinal changes in photoreceptor function following treatment with subretinal injection of voretigene neparvovec. Full-field stimulus threshold testing (FST), Goldmann visual fields (GVF), best-corrected visual acuity (BCVA), and central subfield thickness (CST) on optical coherence tomography (OCT) scans were collected preoperatively and up to 12 months posttreatment. RESULTS: Baseline through 6-12 month follow-up FST and GVF data were obtained for 13 eyes of 7 patients. FST improved for each eye after treatment with a mean improvement of 2.1 log-units (P < 0.001) and GVF improved for each eye with a mean improvement of 221 sum degrees (P < 0.001). BCVA improved from logMAR 0.98 at baseline to logMAR 0.83 at last follow-up (P < 0.001). Across 19 eyes of 10 patients included in CST analysis, there was a small but statistically significant 9-µ decrease in mean CST from baseline to last follow-up (P < 0.001). The most common postoperative issues included elevation in intraocular pressure (59%), persistent intraocular inflammation (15%), and vitreous opacities (26%) that resolved over a period of months. CONCLUSIONS: This report provides some of the earliest longitudinal real-world evidence of the pediatric safety and efficacy of voretigene neparvovec using multiple functional and structural measures of the retina. Outcomes demonstrate significant improvements in visual function consistent with clinical trial results.


Asunto(s)
Amaurosis Congénita de Leber , Niño , Humanos , Amaurosis Congénita de Leber/diagnóstico , Amaurosis Congénita de Leber/genética , Amaurosis Congénita de Leber/terapia , Mutación , Retina , Estudios Retrospectivos , Tomografía de Coherencia Óptica , Agudeza Visual , cis-trans-Isomerasas/genética
2.
Exp Eye Res ; 151: 179-89, 2016 10.
Artículo en Inglés | MEDLINE | ID: mdl-27593915

RESUMEN

Mutations of claudin-19 cause severe ocular deficits that are not easily reconciled with its role in regulating the outer blood retinal barrier. ARPE-19 is a widely used culture model of the retinal pigment epithelium (RPE). ARPE-19 is unique among epithelial cell lines, because it expresses all tight junction proteins except claudin family members. ARPE-19 also loses aspects of the RPE phenotype with cell passage. This study asks whether exogenous expression of the main RPE claudins, claudin-3 and claudin-19, would restore RPE phenotype, and whether these claudins have distinct roles in RPE. An Ussing chamber was used to measure the transepithelial electrical resistance and transepithelial electrical potential. These measurements were used to estimate the permeability co-efficients of ions. The transepithelial diffusion of polyethylene glycols were used to examine the leak pathway of tight junctions. Wound-healing, quantitative RT-PCR and immunoblotting examined diverse aspects of the RPE phenotype. Over-expression of either claudin decreased the permeability of small ions and polyethylene glycol. Both claudins were slightly cation-specific, but claudin-3 was less permeable to large solutes. Claudin expression widely affected gene expression to partially restore RPE phenotype. Claudins redistributed filamentous actin from stress fibers to circumferential bands associated with tight junctions, and made wound-healing more epithelial-like. Both claudins increased the expression of genes related to RPE core functions and increased steady-state levels of phosphorylated-AKT. In conclusion, claudin-3 and claudin-19 formed general permeability barriers and affected cell morphology, proliferation, migration, AKT signaling, and gene expression. When claudins are exogenously expressed, ARPE-19 more closely model native RPE.


Asunto(s)
Claudina-3/genética , Claudinas/genética , Regulación de la Expresión Génica , ARN Mensajero/genética , Enfermedades de la Retina/genética , Epitelio Pigmentado de la Retina/metabolismo , Uniones Estrechas/genética , Células Cultivadas , Claudina-3/biosíntesis , Claudinas/biosíntesis , Humanos , Immunoblotting , Microscopía Confocal , Fenotipo , Enfermedades de la Retina/metabolismo , Enfermedades de la Retina/patología , Epitelio Pigmentado de la Retina/citología , Reacción en Cadena de la Polimerasa de Transcriptasa Inversa , Uniones Estrechas/metabolismo
3.
Retin Cases Brief Rep ; 18(1): 98-100, 2024 Jan 01.
Artículo en Inglés | MEDLINE | ID: mdl-35963010

RESUMEN

BACKGROUND/PURPOSE: Retinal detachment has previously been reported in association with topical miotic use for the treatment of glaucoma. Pilocarpine hydrochloride 1.25% was recently approved by the Food and Drug Administration for the treatment of presbyopia, with no reports of associated retinal detachments in the clinical trial data. METHODS: Case report. RESULTS: Two novel cases of unilateral retinal detachment occurring within 10 days of the initiation of pilocarpine 1.25% for the treatment of presbyopia were described. The patients were pseudophakic men in their 60s or 70s with preexisting retinal detachment risk factors, such as high myopia, lattice degeneration, and prior retinal detachment. Both affected eyes were treated with pars plana vitrectomy and gas endotamponade with an uncomplicated postoperative course. CONCLUSION: Retinal detachment may be associated with the use of pilocarpine 1.25%. Caution should be used when considering prescribing this medication in patients with preexisting retinal abnormality.


Asunto(s)
Presbiopía , Desprendimiento de Retina , Masculino , Humanos , Desprendimiento de Retina/inducido químicamente , Desprendimiento de Retina/cirugía , Pilocarpina/efectos adversos , Presbiopía/complicaciones , Presbiopía/cirugía , Agudeza Visual , Vitrectomía/efectos adversos , Soluciones Oftálmicas , Resultado del Tratamiento , Estudios Retrospectivos
4.
Ophthalmic Genet ; 44(6): 539-546, 2023 Dec.
Artículo en Inglés | MEDLINE | ID: mdl-37381907

RESUMEN

BACKGROUND: To determine the effect of lesion topography on progression in Stargardt disease (STGD1). METHODS: Fundus autofluoresence (excitation 488 nm) images of 193 eyes in patients with proven ABCA4 mutation were semi-automatically segmented for autofluoresence changes: (DDAF) and questionably decreased autofluoresence (QDAF), which are proxies for retinal pigment epithelial (RPE) atrophy. We calculated topographic incidence of DDAF and DDAF + QDAF, as well as velocity of progression of the border of lesions using Euclidean distance mapping. RESULTS: Incidence of atrophy was highest near the fovea, then decreased in incidence with increased foveal eccentricity. However, the rate of atrophy progression followed the opposite pattern; rate of atrophy increased with distance from foveal center. The mean growth rate 500 microns from the foveal center for DDAF + QDAF was 39 microns per year (95% CI = 28-49), whereas the mean growth rate 3000 microns from the foveal center was 342 microns per year (95% CI = 194-522). No difference in growth rate was noted by axis around the fovea. CONCLUSIONS: Incidence and progression of atrophy by fundus autofluorescence follow opposite patterns in STGD1. Further, atrophy progression increases significantly with distance from foveal center, which should be taken into consideration in clinical trials.


Asunto(s)
Degeneración Macular , Epitelio Pigmentado de la Retina , Humanos , Enfermedad de Stargardt/patología , Progresión de la Enfermedad , Epitelio Pigmentado de la Retina/patología , Tomografía de Coherencia Óptica/métodos , Angiografía con Fluoresceína/métodos , Agudeza Visual , Degeneración Macular/diagnóstico , Degeneración Macular/genética , Fondo de Ojo , Atrofia/complicaciones , Transportadoras de Casetes de Unión a ATP/genética
5.
Sci Rep ; 13(1): 9165, 2023 06 06.
Artículo en Inglés | MEDLINE | ID: mdl-37280345

RESUMEN

Diabetic retinopathy is a leading cause of blindness in working-age adults worldwide. Neovascular leakage on fluorescein angiography indicates progression to the proliferative stage of diabetic retinopathy, which is an important distinction that requires timely ophthalmic intervention with laser or intravitreal injection treatment to reduce the risk of severe, permanent vision loss. In this study, we developed a deep learning algorithm to detect neovascular leakage on ultra-widefield fluorescein angiography images obtained from patients with diabetic retinopathy. The algorithm, an ensemble of three convolutional neural networks, was able to accurately classify neovascular leakage and distinguish this disease marker from other angiographic disease features. With additional real-world validation and testing, our algorithm could facilitate identification of neovascular leakage in the clinical setting, allowing timely intervention to reduce the burden of blinding diabetic eye disease.


Asunto(s)
Aprendizaje Profundo , Diabetes Mellitus , Retinopatía Diabética , Adulto , Humanos , Angiografía con Fluoresceína/métodos , Retinopatía Diabética/diagnóstico por imagen , Ojo , Ceguera
6.
Am J Ophthalmol ; 234: 15-19, 2022 02.
Artículo en Inglés | MEDLINE | ID: mdl-34343488

RESUMEN

PURPOSE: To present select cases of paracentral acute middle maculopathy (PAMM) demonstrating progressive vascular occlusion and the ischemic cascade and illustrate scenarios in which therapeutic intervention may be considered. DESIGN: Perspective. METHODS: Review and interpretation of selected literature, with perspective on the evaluation and management of patients with PAMM lesions. Multimodal imaging of 3 illustrative cases of PAMM are presented, including cross-sectional and en face optical coherence tomography (OCT), with progressive vascular occlusion and evidence of the ischemic cascade noted in 2 of the cases. RESULTS: All 3 cases showed evidence of PAMM at baseline, including perivenular PAMM in 2 cases. Progression from a mild central retinal vein occlusion (CRVO) to a more severe CRVO was noted in the first case, while the second case progressed from an incomplete to complete central retinal artery occlusion (CRAO). In the third case, there was resolution of PAMM lesions associated with partial CRAO after the patient began taking aspirin. CONCLUSIONS: Perivenular PAMM can be the only presenting sign of retinal vascular occlusion. In such cases, it is important to differentiate between partial CRVO and incomplete CRAO as the underlying cause. We provide guidance on making this important distinction, which plays a critical role in directing the nature and urgency of the systemic work-up. In patients with PAMM caused by partial retinal arterial occlusion, a benign empirical approach, such as low-dose aspirin (81 mg), may be reasonable to reduce the likelihood of subsequent vision loss from progression to complete arterial occlusion. A clinical trial, however, is necessary to support such a strategy.


Asunto(s)
Degeneración Macular , Enfermedades de la Retina , Estudios Transversales , Angiografía con Fluoresceína/métodos , Humanos , Degeneración Macular/complicaciones , Enfermedades de la Retina/diagnóstico , Enfermedades de la Retina/etiología , Enfermedades de la Retina/terapia , Vasos Retinianos/patología , Tomografía de Coherencia Óptica/métodos , Agudeza Visual
7.
Ophthalmol Retina ; 6(11): 1098-1104, 2022 11.
Artículo en Inglés | MEDLINE | ID: mdl-35644472

RESUMEN

OBJECTIVE: To train a deep learning (DL) algorithm to perform fully automated semantic segmentation of multiple autofluorescence lesion types in Stargardt disease. DESIGN: Cross-sectional study with retrospective imaging data. SUBJECTS: The study included 193 images from 193 eyes of 97 patients with Stargardt disease. METHODS: Fundus autofluorescence images obtained from patient visits between 2013 and 2020 were annotated with ground-truth labels. Model training and evaluation were performed using fivefold cross-validation. MAIN OUTCOMES MEASURES: Dice similarity coefficients, intraclass correlation coefficients, and Bland-Altman analyses comparing algorithm-predicted and grader-labeled segmentations. RESULTS: The overall Dice similarity coefficient across all lesion classes was 0.78 (95% confidence interval [CI], 0.69-0.86). Dice coefficients were 0.90 (95% CI, 0.85-0.94) for areas of definitely decreased autofluorescence (DDAF), 0.55 (95% CI, 0.35-0.76) for areas of questionably decreased autofluorescence (QDAF), and 0.88 (95% CI, 0.73-1.00) for areas of abnormal background autofluorescence (ABAF). Intraclass correlation coefficients comparing the ground-truth and automated methods were 0.997 (95% CI, 0.996-0.998) for DDAF, 0.863 (95% CI, 0.823-0.895) for QDAF, and 0.974 (95% CI, 0.966-0.980) for ABAF. CONCLUSIONS: A DL algorithm performed accurate segmentation of autofluorescence lesions in Stargardt disease, demonstrating the feasibility of fully automated segmentation as an alternative to manual or semiautomated labeling methods.


Asunto(s)
Algoritmos , Imagen Óptica , Humanos , Enfermedad de Stargardt , Estudios Retrospectivos , Estudios Transversales
8.
Retin Cases Brief Rep ; 16(1): 36-39, 2022 Jan 01.
Artículo en Inglés | MEDLINE | ID: mdl-32150117

RESUMEN

BACKGROUND/PURPOSE: To report a case of serology-negative severe disseminated Bartonella neuroretinitis in an immunocompromised patient in which diagnosis was made by detection of B. henselae DNA by universal polymerase chain reaction of brain tissue. METHODS: Case report. RESULTS: A 57-year-old man with immunoglobulin A vasculitis on immunosuppressive therapy presented with lethargy, weight loss, and bilateral decreased vision. Fundus examination revealed bilateral mild vitritis, marked optic disc edema, vascular sheathing, and numerous white inner retinal and preretinal lesions. Brain magnetic resonance imaging revealed multiple foci of restricted diffusion and a ring-enhancing focus in the left parietal lobe. Serologies, cerebrospinal fluid, and vitreous biopsies were all negative for Bartonella. A brain biopsy was performed and B. henselae DNA was detected by universal polymerase chain reaction of the specimen. The patient demonstrated resolution of fundus findings with antibiotic treatment. Repeat serological testing demonstrated seroconversion. CONCLUSION: In immunocompromised patients, infection by Bartonella henselae can present as severe disseminated disease. Establishing the diagnosis can be challenging as serologic testing is often unrevealing in the setting of a blunted immune response. Polymerase chain reaction has been used in select cases to establish the diagnosis.


Asunto(s)
Bartonella henselae , Enfermedad por Rasguño de Gato , Huésped Inmunocomprometido , Bartonella henselae/genética , Bartonella henselae/aislamiento & purificación , Enfermedad por Rasguño de Gato/diagnóstico , ADN Bacteriano/aislamiento & purificación , Humanos , Masculino , Persona de Mediana Edad
9.
Clin Ophthalmol ; 16: 1401-1411, 2022.
Artículo en Inglés | MEDLINE | ID: mdl-35535124

RESUMEN

Purpose: Ocular trauma with intraocular foreign body (IOFB) can have devastating visual consequences. Management and antimicrobial strategies remain variable due to the infrequency and heterogeneity of presentation. Our goal was to identify risk factors for endophthalmitis and poor visual outcomes in cases of IOFB and investigate management strategies. Patients and Methods: A retrospective chart review was conducted in 88 eyes of 88 patients suffering traumatic injury with IOFB at the University of Michigan between January 2000 and December 2019. Medical records were reviewed to characterize the injuries and IOFBs as well as how clinical presentation and treatment modalities were associated with outcomes. Results: Delayed presentation (P=0.016) and organic IOFB (P=0.044) were associated with development of endophthalmitis. Retinal detachment (P=0.012), wound length greater than 5 mm (P=0.041), and poor presenting visual acuity (P=0.003) correlated with poor final visual outcome. Antibiotic prophylaxis was given to all patients, though agents and routes of delivery varied. Endophthalmitis developed in 4.9% of the eyes after initial management, with primary and secondary removal of posterior segment IOFBs associated with similar rates of endophthalmitis (P=1.000). Conclusion: Poor presenting visual acuity and severity of injury, as measured by large wound and retinal detachment, correlate with poor visual outcome. Prompt globe closure and antimicrobial prophylaxis are critical for infection prevention. In cases where IOFB removal and globe closure cannot be performed concurrently, primary globe closure with aggressive antibiotic prophylaxis offers a reasonable alternative to prevent endophthalmitis.

10.
Clin Ophthalmol ; 16: 3339-3350, 2022.
Artículo en Inglés | MEDLINE | ID: mdl-36237492

RESUMEN

Purpose: At the time of open globe injury (OGI), it may be difficult for clinicians to predict which eyes are at highest risk for requiring enucleation. We performed a 17-year retrospective cohort study to report outcomes and risk factors for enucleation following open globe injuryto better aid clinicians counseling patients at OGI diagnosis. Methods: A retrospective cohort study of all patients who presented to the University of Michigan with open globe injury (OGI) and were surgically managed between January 2000 and July 2017 was conducted. At least 30 days of follow-up was required. All eyes that ultimately underwent enucleation following OGI were identified and their clinical course analyzed. The main outcome measured was the rate of enucleation after OGI. Results: There were 587 eyes meeting inclusion criteria. The mean patient age was 40.75 ± 25.1 (range 1-91). 441/585 (75.4%) patients were male. Average follow-up time was 1029.9 ± 1285.9 days. 116/587 eyes (19.8%) required enucleation after OGI, with 81.9% undergoing enucleation less than 30 days from injury. In enucleated eyes, the mean presenting logMAR vision was 2.91 ± 0.47 (Snellen equivalent between hand motion and light perception). The most common mechanism of injury requiring enucleation was globe rupture, 89/116 (76.7%), with 14/116 (12.1%) penetrating injuries and 13/116 (11.2%) perforating injuries. The mean age of patients that underwent enucleation was 45.6 ± 22.5 (range 3-91). Conclusion: Open globe injuries are often visually devastating and a significant number of cases ultimately require enucleation. Despite emergent closure within 24 hours, 19.8% of eyes managed for OGI at our institution required eventual enucleation. 81.2% of these eyes required enucleation within 30 days of injury. Wound length greater than 10 mm, uveal prolapse, higher zone of injury, IOFB, and RAPD were identified as risk factors that predict future need for enucleation.

11.
Cornea ; 41(11): 1345-1352, 2022 Nov 01.
Artículo en Inglés | MEDLINE | ID: mdl-34759204

RESUMEN

PURPOSE: The purpose of this study was to investigate the clinical features, surgical outcomes, and prognostic factors of penetrating keratoplasty (PKP) after open globe injury (OGI). METHODS: A retrospective review of all patients treated for OGI between January 2000 and July 2017 was conducted. Demographic, preoperative, perioperative, and postoperative data were collected for those who underwent PKP after OGI. The predictive value of each preoperative variable on graft failure was assessed using univariate and multivariable Cox proportional hazards models, and the predictive value of variables on post-PKP visual outcome was assessed using both univariate and multivariable logistic regression models. All eyes that underwent PKP after OGI were included unless they had less than 365 days of follow-up. RESULTS: Forty-six eyes that underwent PKP met inclusion criteria. The median age was 46 years (interquartile range = 23.00-61.25), median follow-up was 78.5 months (interquartile range = 38.63-122.02), and 37 of 46 subjects (80.4%) were male. The observed 1- and 5-year graft survival estimates were 80.4% and 41.7%, respectively. Factors statistically associated with graft failure in multivariable analyses were rejection episode, hazard ratio (HR) = 3.29; retinal detachment (RD), HR = 3.47; and endophthalmitis, HR = 6.27. Fifteen of 42 eyes (35.7%) regained ambulatory vision (20/200 or better). The strongest predictors of vision worse than 20/200 at the last follow-up were RD, odds ratio (OR) = 43.88; graft rejection, OR = 12.42; and injury outside the workplace, OR = 25.05. CONCLUSIONS: Despite a high graft survival at 1 year, most of the patients did not regain ambulatory vision. Graft rejection, RD, and endophthalmitis were risk factors for graft failure. These factors should be considered when counseling patients regarding PKP after OGI.


Asunto(s)
Endoftalmitis , Lesiones Oculares , Desprendimiento de Retina , Endoftalmitis/etiología , Lesiones Oculares/etiología , Femenino , Supervivencia de Injerto , Humanos , Queratoplastia Penetrante/efectos adversos , Masculino , Persona de Mediana Edad , Estudios Retrospectivos , Resultado del Tratamiento , Agudeza Visual
12.
JAMA Ophthalmol ; 139(4): 449-455, 2021 Apr 01.
Artículo en Inglés | MEDLINE | ID: mdl-33662097

RESUMEN

IMPORTANCE: The benefits of no-cost genetic testing initiatives have not been characterized. The no-cost My Retina Tracker Genetic Testing Study (MRT-GTS) research registry for inherited retinal degenerations (IRDs) was launched in 2017 in the US. OBJECTIVE: To investigate the associations of MRT-GTS implementation and patient characteristics with access to genetic testing for IRDs. DESIGN, SETTING, AND PARTICIPANTS: In a cross-sectional design, analysis of new patients evaluated 12 months before (July 1, 2016, to June 13, 2017) and 12 months after (June 14, 2017, to June 30, 2018) MRT-GTS implementation at a single academic referral eye center was conducted. Participants included 369 patients with IRD. Data analysis was conducted from February to June 2020. MAIN OUTCOMES AND MEASURES: Change in rates of successfully obtaining genetic testing, odds ratios (ORs) of association between patient characteristics and obtaining testing, and days elapsed from clinic visit to reporting of results. RESULTS: Among 369 patients (mean [SD] age, 39.5 [20.8] years; 193 [52.3%] women), 144 were evaluated in the pre-MRT-GTS period and 225 in the post-MRT-GTS period. The baseline rate of successfully obtaining testing was 51.4% (95% CI, 42.6%-60.2%). The initiation of MRT-GTS was associated with a 28.9-percentage point increase in testing rate (95% CI, 16.7%-41.1%; P < .001). Patient characteristics that increased the odds of obtaining testing were eligibility for MRT-GTS (OR, 14.15; 95% CI, 7.36-27.24; P < .001) and worse visual acuity (logMAR +1.0; Snellen equivalent decrease from 20/20 to 20/200) in the better-seeing eye (OR, 1.92; 95% CI, 1.27-2.91; P < .01). Patients had decreased odds when identifying as Black or African American (OR, 0.10; 95% CI, 0.04-0.24; P < .001) or other race (OR, 0.37; 95% CI, 0.15-0.91; P = .03) compared with White race, and when the primary language was not English (OR, 0.13; 95% CI, 0.03-0.55; P < .01). The proportion of test results reported within 90 days was 81.5% (95% CI, 74.8%-86.4%) when eligible for MRT-GTS compared with 48.1% (95% CI, 35.6%-58.1%) when not eligible (P < .001). CONCLUSIONS AND RELEVANCE: In this study, the implementation of MRT-GTS was associated with an increase in the proportion of patients who successfully obtained testing, suggesting the potential clinical value of this approach. Patient-level demographic and clinical factors appear to be associated with decisions to pursue testing.


Asunto(s)
Pruebas Genéticas , Degeneración Retiniana , Adulto , Femenino , Humanos , Masculino , Estudios Transversales , Degeneración Retiniana/diagnóstico , Degeneración Retiniana/genética , Persona de Mediana Edad
13.
Clin Ophthalmol ; 15: 2077-2087, 2021.
Artículo en Inglés | MEDLINE | ID: mdl-34040343

RESUMEN

BACKGROUND/AIMS: To determine the rate of endophthalmitis and assess risk factors for development of endophthalmitis following open globe injury (OGI). METHODS: A retrospective chart review of all patients treated for OGI at the University of Michigan from January 2000 to July 2017 was conducted. Exclusion criteria included intravitreal injection or intraocular surgery in the 30 days prior to injury or less than 30 days of follow-up. A total of 586 out of 993 open globe injuries were included in the study. The main outcome measure was the rate of endophthalmitis. RESULTS: In this study, 25/586 eyes (4.3%) had endophthalmitis. Of these, 12/25 eyes (48.0%) presented with endophthalmitis and 13/25 eyes (52.0%) developed endophthalmitis after globe closure. Multivariate analysis identified time to globe repair (OR 4.5, CI 1.9-10.7, p = 0.0008), zone I injury (OR 3.6, CI 1.1-11.0, p = 0.0282), and need for additional surgery (OR 5.5, CI 1.5-19.7, p = 0.0092) as factors associated with increased risk of developing endophthalmitis. Subconjunctival antibiotic injection at the time of globe closure (OR 0.3, CI 0.1-0.7, p = 0.0036) was associated with decreased risk of developing endophthalmitis. CONCLUSION: Prompt globe closure and subconjunctival antibiotics may reduce the risk of endophthalmitis in OGI. Furthermore, our practice of a one-time dose of systemic prophylactic antibiotics, and intravitreal antibiotics if intraocular foreign body (IOFB) removal is delayed, was not found to increase the rate of endophthalmitis.

14.
Stem Cell Reports ; 16(10): 2415-2421, 2021 10 12.
Artículo en Inglés | MEDLINE | ID: mdl-34597599

RESUMEN

A wide variety of stem cell-derived therapies are under development for the treatment of retinal degeneration. In order to better understand patient perspectives about these therapies, we assessed risk tolerance using an in-person survey of 178 patients at an academic eye center. Risk of malignancy served as a hypothetical, readily understood, and serious adverse event to be considered in trade for potential visual improvement from a stem cell-derived treatment. The results indicate that patients were willing to trade visual improvement against a risk of malignancy that far exceeds actual risk. Two novel findings were that older patients and those with an intermediate level of visual loss were particularly risk tolerant. The quantitative survey results provide a step toward understanding patient perspectives that will, over the long term, guide the development of ocular stem cell-derived therapies.


Asunto(s)
Prioridad del Paciente , Percepción , Degeneración Retiniana/terapia , Trasplante de Células Madre , Anciano , Anciano de 80 o más Años , Tratamiento Basado en Trasplante de Células y Tejidos/métodos , Femenino , Humanos , Masculino , Persona de Mediana Edad , Pacientes , Factores de Riesgo , Encuestas y Cuestionarios
15.
Ophthalmic Genet ; 41(3): 275-278, 2020 06.
Artículo en Inglés | MEDLINE | ID: mdl-32400255

RESUMEN

BACKGROUND: X-linked retinitis pigmentosa (XLRP) is a hereditary retinopathy that may present with cystoid macular edema (CME). The exact cause of CME in XLRP is unknown. We describe a case report of new-onset CME precipitated by travel to high altitude in an adult with XLRP, but no known prior history of CME. CASE DESCRIPTION: A 38-year-old man with XLRP caused by a hemizygous pathogenic variant in RPGR (c.372del; p.Glu125fs) reported sudden onset bilateral blurry vision 4 days after ascending to an altitude of 3,700 m. He sought local ophthalmic care and was found to have severe bilateral CME. He was treated with topical and oral carbonic anhydrase inhibition and instructed to return to normal altitude. Follow-up imaging at normal altitude revealed that the CME was nearly completely resolved 4 days after initial presentation, and completely resolved 2 weeks after initial presentation. CONCLUSION: Vascular and metabolic changes caused by retinal degeneration in XLRP may predispose to the development of CME under the hypoxic conditions experienced at high altitudes. We advise that retinal specialists treating patients with RP should caution them on traveling to high altitudes that could precipitate or exacerbate CME.


Asunto(s)
Altitud , Enfermedades Genéticas Ligadas al Cromosoma X/complicaciones , Edema Macular/etiología , Retinitis Pigmentosa/complicaciones , Adulto , Humanos , Edema Macular/patología , Masculino , Pronóstico
16.
JAMA Ophthalmol ; 142(4): 375-376, 2024 Apr 01.
Artículo en Inglés | MEDLINE | ID: mdl-38386327
18.
JAMA Ophthalmol ; 141(1): 102-104, 2023 01 01.
Artículo en Inglés | MEDLINE | ID: mdl-36480192

RESUMEN

This case report describes the development of posterior subcapsular cataract and retinal neovascularization in a patient with posterior polar annular choroidal dystrophy.


Asunto(s)
Enfermedades de la Coroides , Neovascularización Coroidal , Neovascularización Retiniana , Humanos , Neovascularización Retiniana/diagnóstico , Enfermedades de la Coroides/diagnóstico , Neovascularización Coroidal/diagnóstico , Angiografía con Fluoresceína
19.
JAMA Ophthalmol ; 141(9): 906-907, 2023 09 01.
Artículo en Inglés | MEDLINE | ID: mdl-37535363

RESUMEN

This cross-sectional study compares 2 sets of responses by 1 chatbot to frequently asked questions about vitreoretinal conditions and procedures.


Asunto(s)
Inteligencia Artificial , Degeneración Retiniana , Humanos , Degeneración Retiniana/diagnóstico
20.
JAMA Ophthalmol ; 136(11): 1271-1279, 2018 11 01.
Artículo en Inglés | MEDLINE | ID: mdl-30178000

RESUMEN

Importance: Medical and surgical interventions for glaucoma are effective only if they are affordable to patients. Little is known about how affordable glaucoma interventions are in developing and developed countries. Objective: To compare the prices of topical glaucoma medications, laser trabeculoplasty, and trabeculectomy relative with median annual household income (MA-HHI) for countries worldwide. Design, Setting and Participants: Cross-sectional observational study. For each country, we obtained prices for glaucoma medications, laser trabeculoplasty, and trabeculectomy using government pricing data, drug databases, physician fee schedules, academic publications, and communications with local ophthalmologists. Prices were adjusted for purchasing power parity and inflation to 2016 US dollars, and annual therapy prices were examined relative to the MA-HHI. Interventions costing less than 2.5% of the MA-HHI were considered affordable. Main Outcomes and Measures: Daily cost for topical glaucoma medications, cost of annual therapy with glaucoma medications, laser trabeculoplasty, and trabeculectomy relative to MA-HHI in each country. Results: Data were obtained from 38 countries, including 17 developed countries and 21 developing countries, as classified by the World Economic Outlook. We observed considerable variability in intervention prices compared with MA-HHI across the countries and across interventions, ranging from 0.1% to 5% of MA-HHI for timolol, 0.1% to 27% for latanoprost, 0.2% to 17% for laser trabeculoplasty, and 0.3% to 42% for trabeculectomy. Timolol was the most affordable medication in all countries studied and was 2.5% or more of MA-HHI in only 2 countries (5%). The annual cost of latanoprost was 2.5% or more of MA-HHI in 15 countries (41%) (15 developing countries [75%] and no developed countries). The cost of laser trabeculoplasty was 2.5% or more of the MA-HHI in 15 countries (44%) (11 developing countries [65%] and 4 developed countries [24%]). The cost of trabeculectomy was 2.5% or more of the MA-HHI in 28 countries (78%) (18 developing countries [95%] and 10 developed countries [59%]). In 18 countries (53%), laser trabeculoplasty cost less than a 3-year latanoprost supply. Conclusions and Relevance: For many patients worldwide, the costs of medical, laser, and incisional surgical interventions were 2.5% or more of the MA-HHI. Successfully reducing global blindness from glaucoma requires addressing multiple contributing factors, including making glaucoma interventions more affordable.


Asunto(s)
Antihipertensivos/economía , Glaucoma/economía , Glaucoma/terapia , Coagulación con Láser/economía , Trabeculectomía/economía , Administración Oftálmica , Costos y Análisis de Costo , Estudios Transversales , Costos de los Medicamentos , Femenino , Glaucoma/tratamiento farmacológico , Glaucoma/cirugía , Salud Global , Accesibilidad a los Servicios de Salud/economía , Humanos , Renta , Presión Intraocular , Latanoprost/economía , Masculino , Persona de Mediana Edad , Timolol/economía
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