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1.
Ophthalmol Retina ; 2020 Aug 28.
Artículo en Inglés | MEDLINE | ID: mdl-32866664

RESUMEN

PURPOSE: To compare the relative retinal pixels and retinal area imaged using the Optos P200DTx and Zeiss Clarus 500 ultra-wide field (UWF) fundus cameras. DESIGN: Single-center retrospective cross-sectional analysis SUBJECTS: 78 eyes of 46 patients METHODS: Eyes were imaged with both the Optos P200DTx and Zeiss Clarus 500 UWF fundus cameras. The Optos P200DTx photos were single capture images without montage. Clarus images were two single capture images that were montaged, when possible. The relative pixels encompassing all visible retina in four quadrants centered on the fovea were measured. Measurements were normalized to the optic disc area for each image to account for pixel density and image size differences. Additionally, retinal area was measured in all four quadrants using Optos proprietary software Area-Measurement Tool and ImageJ software, using Optos P200DTx images and Zeiss Clarus 500 images that were registered to the Optos P200DTx images. Patients and technicians were asked for their preference between the machines. Imaging session times were recorded. MAIN OUTCOME MEASURE: Relative retinal pixels and retina area captured by each fundus camera. RESULTS: When comparing all 78 images, the Optos P200DTx consistently captured more relative pixels in all four quadrants compared to the Zeiss Clarus 500: 93.8 vs. 75.0 superiorly (p < 0.001), 102.6 vs. 61.6 inferiorly (p < 0.001), 146.9 vs. 91.2 temporally (p < 0.001), and 167.1 vs. 127.8 nasally (p < 0.001). For the area calculation, 70 (89.7%) of the 78 images had successful registration of Zeiss Clarus 500 images to Optos P200DTx images. The Optos captured a larger retinal area in all four quadrants: 169.3 mm2 vs 131.6 mm2 superiorly (p < 0.001), 169.3 mm2 vs. 112.5 mm2 inferiorly (p < 0.001), 213.3 mm2 vs 143.6 mm2 temporally (p < 0.001), and 220.7 mm2 vs 178.8 mm2 nasally (p < 0.001). Eighteen (21.8%) of the 78 Zeiss Clarus 500 images were single capture images due to the patient being unable to take multiple images and were unable to be montaged. When comparing only the 52 (74.3%) of 70 Zeiss Clarus 500 images that were able to be montaged with the Optos P200DTx images, there was similarly significantly more relative pixel count and retina area imaged by the Optos P200DTx images. Among the 66 images with peripheral pathology, the Optos P200DTx captured findings not captured by the Zeiss Clarus 500 in 28 (42.4%) images, and the Zeiss Clarus 500 captured findings not captured by the Optos P200DTx in 1 (1.5%) image (P < 0.001). Among the 48 imaging sessions in which technicians responded with a preference, the Optos P200DTx was preferred for 28 imaging sessions (58%) compared to the Zeiss Clarus 500 for 20 imaging sessions (42%, P = 0.15). Among 44 patients who responded with a preference, 24 preferred the Optos P200DTx, 20 preferred the Zeiss Clarus 500 (P = 0.52). The average imaging session time was 4.6 minutes (SD: 3.0) for the Optos P200DTx and 5.3 minutes (SD: 3.1) for the Zeiss Clarus 500 (p=0.17). CONCLUSION: In the current study, the Optos P200DTx captured statistically significantly more retinal area in all four quadrants compared to the Zeiss Clarus 500. There was no statistically significant difference in patient or technician preference or image acquisition time between the two devices.

2.
JAMA Ophthalmol ; 137(6): 603-609, 2019 06 01.
Artículo en Inglés | MEDLINE | ID: mdl-30896765

RESUMEN

Importance: Targeting the early pathogenic steps in Stargardt disease type 1 (STGD1) is critical to advance our understanding of this condition and to develop potential therapies. Lipofuscin precursors may accumulate within photoreceptors, leading to photoreceptor damage and preceding retinal pigment epithelial (RPE) cell death. Fluorescence adaptive optics scanning light ophthalmoscopy can provide autofluorescence (AF) images in vivo with microscopic resolution to elucidate the cellular origin of AF abnormalities in STGD1. Objective: To study the spatial distribution of photoreceptor, RPE, and AF abnormalities in patients with STGD1 at a cellular level. Design, Setting, and Participants: Cross-sectional study using fluorescence adaptive optics scanning light ophthalmoscopy to compare the cones, rods, and RPE cells between 3 patients with STGD1 and 1 control individual. Imaging sessions were conducted at the University of Rochester. Further image analyses were performed at Beijing Tongren Eye Center and the University of Pittsburgh. Data were collected from August 2015 to February 2016, and analysis began in March 2016. Main Outcomes and Measures: Structural appearance of cones, rods, and AF structures at different retinal locations. Results: Two women and 1 man with macular atrophy phenotype of STGD1 and visual acuity loss ranging from 20/30 to 20/150 and 1 woman without STGD1 with 20/20 visual acuity were analyzed. Cone and rod spacing was increased in all 3 patients at all locations where photoreceptors were detectable; most cones had a dark appearance. Autofluorescence was low contrast but contained structures consistent with RPE cells in the periphery. In the transition zone peripheral to the foveal atrophic lesion, the structural pattern of AF was more consistent with photoreceptors than RPE cells. The microscopic AF was disrupted within areas of clinically detectable atrophy. Conclusions and Relevance: Adaptive optics high-resolution images of cones, rods, and RPE cells at the leading disease front of STGD1 macular atrophy show an AF pattern that appears to colocalize with photoreceptors or may result from a combination of AF signals from both RPE cells and photoreceptors. This in vivo observation is consistent with histologic reports of fluorescence arising from photoreceptors in STGD1. The detection of bisretinoid accumulation in the photoreceptors may represent an early pathologic step in STGD1 and can provide an in vivo imaging tool to act as a biomarker of disease progression.


Asunto(s)
Células Fotorreceptoras de Vertebrados/patología , Epitelio Pigmentado de la Retina/patología , Enfermedad de Stargardt/diagnóstico , Adulto , Estudios Transversales , Femenino , Angiografía con Fluoresceína , Fondo de Ojo , Humanos , Masculino , Oftalmoscopía , Imagen Óptica , Óptica y Fotónica , Adulto Joven
3.
Invest Ophthalmol Vis Sci ; 59(15): 5705-5716, 2018 12 03.
Artículo en Inglés | MEDLINE | ID: mdl-30513531

RESUMEN

Purpose: To characterize in vivo morphometry and multispectral autofluorescence of the retinal pigment epithelial (RPE) cell mosaic and its relationship to cone cell topography across the macula. Methods: RPE cell morphometrics were computed in regularly spaced regions of interest (ROIs) from contiguous short-wavelength autofluorescence (SWAF) and photoreceptor reflectance images collected across the macula in one eye of 10 normal participants (23-65 years) by using adaptive optics scanning light ophthalmoscopy (AOSLO). Infrared autofluorescence (IRAF) images of the RPE were collected with AOSLO in seven normal participants (22-65 years), with participant overlap, and compared to SWAF quantitatively and qualitatively. Results: RPE cell statistics could be analyzed in 84% of SWAF ROIs. RPE cell density consistently decreased with eccentricity from the fovea (participant mean ± SD: 6026 ± 1590 cells/mm2 at fovea; 4552 ± 1370 cells/mm2 and 3757 ± 1290 cells/mm2 at 3.5 mm temporally and nasally, respectively). Mean cone-to-RPE cell ratio decreased rapidly from 16.6 at the foveal center to <5 by 1 mm. IRAF revealed cells in six of seven participants, in agreement with SWAF RPE cell size and location. Differences in cell fluorescent structure, contrast, and visibility beneath vasculature were observed between modalities. Conclusions: Improvements in AOSLO autofluorescence imaging permit efficient visualization of RPE cells with safe light exposures, allowing individual characterization of RPE cell morphometry that is variable between participants. The normative dataset and analysis of RPE cell IRAF and SWAF herein are essential for understanding microscopic characteristics of cell fluorescence and may assist in interpreting disease progression in RPE cells.


Asunto(s)
Células Fotorreceptoras Retinianas Conos/citología , Epitelio Pigmentado de la Retina/citología , Adulto , Anciano , Recuento de Células , Femenino , Voluntarios Sanos , Humanos , Masculino , Persona de Mediana Edad , Mosaicismo , Oftalmoscopía/métodos , Imagen Óptica , Óptica y Fotónica , Epitelio Pigmentado de la Retina/diagnóstico por imagen , Tomografía de Coherencia Óptica , Adulto Joven
4.
Proc Natl Acad Sci U S A ; 114(39): E8214-E8223, 2017 09 26.
Artículo en Inglés | MEDLINE | ID: mdl-28878022

RESUMEN

Age-related macular degeneration (AMD) and related macular dystrophies (MDs) are a major cause of vision loss. However, the mechanisms underlying their progression remain ill-defined. This is partly due to the lack of disease models recapitulating the human pathology. Furthermore, in vivo studies have yielded limited understanding of the role of specific cell types in the eye vs. systemic influences (e.g., serum) on the disease pathology. Here, we use human induced pluripotent stem cell-retinal pigment epithelium (hiPSC-RPE) derived from patients with three dominant MDs, Sorsby's fundus dystrophy (SFD), Doyne honeycomb retinal dystrophy/malattia Leventinese (DHRD), and autosomal dominant radial drusen (ADRD), and demonstrate that dysfunction of RPE cells alone is sufficient for the initiation of sub-RPE lipoproteinaceous deposit (drusen) formation and extracellular matrix (ECM) alteration in these diseases. Consistent with clinical studies, sub-RPE basal deposits were present beneath both control (unaffected) and patient hiPSC-RPE cells. Importantly basal deposits in patient hiPSC-RPE cultures were more abundant and displayed a lipid- and protein-rich "drusen-like" composition. Furthermore, increased accumulation of COL4 was observed in ECM isolated from control vs. patient hiPSC-RPE cultures. Interestingly, RPE-specific up-regulation in the expression of several complement genes was also seen in patient hiPSC-RPE cultures of all three MDs (SFD, DHRD, and ADRD). Finally, although serum exposure was not necessary for drusen formation, COL4 accumulation in ECM, and complement pathway gene alteration, it impacted the composition of drusen-like deposits in patient hiPSC-RPE cultures. Together, the drusen model(s) of MDs described here provide fundamental insights into the unique biology of maculopathies affecting the RPE-ECM interface.


Asunto(s)
Lámina Basal de la Coroides/patología , Enfermedades Hereditarias del Ojo/patología , Células Madre Pluripotentes Inducidas/citología , Degeneración Macular/patología , Drusas Retinianas/patología , Epitelio Pigmentado de la Retina/citología , Ceguera/genética , Ceguera/patología , Células Cultivadas , Colágeno Tipo IV/metabolismo , Proteínas de la Matriz Extracelular/genética , Proteínas de la Matriz Extracelular/metabolismo , Humanos , Drusas del Disco Óptico/congénito , Drusas del Disco Óptico/patología , Epitelio Pigmentado de la Retina/patología , Inhibidor Tisular de Metaloproteinasa-3/genética
5.
Proc Natl Acad Sci U S A ; 114(3): 586-591, 2017 01 17.
Artículo en Inglés | MEDLINE | ID: mdl-28049835

RESUMEN

Although imaging of the living retina with adaptive optics scanning light ophthalmoscopy (AOSLO) provides microscopic access to individual cells, such as photoreceptors, retinal pigment epithelial cells, and blood cells in the retinal vasculature, other important cell classes, such as retinal ganglion cells, have proven much more challenging to image. The near transparency of inner retinal cells is advantageous for vision, as light must pass through them to reach the photoreceptors, but it has prevented them from being directly imaged in vivo. Here we show that the individual somas of neurons within the retinal ganglion cell (RGC) layer can be imaged with a modification of confocal AOSLO, in both monkeys and humans. Human images of RGC layer neurons did not match the quality of monkey images for several reasons, including safety concerns that limited the light levels permissible for human imaging. We also show that the same technique applied to the photoreceptor layer can resolve ambiguity about cone survival in age-related macular degeneration. The capability to noninvasively image RGC layer neurons in the living eye may one day allow for a better understanding of diseases, such as glaucoma, and accelerate the development of therapeutic strategies that aim to protect these cells. This method may also prove useful for imaging other structures, such as neurons in the brain.


Asunto(s)
Oftalmoscopía/métodos , Células Ganglionares de la Retina/citología , Animales , Femenino , Glaucoma/diagnóstico por imagen , Humanos , Macaca fascicularis/anatomía & histología , Macaca mulatta/anatomía & histología , Degeneración Macular/diagnóstico por imagen , Degeneración Macular/patología , Masculino , Fenómenos Ópticos , Células Fotorreceptoras Retinianas Conos/citología , Especificidad de la Especie
6.
JAMA Ophthalmol ; 133(4): 421-6, 2015 Apr.
Artículo en Inglés | MEDLINE | ID: mdl-25590968

RESUMEN

IMPORTANCE: Aflibercept-related sterile inflammation, an event that is poorly understood, has been the subject of ongoing postmarketing reports. OBJECTIVE: To analyze cases of aflibercept-related sterile inflammation reported to the American Society of Retina Specialists (ASRS) Therapeutic Surveillance Committee (TSC), an independent task force formed to monitor drug- and device-related safety events. DESIGN, SETTING, AND PARTICIPANTS: A retrospective review of 56 cases in 55 patients was performed of all cases of sterile inflammation after aflibercept injection that were voluntarily reported by 12 practices throughout the United States to the ASRS TSC from December 1, 2011, through February 12, 2014. MAIN OUTCOMES AND MEASURES: Cases of aflibercept-related sterile inflammation were analyzed for baseline and demographic information, presenting symptoms and findings, visual acuity changes, injection technique, and management details. RESULTS: Among 56 reported cases of sterile inflammation, mean time to onset was 3.5 days (median, 2 days; range, 0-30 days). Most cases consisted of initial loss of vision and intraocular inflammation without prominent redness, severe pain, or hypopyon. Thirty-seven cases (66%) were treated with topical corticosteroids and/or observation alone. Mean time to resolution was 28.6 days (median, 28 days; range, 4-84 days). Although final vision was overall unchanged, some patients developed permanent vision loss, which may have resulted from inflammation and/or progression of the underlying disease. Age older than 80 years was associated with worse visual outcomes. No difference in visual outcome was detected in patients with sterile inflammation undergoing topical therapy alone vs invasive procedures (vitreous biopsy and/or intravitreal antibiotic administration and/or vitrectomy). CONCLUSIONS AND RELEVANCE: With the largest number of reported cases of aflibercept-related sterile inflammation to our knowledge, this analysis suggests typical findings and an often favorable prognosis of this event. Analysis of real-world, postmarketing data has limitations, and these findings should be considered as hypothesis-generating assessments rather than a definitive reflection of this event or its typical course. Distinguishing sterile inflammation and infectious endophthalmitis at the time of presentation may often be difficult, and cautious evaluation and management of these patients are warranted. The ASRS TSC encourages active postmarketing surveillance by all physicians.


Asunto(s)
Inhibidores de la Angiogénesis/efectos adversos , Endoftalmitis/inducido químicamente , Vigilancia de Productos Comercializados/estadística & datos numéricos , Receptores de Factores de Crecimiento Endotelial Vascular/efectos adversos , Proteínas Recombinantes de Fusión/efectos adversos , Anciano , Anciano de 80 o más Años , Neovascularización Coroidal/tratamiento farmacológico , Endoftalmitis/diagnóstico , Endoftalmitis/tratamiento farmacológico , Dolor Ocular/diagnóstico , Dolor Ocular/tratamiento farmacológico , Femenino , Glucocorticoides/uso terapéutico , Humanos , Inyecciones Intravítreas , Degeneración Macular/tratamiento farmacológico , Masculino , Persona de Mediana Edad , Oclusión de la Vena Retiniana/tratamiento farmacológico , Estudios Retrospectivos , Factores de Tiempo , Factor A de Crecimiento Endotelial Vascular/antagonistas & inhibidores , Agudeza Visual
7.
Retina ; 35(6): 1128-34, 2015 Jun.
Artículo en Inglés | MEDLINE | ID: mdl-25635575

RESUMEN

PURPOSE: After the recent approval of ocriplasmin by the Food and Drug Administration, postmarketing safety concerns have been raised by the vitreoretinal community. The American Society of Retina Specialists Therapeutic Surveillance Committee was commissioned to monitor postmarketing drug-related and device-related adverse events. The purpose of this report is to analyze the postmarketing safety experience in the context of available premarketing safety data. METHODS: Periodic aggregate safety reports consisting of premarketing, or clinical trial, data (n = 999 injections) and postmarketing reports through July 16, 2013 (n = 4,387 injections), were retrospectively analyzed by the TSC. The aggregate data were analyzed to classify adverse events, and the postmarketing safety data for each event type were compared with the premarketing data. RESULTS: Eight categories of adverse events were identified. Acute reduction in visual acuity attributable to either worsening of macular pathology or development of subretinal fluid, electroretinogram changes, dyschromatopsia, retinal tears and detachments, lens subluxation or phacodonesis, impaired pupillary reflex, and retinal vessel findings were reported in both the premarketing and postmarketing experiences. Ellipsoid zone (inner segment/outer segment) findings were only reported in the postmarketing experience. Rates of postmarketing reports were lower than in the premarketing data. Adverse events were generally transient, and characteristics of these adverse events were generally similar between the premarketing and postmarketing experience. CONCLUSION: Postmarket analyses are limited by significant underreporting, and in the case of ocriplasmin as a first in-class drug, they may not have captured safety events that have only more recently been identified. Nonetheless, postmarket analyses can identify the scope of potential safety events based on real-world experiences. Ocriplasmin administration should be guided by an appropriate and informed risk-benefit discussion with the patient. Ongoing active postmarket surveillance by all practitioners will continue to be critical to better understand this safety profile.


Asunto(s)
Efectos Colaterales y Reacciones Adversas Relacionados con Medicamentos/etiología , Oftalmopatías/tratamiento farmacológico , Fibrinolisina/efectos adversos , Fibrinolíticos/efectos adversos , Fragmentos de Péptidos/efectos adversos , Vigilancia de Productos Comercializados , Enfermedades de la Retina/tratamiento farmacológico , Cuerpo Vítreo/efectos de los fármacos , Ensayos Clínicos como Asunto , Defectos de la Visión Cromática/inducido químicamente , Evaluación Preclínica de Medicamentos , Electrorretinografía/efectos de los fármacos , Fibrinolisina/uso terapéutico , Fibrinolíticos/uso terapéutico , Humanos , Inyecciones Intravítreas , Subluxación del Cristalino/inducido químicamente , Fragmentos de Péptidos/uso terapéutico , Células Fotorreceptoras de Vertebrados/efectos de los fármacos , Células Fotorreceptoras de Vertebrados/patología , Reflejo Pupilar/efectos de los fármacos , Desprendimiento de Retina/inducido químicamente , Perforaciones de la Retina/inducido químicamente , Estudios Retrospectivos , Adherencias Tisulares/tratamiento farmacológico , Agudeza Visual/efectos de los fármacos
8.
Biomed Opt Express ; 4(11): 2527-39, 2013.
Artículo en Inglés | MEDLINE | ID: mdl-24298413

RESUMEN

Morgan and colleagues demonstrated that the RPE cell mosaic can be resolved in the living human eye non-invasively by imaging the short-wavelength autofluorescence using an adaptive optics (AO) ophthalmoscope. This method, based on the assumption that all subjects have the same longitudinal chromatic aberration (LCA) correction, has proved difficult to use in diseased eyes, and in particular those affected by age-related macular degeneration (AMD). In this work, we improve Morgan's method by accounting for chromatic aberration variations by optimizing the confocal aperture axial and transverse placement through an automated iterative maximization of image intensity. The increase in image intensity after algorithmic aperture placement varied depending upon patient and aperture position prior to optimization but increases as large as a factor of 10 were observed. When using a confocal aperture of 3.4 Airy disks in diameter, images were obtained using retinal radiant exposures of less than 2.44 J/cm(2), which is ~22 times below the current ANSI maximum permissible exposure. RPE cell morphologies that were strikingly similar to those seen in postmortem histological studies were observed in AMD eyes, even in areas where the pattern of fluorescence appeared normal in commercial fundus autofluorescence (FAF) images. This new method can be used to study RPE morphology in AMD and other diseases, providing a powerful tool for understanding disease pathogenesis and progression, and offering a new means to assess the efficacy of treatments designed to restore RPE health.

10.
Surg Neurol Int ; 3: 4, 2012.
Artículo en Inglés | MEDLINE | ID: mdl-22347674

RESUMEN

OBJECTIVE: Leber congenital amaurosis (LCA) is a rare, clinically and genetically heterogeneous disorder characterized by severe loss of vision in the first year of life, affecting approximately 3000 people in the United States. Some LCA patients manifest developmental abnormalities of the central nervous system (CNS) and neuroradiological studies have revealed a variety of cerebral anomalies in association with LCA; however, Chiari I malformations (CMI) have never been described. CASE DESCRIPTION: We report two sisters who were referred to the pediatric neurosurgery clinic for evaluation of CMI. The elder sister presented with convergence nystagmus from 3 months of age and magnetic resonance imaging (MRI) demonstrated evidence of significant CMI. Her younger sister began developing nystagmus at 4 months of age. Both had symptomatic progression and underwent suboccipital decompression. Both were subsequently diagnosed with LCA. Case specifics and imaging findings are presented. CONCLUSIONS: CMI have been found in association with several genetic syndromes, but not with LCA. These patients represent the first reported cases of CMI with LCA and suggest an additional potential CNS anomaly. The unique occurrence in siblings and the association with another inherited disorder are suggestive of a genetic basis for CMI.

11.
Ophthalmology ; 119(4): 674-81, 2012 Apr.
Artículo en Inglés | MEDLINE | ID: mdl-22266108

RESUMEN

PURPOSE: To report the clinical characteristics of infectious endophthalmitis after Boston type I keratoprosthesis (K-Pro) implantation. DESIGN: Retrospective study. PARTICIPANTS: One hundred forty-one adult eyes receiving a K-Pro at a single institution from May 2004 through July 2008. METHODS: A retrospective chart review was performed of all adult eyes receiving a K-Pro at the University of Rochester from May 2004 through July 2008. Those patients identified as having been treated for exogenous bacterial endophthalmitis were reviewed for demographic data, indication for K-Pro, bandage contact lens use, prophylactic antibiotic use, timing and clinical presentation of endophthalmitis, gram stain and culture results of intraocular fluid, timing and presentation of any subsequent episodes of endophthalmitis (recurrent endophthalmitis), and preoperative and postoperative visual acuity through August 2010. MAIN OUTCOME MEASURES: Incidence of endophthalmitis, time to occurrence, recurrence rates, visual outcomes, and risk factors associated with K-Pro endophthalmitis. RESULTS: Ten (7.1%) of 141 eyes of 130 adult patients were diagnosed and treated for bacterial endophthalmitis. Average time to endophthalmitis developing after K-Pro was 9.8 months (standard deviation [SD], 6.2 months; range, 2-25 months). Coagulase-negative staphylococci were identified in 7 eyes. In 7 of the 10 eyes, recurrent endophthalmitis developed that occurred at a mean of 4 months (SD, 3.9 months; range, 1-13 months) after resolution of the initial episode. At each episode of endophthalmitis, no eye was receiving vancomycin ophthalmic drops and most eyes were receiving only fluoroquinolone ophthalmic drops for prophylaxis. CONCLUSIONS: Infectious endophthalmitis after K-Pro implantation has a higher incidence, delayed onset, and high risk for recurrence compared with postoperative endophthalmitis associated with more common intraocular procedures such as cataract surgery. The concurrent use of topical vancomycin is recommended because it seems to be important in reducing the incidence and recurrence of endophthalmitis and because fluoroquinolone ophthalmic drops do not seem to be sufficient prophylaxis in these eyes.


Asunto(s)
Órganos Artificiales , Trasplante de Córnea , Endoftalmitis/microbiología , Infecciones Bacterianas del Ojo/microbiología , Complicaciones Posoperatorias , Implantación de Prótesis , Anciano , Anciano de 80 o más Años , Profilaxis Antibiótica , Opacidad de la Córnea/cirugía , Endoftalmitis/diagnóstico , Endoftalmitis/tratamiento farmacológico , Infecciones por Enterobacteriaceae/diagnóstico , Infecciones por Enterobacteriaceae/tratamiento farmacológico , Infecciones por Enterobacteriaceae/microbiología , Infecciones Bacterianas del Ojo/diagnóstico , Infecciones Bacterianas del Ojo/tratamiento farmacológico , Estudios de Seguimiento , Humanos , Pruebas de Sensibilidad Microbiana , Persona de Mediana Edad , Prótesis e Implantes , Recurrencia , Estudios Retrospectivos , Factores de Riesgo , Infecciones Estafilocócicas/diagnóstico , Infecciones Estafilocócicas/tratamiento farmacológico , Infecciones Estafilocócicas/microbiología , Agudeza Visual/fisiología , Cuerpo Vítreo/microbiología
12.
Arch Ophthalmol ; 130(3): 357-64, 2012 Mar.
Artículo en Inglés | MEDLINE | ID: mdl-22084158

RESUMEN

OBJECTIVE: To describe the anatomical phenotypes of Best vitelliform macular dystrophy (BVMD) with spectral-domain optical coherence tomography (SD-OCT) in a large series of patients with confirmed mutations in the BEST1 gene. METHODS: In our retrospective observational case series, we assessed 15 patients (30 eyes) with a clinical diagnosis of vitelliform macular dystrophy who were found to have mutations in the BEST1 gene. Color fundus photographs and SD-OCT images were evaluated and compared with those of 15 age-matched controls (30 eyes). Using a validated 3-dimensional SD-OCT segmentation algorithm, we calculated the equivalent thickness of photoreceptors and the equivalent thickness of the retinal pigment epithelium for each patient. The photoreceptor equivalent thickness and the retinal pigment epithelium (RPE) equivalent thickness were compared in all patients, in a region of the macula outside the central lesion for patients with BVMD and outside the fovea in control patients. Paired t tests were used for statistical analysis. RESULTS: The SD-OCT findings revealed that the vitelliform lesion consists of material above the RPE and below the outer segment tips. Additionally, drusen-like deposition of sub-RPE material was notable, and several patients exhibited a sub-RPE fibrotic nodule. Patients with BVMD had a mean photoreceptor equivalent thickness of 28.3 µm, and control patients had a mean photoreceptor equivalent thickness of 21.8 µm, a mean difference of 6.5 µm (P < .01), whereas the mean RPE equivalent thickness was not statistically different between patients with BVMD and control patients (P = .53). CONCLUSIONS: The SD-OCT findings suggest that vitelliform material is located in the subretinal space and that BVMD is associated with diffuse photoreceptor outer segment abnormalities overlying a structurally normal RPE. CLINICAL RELEVANCE: These findings provide new insight into the pathophysiology of BVMD and thus have implications for the development of therapeutic interventions.


Asunto(s)
Mácula Lútea/patología , Células Fotorreceptoras Retinianas Conos/patología , Epitelio Pigmentado de la Retina/patología , Distrofia Macular Viteliforme/patología , Adolescente , Adulto , Anciano , Anciano de 80 o más Años , Bestrofinas , Canales de Cloruro/genética , Proteínas del Ojo/genética , Femenino , Fibrosis , Humanos , Imagenología Tridimensional , Masculino , Persona de Mediana Edad , Drusas Retinianas/genética , Drusas Retinianas/patología , Estudios Retrospectivos , Tomografía de Coherencia Óptica/métodos , Distrofia Macular Viteliforme/genética , Adulto Joven
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