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PubMed2005

Fundus autofluorescence in children and teenagers with hereditary retinal diseases.

INTRODUCTION: In adults, evaluation of fundus autofluorescence (AF) plays an important role in the differential diagnosis of retinal diseases. The aim of this study was to evaluate the feasibility of recording AF in children and teenagers and to define typical AF findings of various hereditary retinal diseases during childhood. METHODS: Fifty patients aged 2 to 16 years with hereditary retinal diseases were analysed using the HRA (Heidelberg Retina Angiograph). To enhance the AF signal, a mean of up to 16 single images was calculated. Twenty healthy children (aged 4-16 years) served as controls. RESULTS: In many children as young as 5 years of age and even in one 2-year-old child good AF images could be obtained. To achieve high quality images, larger image series (about 50 single images) were taken and appropriate single images were chosen manually to calculate the mean. Characteristically, Stargardt disease shows a central oval area of reduced AF, often surrounded by more irregular AF. In patients with Best disease, a central round structure with regular or irregular intense AF is visualised. Some patients with X-linked retinoschisis show central radial structures. In many patients with rod-cone dystrophies, a central oval ring-shaped area of increased AF is present. In early-onset severe retinal dystrophy (EOSRD) with RPE65 mutations AF is completely absent, whereas in other forms of Leber congenital amaurosis, AF is normal. DISCUSSION: Fundus autofluorescence may visualise disease-specific distributions of lipofuscin in the retinal pigment epithelium, often not (yet) visible on ophthalmoscopy. AF images can be used in children to differentiate hereditary retinal diseases and to facilitate follow-up controls. In many cases, four single images are sufficient to analyse the AF pattern.

Adolescent↗

PubMed1982

Temporal modulation transfer function in patients with retinal diseases.

The temporal modulation transfer function (temporal MTF) was measured in normal controls and in patients with various retinal diseases. Temporal MTF of controls showed band-pass filter properties, having a peak sensitivity at about 10 Hz at 1,000 Troland of retinal luminance. As the retinal luminance was reduced, the sensitivity decreased first at high temporal frequencies; then the peak sensitivity decreased, and finally at low temporal frequencies the sensitivity decreased and the peak shifted to the left. In retinal diseases, abnormal temporal MTFs were detected, and the curves were very similar to those of normal controls when retinal luminance was reduced. These patterns were not specific to any retinal diseases, and the severity of the conditions seemed to determine the pattern.

Adult↗

PubMed1997

Localization of tissue inhibitor of metalloproteinases-3 in neurodegenerative retinal disease.

Tissue inhibitor of metalloproteinases-3 (TIMP-3) is one of a family of genes whose products are implicated in the regulation of remodelling of the extracellular matrix. The level of mRNA coding for TIMP-3 is increased in retinas affected by the photoreceptor degenerative disease, simplex retinitis pigmentosa (RP), and mutations in TIMP-3 are associated with an inherited form of macular dystrophy. Here we compare TIMP-3 protein expression in normal retina and in those affected by RP and by age-related macular degeneration. Immunoreactive TIMP-3 is present in normal retinal pigment epithelium, and in degenerative retinas particularly at Bruch's membrane and additionally in photoreceptor-retaining regions in simplex RP. The pattern suggests a role for TIMP-3 in normal retinal homeostasis, and, in the disease state, in the modulation of extracellular matrix metabolism and neovascularization.

Aged↗

PubMed1995

The first and second harmonics of the macular flicker electroretinogram: differential effects of retinal diseases.

We evaluated the effects of retinal diseases on the macular electroretinogram first and second harmonic components, which are dominated by outer and inner retinal activity, respectively. Macular electroretinograms in response to a uniform field (9 degrees x 9 degrees) flickering sinusoidally at either 32 or 8 Hz (peak frequencies of the first and second harmonics, respectively) were recorded in 14 patients with maculopathies involving photoreceptors (e.g., age-related macular degeneration), in 16 patients with postreceptoral macular diseases (e.g., branch occlusion of central retinal artery), and in 38 normal controls. Amplitude and phase of the first and second harmonic response components were evaluated by Fourier analysis. When compared to controls, patients with photoreceptor diseases had reduction in both first and second harmonic mean amplitudes and second harmonic phase delay; patients with postreceptoral diseases had normal first harmonic components but reduced and delayed second harmonic components. A discriminant analysis, by using first and second harmonic values, correctly classified 13 of 14 patients with photoreceptor diseases and 14 of 16 patients with postreceptoral disorders. These results indicate that combined evaluation of the macular electroretinogram first and second harmonic components is a useful test for identifying the site(s) of retinal dysfunction in patients with macular diseases.

Adolescent↗

PubMed1991

Photoreceptor cell rescue at early and late RPE-cell transplantation periods during retinal disease in RCS dystrophic rats.

Normal retinal pigment epithelial (RPE) cells were transplanted into retinas of Royal College of Surgeons (RCS) dystrophic rats at different stages of the retinal disease process. RPE-cell transplantation at 10, 17 and 26 days resulted in rescue of photoreceptor cells, such that at 4 months the outer nuclear layer (ONL) was 8-10 cells in thickness as shown in retinas of age-matched control rats. Of these transplantation times, day 17 appeared to affect the best rescue of photoreceptor cells. Nongrafted retinas of 4 month-old RCS dystrophic rats exhibited scattered PRC's, most prevalent in the peripheral retina. In addition, a small, but significant increase in the ONL thickness was detected in vehicle-injected retinas (sham control) of 17 day-old RCS dystrophic rats at 2 months; however, at 3 months, the ONL thickness was reduced to control levels. A normal distribution of (Na+ + K+)-ATPase immunostain was demonstrated beneath grafted RPE cells in retinas of 4 month-old RCS dystrophic rats. Dense immunostaining was shown along rescued photoreceptor cell inner segments (IS), within the inner (IPL) and outer (OPL) plexiform layers and on plasmalemma of cell bodies in the inner nuclear layer (INL). In nongrafted retinas of age-matched RCS dystrophic rats, immunostaining for (Na+ + K+)-ATPase was observed only in the INL and IPL. Under RPE-cells transplants in retinas of 4 month-old RCS dystrophic rats, opsin immunostaining was detected along both rescued photoreceptor cell inner and outer (OS) segments and on plasmalemma of ONL cell bodies. However, immunostaining for opsin was restricted to a debris zone in nongrafted retinas of age-matched RCS dystrophic rats.(ABSTRACT TRUNCATED AT 250 WORDS)

Animals↗

PubMed1981

Contrast sensitivity and retinal disease.

A new and practical test for contrast sensitivity (Arden gratings) was evaluated in retinal diseases and found to be a more sensitive indicator of dysfunction than Snellen acuity. Most patients with acuity of 20/30 or better (e.g., with retinitis pigmentosa or toxic retinopathy) showed diminished sensitivity to finely spaced gratings, while patients with poor acuity (e.g., with cone dystrophy) showed normal sensitivity to coarsely spaced gratings. Grating tests may allow earlier diagnosis and more precise follow-up of retinal disease, but their specificity for particular disorders remains to be established.

Adult↗

PubMed1999

Validity of the visual function index (VF-14) in patients with retinal disease.

OBJECTIVE: To test the validity of the Visual Function Index (VF-14) in patients with retinal disease. DESIGN: A self-administered questionnaire package in association with clinical examination findings. PARTICIPANTS: Consecutive patients attending the Vancouver General Hospital Eye Care Centre, Vancouver, British Columbia, retina clinic between May 1 and August 15, 1998. MAIN OUTCOME MEASURES: Responses to the questionnaire package as they relate to global self-assessment scales and visual acuity. In addition, correlations were calculated between the VF-14, the 36-Item Short-Form Health Survey, a Weighted Comorbidity Scale, and visual acuity scores. RESULT: Five hundred forty-seven patients were given the questionnaire package to complete. The VF-14 demonstrated a moderately strong positive association with patient self-rating of amount of trouble, satisfaction, and overall quality of vision. Correlations between the 36-Item Short-Form Health Survey, visual acuity, and the global scales were mild to moderate. The VF-14 was moderately correlated with visual acuity in the better and the worse eyes. CONCLUSIONS: This study provides support for the validity of the VF-14 as a measure of functional impairment in patients with retinal disease. Once responsiveness has been measured and an analysis of disease subtypes has been carried out, the VF-14 will be ready for inclusion in clinical trials to evaluate patients' functional ability. Further implementation and development of this outcome measure will better our understanding of the utility of the functional assessment format for patients with retinal disease.

Adolescent↗

PubMed1999

Distribution of pigment epithelium autofluorescence in retinal disease state recorded in vivo and its change over time.

BACKGROUND: Recently a technique of imaging the retinal pigment epithelium (RPE) has been developed that takes advantages of its intrinsic fluorescence derived from lipofuscin. The purpose of this study was to document the distribution of fundus autofluorescence in patients with various retinal diseases and its change over time. METHODS: The intensity and spatial distribution of fundus autofluorescence was documented in 318 eyes from 159 patients with various retinal diseases using a confocal Laser Scanning Ophthalmoscope. Thirty patients with macular dystrophies and 30 with age-related macular disease underwent serial examinations over a period of 1-3 years in order to monitor the changes over time of fundus autofluorescence. RESULTS: Absent autofluorescence corresponded well spatially with outer retinal atrophy in eyes with retinitis pigmentosa and rod-cone dystrophy. Abnormally high background autofluorescence was seen in the macular region in some patients with dominant and recessive retinitis pigmentosa and rod-cone dystrophies. In areas of macular edema fundus autofluorescence was abnormal. Fundus autofluorescence showed changes over time in most of the eyes with retinal diseases studied. CONCLUSION: Fundus autofluorescence allows documentation of areas of photoreceptor cell loss in eyes with retinitis pigmentosa and rod-cone dystrophies. If abnormal high background autofluorescence in the surviving areas occurs only in some patients with retinitis pigmentosa, the technique may serve to distinguish the regional from the diffuse type of disease. Over time, fundus autofluorescence may demonstrate change or may remain stable.

Adolescent↗

PubMed1983

Spatio-temporal modulation transfer function in retinal diseases.

Spatio-temporal modulation transfer function was measured in patients with retinal disease (central serous choroidopathy, retinal branch vein occlusion, retinitis pigmentosa) using microcomputer controlled monitor-TV display system. Sensitivity loss in high spatial frequency ranges at 1.0 Hz temporal frequency was the characteristic finding in those diseases, and the results suggest that the sustained channel of the visual perception system is more impaired than the transient channel in retinal diseases involving the macula.

Adult↗

PubMed1998

Levodopa-carbidopa and childhood retinal disease.

PURPOSE: Our purpose was to determine the influence of levodopa-carbidopa on visual function in children with retinal disease. METHOD: Two studies were undertaken, a single-dose study and a longitudinal dosing study. A double-masked, placebo controlled single-dose study was undertaken of levodopa-carbidopa (2.08 mg/kg of body weight levodopa with 25% carbidopa) on monocular visual acuity in 14 children with retinal disease. Subjects received two capsules approximately 2.5 hours apart, and monocular visual acuity was measured 2 hours after each capsule ingestion. The second study was a double-masked, placebo-controlled 12-week longitudinal dosing (0.62 mg/kg of body weight) crossover study in which subjects received levodopa-carbidopa for 6 weeks and placebo for 6 weeks. RESULTS: The single-dose study revealed a small but statistically significant improvement in monocular visual acuity after levodopa-carbidopa ingestion. The longitudinal study revealed a small but statistically significant improvement in binocular visual acuity after levodopa ingestion. In both studies placebo had no significant effect on visual acuity. Six subjects participated in both studies and demonstrated a significant correlation (r = 0.76, p < 0.05) between change in visual acuity in the single-dose study and the longitudinal dosing study. CONCLUSION: The results are consistent with the hypothesis that dopamine influences the receptive field characteristics of retinal cells. The results also suggest that there may be low retinal dopamine levels in some types of retinal disease, which may be amenable to treatment.

Adolescent↗

PubMed1999

Automated scanning laser ophthalmoscope image montages of retinal diseases.

PURPOSE: To generate wide-field automated seamless retinal montage images. DESIGN: Prospective, observational, case series study. PARTICIPANTS: Eighteen eyes of 14 patients were studied. INTERVENTION: A digital scanning laser ophthalmoscope (SLO), the Heidelberg Retina Angiograph (HRA), was used to obtain overlapping images of the fundus during fluorescein angiograms, indocyanine green angiograms, or monochromatic retinal imaging using infrared or green light. Software was used to generate seamless montages. MAIN OUTCOME MEASURES: Ability to electronically synthesize retinal montages. Four laser wavelengths were used. The wide-field degrees, vertical and horizontal number of pixels, and diameter of the individual images were measured. RESULTS: High-resolution (pixel size, 10 microm), wide-field, typically 100 degrees to 140 degrees, digital montages of the postequatorial retina can be generated from HRA images. CONCLUSIONS: The software automatically and rapidly aligned the retinal blood vessels and synthesized a montage of the entire fundus that could then be overlaid on images taken at different times to illustrate change. These montages will allow improved ability to understand and follow retinal diseases.

Adolescent↗

PubMed2004

Pattern of retinal diseases at a teaching eye department, Addis Ababa, Ethiopia.

The purpose of this study was to evaluate the pattern of retinal diseases as seen at a teaching and tertiary eye care center in Addis Ababa. In a descriptive, cross-sectional study, all consecutive patients seen at the retina clinic of Menelik II Hospital during a 19 months period (January 2000 to August 2001) were included in this series. Pertinent demographic and clinical data were recorded for all patients. A total of 1390 new patients with retinal diseases were seen at the retina clinic during the study period, accounting for 12.5% of the total outpatient population of the eye department. The male to female ratio was 1.8 : 1. The mean age was 45.2 years +/- 17.3 years (range 2 months to 92 years) and median of 44.5 years. Two hundred and twenty-four (16.1%) patients were bilaterally blind, 465 (33.5%) patients were unilaterally blind, 280 (20.1%) patients had bilateral visual impairment and 195 (14.0%) patients had unilateral visual impairment, while 213 (15.3%) patients had normal vision. Retinal detachment was the commonest cause of both bilateral (54.9%) and unilateral blindness (41.2%), while diabetic retinopathy and myopia were the leading causes of bilateral visual impairment accounting for 36.8% and 28.2% respectively. Retinal vascular diseases accounted for the largest group of patients (38.1%) of which diabetic retinopathy accounted for 75.1%. Retinal detachment was the second largest group of diseases, accounting for 24.5% of the total. The proportion of patients with age-related macular degeneration was only 2.7%. Most of the patients presented with advanced disease, which required vitreo-retinal surgery. There is a need to improve on the early diagnosis and early referral of retinal diseases at primary and secondary care levels. Selected tertiary care centers should develop capacity to provide laser and vitreo-retinal surgery. The introduction of posterior vitrectomy in Ethiopia is long overdue.

Adolescent↗

PubMed1976

Retinal diseases in Nigerians--a preliminary report.

Of 595 new patients seen in the Eye Clinic of Lagos University Teaching Hospital, twenty-seven had retinal diseases, a prevalence of 4.5% Seven of these patients had retinopathy associated with sickle cell haemoglobinopathy and another seven had macular degenerations. Other causes of retinal diseases are analysed and the findings compared with reports from other countries. It is suggested that haemoglobin electrophoretic fractionation should be done in young Nigerians presenting with retinal diseases.

Adult↗

PubMed1994

Symptoms and signs of retinal disease.

Of all the diseases of the eye, the retinal disorders are perhaps the most frequently misdiagnosed by the general medical community. The content of this article is restricted to retinal diseases and the importance of symptoms and signs in their accurate diagnosis.

Fluorescein Angiography↗

PubMed1985

Color contrast perimetry. The spatial distribution of color defects in optic nerve and retinal diseases.

Color contrast perimetry was used to evaluate central visual field defects in a group of 28 patients with visual loss resulting from optic nerve or retinal diseases. Kinetic perimetry was performed using colored test objects of constant luminance, equated to a white surround of 10 ft lamberts. Colored test objects were varied in size and in extent of color saturation. Test object color saturation was varied from a white that matched the color and luminance of the adapting background toward either the blue or the red color maxima of a video tangent screen. All central visual field defects that were demonstrable by luminance contrast perimetry were also detected by color contrast testing, and no defects were found for color contrast detection that could not also be demonstrated by conventional luminance increment perimetry. Retinal diseases usually produced scotomas for both color and luminance contrast detection, while optic nerve disorders tended to produce global depressions of both color and luminance contrast sensitivity across the entire visual field in addition to scotomas. There was no systematic difference in visual field defects for either class of disease when comparing color contrast in the blue (tritan) versus the red (protan) axes of color space. The apparent tritan or protan/deutan axes of color confusion found by hue discrimination testing in acquired dyschromatopsias may be determined by the relative spatial distribution of defects in the central visual field rather than by selective impairment of neural mechanisms for color or luminance information processing.

Adult↗

PubMed2001

Visual hallucinations in patients with retinal disease.

PURPOSE: To investigate the prevalence and phenomenologic nature of visual hallucinations among patients with retinal disease and to investigate whether presence of hallucinations is a significant predictor of functional status, quality of life, and/or emotional distress after adjusting for visual acuity. DESIGN: Cross-sectional study. METHODS: Eighty-six consecutive patients at the Wilmer Ophthalmologic Institute Retinal Vascular Center were interviewed using the Sickness Impact Profile, Community Disability Scale, General Health Questionnaire, Visual Phenomena Interview, Eysenck Personality Questionnaire, and Telephone Interview for Cognitive Status. RESULTS: The prevalence of visual hallucinations was 15.1%. Most were formed hallucinations in clear consciousness that lasted for seconds to minutes. The majority of patients had been experiencing visual hallucinations for less than 1 year (61.5%) or for 1 to 2 years (23.1%). Only two of the 13 patients with hallucinations had informed a physician of their hallucinations. Univariate analyses revealed that variables significantly associated with experiencing hallucinations were female sex, worse visual acuity, bilateral visual impairment, emotional distress, decreased functional status, and decreased quality of life. Regression analysis demonstrated that among patients with relatively good vision, those who experienced hallucinations were more emotionally distressed and had a lower quality of life than patients without hallucinations. CONCLUSIONS: Visual hallucinations among patients with retinal disease are common, underdiagnosed, and not associated with cognitive deficits, abnormal personality traits, or a family or personal history of psychiatric morbidity. Among patients with relatively good vision, hallucinations are associated with increased emotional distress and decreased quality of life.

Cross-Sectional Studies↗

PubMed1982

Contrast sensitivity measurement in retinal diseases by laser generated sinusoidal grating.

The contrast sensitivity function (CSF) of 83 patients with retinal diseases was measured by laser-generated sinusoidal grating pattern, which enables the direct measurement of the CSF between the retina and the brain. The CSF abnormality in various retinal diseases was not specific to each disease, and the severity may determine the pattern. The abnormal patterns and rough correlation with visual acuity (V.A.) are: (1) attenuation at high frequency ranges (V.A. greater than 0.8), (2) attenuation at intermediate and high frequency ranges (0.4 less than V.A. less than 0.7), (3) attenuation at all frequency ranges (V.A. less than 0.3). Some cases, however, showed discrepancy between visual acuity and CSF. At the recovery stage of central serous retinopathy and retinal branch vein occlusion, CSF showed delayed recovery or attenuated sensitivity even when visual acuity was restored to 1.0 or more. In retinitis pigmentosa and pre-retinal macular fibrosis, CSF showed more sensitive detection of abnormal macular function than visual acuity measurement. At the post-corticosteroid therapy of Harada's disease and Sympathetic Ophthalmia, CSF recovered more rapidly than visual acuity when retinal detachment was absorbed. Those discrepancies suggest that CSF measurement may be useful for evaluating progression or regression of these diseases.

Adolescent↗

PubMed2003

HPMPC therapy of MCMV-induced retinal disease in the SCID mouse measured by electroretinography, a non-invasive technique.

The purpose of these studies was to investigate the use of non-invasive electroretinography for the evaluation of retinal disease and its treatment in an ocular murine cytomegalovirus (MCMV) disease model. While under anesthesia, 10(2.6)plaque forming units (pfu) of salivary gland passaged, Smith strain MCMV was injected in the anterior chamber of 6- to 8-week-old severe combined immunodeficiency (SCID) mice. At various times post-inoculation, bright-flash scotopic electroretinogram, viral titer, and histology were obtained from the injected eye. Antiviral therapy was tested using 0.1 and 5mg/kg/day subcutaneous injections of HPMPC (Cidofovir) once daily for 5 consecutive days. In infected animals, the a- and b-waves of the electroretinographic (ERG) signal were significantly reduced as of 10 days post-inoculation when compared to control animals. Therapy with HPMPC 0.1mg/kg/day subcutaneously (s.c.) once daily for 5 consecutive days was able to delay the decrease in ERG wave amplitude and inhibit viral replication, whereas 5mg/kg/day s.c. significantly protected the ERG, completely inhibited viral replication, and maintained ocular viral titer below the limit of detection for up to 17 days post-infection. The reduction of ERG activity during progression of retinal disease correlated well with reduction of disease pathology. ERG recording represents a valuable non-invasive technique to measure the progression of the retinal disease induced by MCMV and the efficacy of antiviral treatment in the ocular MCMV disease model.

Animals↗