Multifocal electroretinographic responses in sector retinitis pigmentosa
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Electroretinography
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In clinical electrophysiology AC-recorded ERGs are used for the investigation of a- and b-wave parameters. The direct coupled (DC-ERG) ist used to record the c-wave. Theoretically the DC-ERG technique has less influence on the recorded potentials than the AC-ERG technique. We recorded AC- and DC-ERGs of 20 normal eyes, 17 eyes with x-linked congenital retinoschisis and 30 eyes with retinal degenerations. In normal eyes no difference was found between AC- und DC-ERG concerning a- and b-wave amplitudes, latencies and implicit times, the b/a-ratios and the oscillatory potentials. The 30 Hz flicker amplitude was higher in the DC-ERG (p less than 0.0003). The intraindividual variability of all measured parameters was comparable between AC- and DC-ERG. In x-linked retinoschisis the b-wave amplitudes and the b/a-ratios were lower in the DC-ERG than in the AC-ERG. In retinal degenerative diseases the a-wave amplitudes were higher and the dark-adapted b-wave amplitudes lower in the DC-ERG, therefore the b/a-ratio was lower in the DC-ERG. We conclude, the DC-ERG is a reproducible method for clinical routine investigations. It is comparable to the conventional AC-ERG technique. In certain diseases the pathological features of the electroretinogram were more distinct in the DC-ERG than the AC-ERG.
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Acute experimental retinal degeneration was induced in 3-month-old mice with sodium iodate (NaIO3) injection to investigate the effect of bright light with electroretinography (ERG). Eight C 57 black male mice were anesthetized. The ERG was recorded before and 24 h after the injection of NaIO3. Next, only one eye of each mouse was exposed to a bright light of 3.0 x 10(4) lx white light for 30 min. Ten hours later, ERGs of both the light-exposed and the unexposed eye were recorded. The amplitudes and peak latencies of the a wave and b wave were measured. The ERG was recorded as both eyes were exposed to stimulating flashes that were given in an increasing order of 0.6 log unit steps from the dimmest flash to produce a detectable ERG. The ERG from the light-exposed eye showed a distinctly elevated threshold (approx 2.0 log), while the contralateral unexposed eye did not. The peak latencies of both waves were significantly prolonged by exposure to light. In conclusion, light exposure affected ERG thresholds in mice with experimentally induced acute retinal degeneration.
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Psychophysical and electroretinographic (ERG) testing are used to evaluate retinal function in a Negro family with a recessively inherited retinitis pigmentosa. The abnormal ERGs with the autosomal recessive pattern of inheritance distinguish the retinal pigmentary degeneration in this family from other genetic types previously classified with electroretinography. All affected children have retinal disease visible with the ophthalmoscope. The youngest child has a reduction in amplitude and delay in timing of her cone ERG before any rod system abnormality can be detected. The older affected children have a reduction in amplitude and delay in implicit time of both cone and rod components. The reduced and delayed rod ERG to a relatively bright short wavelength light was simulated by presenting a relatively dim short wavelength light to normal subjects. The relative preservation of cone ERG amplitudes is discussed as a measure of severity of different types of retinitis pigmentosa in childhood.
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Many retinal degenerative diseases result in vision impairment or permanent blindness due to photoreceptor loss or dysfunction. It has been observed that Pde6brd1 mice (rd1), which carry a spontaneous nonsense mutation in the pde6b gene, have a strong phenotypic similarity to patients suffering from autosomal recessive retinitis pigmentosa. In this study, we present a novel mouse model of retinitis pigmentosa generated through pde6b gene knockout using CRISPR/Cas9 technology. We compare this Pde6b-KO mouse model to the rd1 mouse model to gain insights into the progression of retinal degeneration. The functional assessment of the mouse retina and the tracking of degeneration dynamics were performed using electrophysiological methods, while retinal morphology was analyzed through histology techniques. Interestingly, the Pde6b-KO mouse model demonstrated a higher amplitude of photoresponse than the rd1 model of the same age. At postnatal day 12, the thickness of the photoreceptor layer in both mouse models did not significantly differ from that of control animals; however, by day 15, a substantial reduction was observed. Notably, the decline in the number of photoreceptors in the rd1 model occurred at a significantly faster rate. These findings suggest that the C3H background may play a significant role in the early stages of retinal degeneration.
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The Electroretinography (ERG) is a noninvasive technique that allows the assessment of functional integrity of the retina. The ERG recordings are biopotencials acquired in the corneal surface as a response of retinal tissue against controlled light stimuli. In clinical ophthalmology ERG is not commonly used but nowadays, because of the high incidence of degenerative diseases of the retina (RD), its use should be increased. Like other biopotentials as electrocardiography (ECG), electroencephalogram (EEG) and electromyography (EMG), ERG is a low amplitude signal, in this case a few hundred of microvolts (µV), which must be fitted and processed. The ERG signals are affected in morphology in the presence of pathologies that affects the integrity of the different retinal cell groups, for example due to some RD. In advanced cases of RD recordings can be abolished in the time domain; and yet in them it is believed that there is relevant clinical information making the ERG a great potential diagnostic tool.
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Abstract Retinitis pigmentosa (RP) is a heterogeneous disease and the main cause of vision loss within the group of inherited retinal diseases (IRDs). IRDs are a group of rare disorders caused by mutations in one or more of over 280 genes which ultimately result in blindness. Modifier genes play a key role in modulating disease phenotypes, and mutations in them can affect disease outcomes, rate of progression, and severity. Our previous studies have demonstrated that the nuclear hormone receptor 2 family e, member 3 ( Nr2e3 ) gene reduced disease progression and loss of photoreceptor cell layers in Rho P23H − / − mice. This follow up, pharmacology study evaluates a longitudinal NR2E3 dose response in the clinically relevant heterozygous Rho P23H mouse. Reduced retinal degeneration and improved retinal morphology was observed 6 months following treatment evaluating three different NR2E3 doses. Histological and immunohistochemical analysis revealed regions of photoreceptor rescue in the treated retinas of Rho P23H+/ − mice. Functional assessment by electroretinogram (ERG) showed attenuated photoreceptor degeneration with all doses. This study demonstrates the effectiveness of different doses of NR2E3 at reducing retinal degeneration and informs dose selection for clinical trials of Rho P23H -associated RP.
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