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Retinal Development in Infants and Young Children with Achromatopsia.

Lee H, Purohit R, Sheth V, McLean RJ, Kohl S, Leroy BP, Sundaram V, Michaelides M, Proudlock FA, Gottlob I - Ophthalmology (2015)

View Article: PubMed Central - PubMed

Affiliation: The University of Leicester Ulverscroft Eye Unit, Robert Kilpatrick Clinical Sciences Building, Leicester Royal Infirmary, Leicester, UK.

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Normally, postnatal development of the human retina involves centrifugal displacement of the inner retinal layers (IRLs) from the fovea, centripetal migration of the cone photoreceptors into the fovea, and elongation of the photoreceptors with age... In all of the participants with ACHM, there was evidence of foveal hypoplasia (presence of the normally absent IRLs at the fovea) at each visit on OCT examination... To define the differences between the ACHM and control groups with regard to retinal layer thickness measurements and their rate of change over time, linear mixed modelling was performed using STATA (Copyright 1996–2014, StataCorp)... Overall retinal thickness measurements and rate of increase in retinal thickness with age were reduced significantly at the fovea, parafovea (500 μm from the central fovea), and perifovea (1500 μm from the central fovea) in ACHM (Fig 2A)... The foveal IRLs were 5 times thicker in ACHM than mean control values (P < 0.0001), as a result of significantly increased thicknesses of the ganglion cell (GCL), inner plexiform, inner nuclear (INL), and outer plexiform (OPL) layers... There was an age-related decrease in foveal IRL thickness in ACHM, owing to regression of the GCL, INL, and OPL (Fig 2B)... In contrast, the foveal outer retinal layers (ORLs) in ACHM were significantly thinner, being 0.6 times thinner than mean control values (P < 0.0001), which was attributable to reductions in the photoreceptor inner segment (IS), outer segment (OS), and outer nuclear layer (Fig 2C)... Mixed linear regression analysis showed a significant negative correlation between the thickness of the ORLs and the thickness of the IRLs at the fovea (β = −0.52; P < 0.0001), which suggests that cone photoreceptors influence IRL migration during retinal development... The parafoveal ORLs were uniformly thinner in ACHM at all ages (P < 0.0001), owing to reductions in IS, OS, and retinal pigment epithelium (RPE) measurements... Perifoveal ORL thickness in ACHM did not differ from controls at birth... A contrasting pattern of changes in the RPE takes place across all measured retinal locations, where the RPE becomes thinner with age in ACHM and thicker with age in controls... We have shown that retinal development is not arrested in children with ACHM, but is ongoing albeit at a reduced rate and magnitude in comparison with controls, with consequences for all retinal layers.

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Examples of the developmental trajectories for the retinal thickness (RT), inner retinal layers (IRLs), outer retinal layers (ORLs), inner plexiform layer (IPL), and outer plexiform layer (OPL) at the fovea and temporal perifovea and the photoreceptor inner segment (IS) and photoreceptor outer segment (OS) at the fovea. The upper plots for each panel show the trajectories plotted over a time period spanning 0–120 months postnatal age. Each point represents a single value from each optical coherence tomography (OCT) examination. The lines of best fit (trend lines) are shown in red and blue for the achromatopsia (ACHM) and control groups, respectively. The lower plots for each panel represent the difference between the best fit lines for ACHM and control groups (in the upper plots) with the error bars representing the 95% confidence intervals. By calculating partial derivatives of the interaction term the significant differences between ACHM and control groups were estimated at 13 specified time points, namely, 6, 12, 18, 24, 36, 42, 48, 54, 60, 66, 72, and 78 months postnatal age. Red colors indicate where there is a significantly thinner retinal layer in ACHM and blue colors a significantly thicker retinal layer in ACHM with increasing depth of color representing the level of significance.
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fig2: Examples of the developmental trajectories for the retinal thickness (RT), inner retinal layers (IRLs), outer retinal layers (ORLs), inner plexiform layer (IPL), and outer plexiform layer (OPL) at the fovea and temporal perifovea and the photoreceptor inner segment (IS) and photoreceptor outer segment (OS) at the fovea. The upper plots for each panel show the trajectories plotted over a time period spanning 0–120 months postnatal age. Each point represents a single value from each optical coherence tomography (OCT) examination. The lines of best fit (trend lines) are shown in red and blue for the achromatopsia (ACHM) and control groups, respectively. The lower plots for each panel represent the difference between the best fit lines for ACHM and control groups (in the upper plots) with the error bars representing the 95% confidence intervals. By calculating partial derivatives of the interaction term the significant differences between ACHM and control groups were estimated at 13 specified time points, namely, 6, 12, 18, 24, 36, 42, 48, 54, 60, 66, 72, and 78 months postnatal age. Red colors indicate where there is a significantly thinner retinal layer in ACHM and blue colors a significantly thicker retinal layer in ACHM with increasing depth of color representing the level of significance.

Mentions: To define the differences between the ACHM and control groups with regard to retinal layer thickness measurements and their rate of change over time, linear mixed modelling was performed using STATA (Copyright 1996–2014, StataCorp). Overall retinal thickness measurements and rate of increase in retinal thickness with age were reduced significantly at the fovea, parafovea (500 μm from the central fovea), and perifovea (1500 μm from the central fovea) in ACHM (Fig 2A).


Retinal Development in Infants and Young Children with Achromatopsia.

Lee H, Purohit R, Sheth V, McLean RJ, Kohl S, Leroy BP, Sundaram V, Michaelides M, Proudlock FA, Gottlob I - Ophthalmology (2015)

Examples of the developmental trajectories for the retinal thickness (RT), inner retinal layers (IRLs), outer retinal layers (ORLs), inner plexiform layer (IPL), and outer plexiform layer (OPL) at the fovea and temporal perifovea and the photoreceptor inner segment (IS) and photoreceptor outer segment (OS) at the fovea. The upper plots for each panel show the trajectories plotted over a time period spanning 0–120 months postnatal age. Each point represents a single value from each optical coherence tomography (OCT) examination. The lines of best fit (trend lines) are shown in red and blue for the achromatopsia (ACHM) and control groups, respectively. The lower plots for each panel represent the difference between the best fit lines for ACHM and control groups (in the upper plots) with the error bars representing the 95% confidence intervals. By calculating partial derivatives of the interaction term the significant differences between ACHM and control groups were estimated at 13 specified time points, namely, 6, 12, 18, 24, 36, 42, 48, 54, 60, 66, 72, and 78 months postnatal age. Red colors indicate where there is a significantly thinner retinal layer in ACHM and blue colors a significantly thicker retinal layer in ACHM with increasing depth of color representing the level of significance.
© Copyright Policy - CC BY
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4582068&req=5

fig2: Examples of the developmental trajectories for the retinal thickness (RT), inner retinal layers (IRLs), outer retinal layers (ORLs), inner plexiform layer (IPL), and outer plexiform layer (OPL) at the fovea and temporal perifovea and the photoreceptor inner segment (IS) and photoreceptor outer segment (OS) at the fovea. The upper plots for each panel show the trajectories plotted over a time period spanning 0–120 months postnatal age. Each point represents a single value from each optical coherence tomography (OCT) examination. The lines of best fit (trend lines) are shown in red and blue for the achromatopsia (ACHM) and control groups, respectively. The lower plots for each panel represent the difference between the best fit lines for ACHM and control groups (in the upper plots) with the error bars representing the 95% confidence intervals. By calculating partial derivatives of the interaction term the significant differences between ACHM and control groups were estimated at 13 specified time points, namely, 6, 12, 18, 24, 36, 42, 48, 54, 60, 66, 72, and 78 months postnatal age. Red colors indicate where there is a significantly thinner retinal layer in ACHM and blue colors a significantly thicker retinal layer in ACHM with increasing depth of color representing the level of significance.
Mentions: To define the differences between the ACHM and control groups with regard to retinal layer thickness measurements and their rate of change over time, linear mixed modelling was performed using STATA (Copyright 1996–2014, StataCorp). Overall retinal thickness measurements and rate of increase in retinal thickness with age were reduced significantly at the fovea, parafovea (500 μm from the central fovea), and perifovea (1500 μm from the central fovea) in ACHM (Fig 2A).

View Article: PubMed Central - PubMed

Affiliation: The University of Leicester Ulverscroft Eye Unit, Robert Kilpatrick Clinical Sciences Building, Leicester Royal Infirmary, Leicester, UK.

AUTOMATICALLY GENERATED EXCERPT
Please rate it.

Normally, postnatal development of the human retina involves centrifugal displacement of the inner retinal layers (IRLs) from the fovea, centripetal migration of the cone photoreceptors into the fovea, and elongation of the photoreceptors with age... In all of the participants with ACHM, there was evidence of foveal hypoplasia (presence of the normally absent IRLs at the fovea) at each visit on OCT examination... To define the differences between the ACHM and control groups with regard to retinal layer thickness measurements and their rate of change over time, linear mixed modelling was performed using STATA (Copyright 1996–2014, StataCorp)... Overall retinal thickness measurements and rate of increase in retinal thickness with age were reduced significantly at the fovea, parafovea (500 μm from the central fovea), and perifovea (1500 μm from the central fovea) in ACHM (Fig 2A)... The foveal IRLs were 5 times thicker in ACHM than mean control values (P < 0.0001), as a result of significantly increased thicknesses of the ganglion cell (GCL), inner plexiform, inner nuclear (INL), and outer plexiform (OPL) layers... There was an age-related decrease in foveal IRL thickness in ACHM, owing to regression of the GCL, INL, and OPL (Fig 2B)... In contrast, the foveal outer retinal layers (ORLs) in ACHM were significantly thinner, being 0.6 times thinner than mean control values (P < 0.0001), which was attributable to reductions in the photoreceptor inner segment (IS), outer segment (OS), and outer nuclear layer (Fig 2C)... Mixed linear regression analysis showed a significant negative correlation between the thickness of the ORLs and the thickness of the IRLs at the fovea (β = −0.52; P < 0.0001), which suggests that cone photoreceptors influence IRL migration during retinal development... The parafoveal ORLs were uniformly thinner in ACHM at all ages (P < 0.0001), owing to reductions in IS, OS, and retinal pigment epithelium (RPE) measurements... Perifoveal ORL thickness in ACHM did not differ from controls at birth... A contrasting pattern of changes in the RPE takes place across all measured retinal locations, where the RPE becomes thinner with age in ACHM and thicker with age in controls... We have shown that retinal development is not arrested in children with ACHM, but is ongoing albeit at a reduced rate and magnitude in comparison with controls, with consequences for all retinal layers.

Show MeSH
Related in: MedlinePlus