Pattern-reversal visual evoked potentials in Down's syndrome

Kakigi, Ryusuke; Oono, Shinji; Matsuda, Yuki; Kuroda, Yasuo

doi:10.1111/j.1600-0404.1993.tb04126.x

Cited by 9 publications

(11 citation statements)

References 16 publications

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“…Nevertheless, we are also aware that we have not addressed other ophthalmological and neuroophthalmological disorders frequently seen in persons with DS, such as strabismus, spontaneous and latent nystagmi, deficiencies in accommodation, and refractive errors, which may also be phenotypes of the Ts65Dn mouse and may help explain some of our findings. It is important to point out, however, that in a previously published human VEP study, even subjects with DS who did not present any of these ophthalmological and neuroophthalmological disorders showed significant reductions in VEP amplitude as compared to typical control individuals 12. In addition, altered dendrite arbors and dendritic spine morphology have been described in brain specimens from persons with DS as well as Ts65Dn mice 19, 20, which provide some support to a potential neural sensory origin to the pVEP phenotypes we observed in Ts65Dn mice.…”

Section: Discussionmentioning

confidence: 86%

“…Because a significant component of the visual deficits seen in persons with DS cannot be ameliorated through the use of corrective lenses, there is a strong possibility that a portion of such deficits are due to dysfunction of central visual pathways 17. This notion is supported by the finding of abnormal visual evoked potentials (VEPs) in both adults 12, 18 and children 15, 17 with DS. Additional evidence for a neuronal origin of some visual deficits in persons with DS comes from the finding of altered dendritic and synaptic morphology in the cortex of individuals with DS 19, 20.…”

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confidence: 99%

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The Mouse Model of Down Syndrome Ts65Dn Presents Visual Deficits as Assessed by Pattern Visual Evoked Potentials

Scott-McKean¹,

Chang

Hurd

et al. 2010

Invest. Ophthalmol. Vis. Sci.

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Purpose The Ts65Dn mouse is the most complete widely available animal model for Down syndrome (DS). To generate quantitative information about visual function in the Ts65Dn mouse, we investigated their visual capabilities by means of electroretinography (ERG) and patterned visual evoked potentials (pVEPs). Methods pVEPs were recorded directly from specific regions of the binocular visual cortex of anesthetized mice in response to horizontal sinusoidal gratings of different spatial frequency, contrast, and luminance generated by a specialized video card and presented on a 21” computer display suitably linearized by gamma correction. Results ERG assessments indicated no significant deficit in retinal physiology in Ts65Dn mice compared to euploid controls mice. We found, however, that Ts65Dn mice exhibit deficits in luminance threshold, spatial resolution, and contrast threshold compared to euploid control mice. The behavioral counterparts of these parameters are luminance sensitivity, visual acuity, and the inverse of contrast sensitivity, respectively. Conclusions DS includes various phenotypes associated with the visual system, including deficits in visual acuity, accommodation, and contrast sensitivity. The present study provides electrophysiological evidence of visual deficits in Ts65Dn mice that are similar to those reported in persons with DS. These findings strengthen the role of the Ts65Dn mouse as a model for DS. Also, given the historical assumption of integrity of the visual system most behavioral assessments of Ts65Dn mice, such as the hidden platform component of the Morris water maze, the visual deficits described here might represent a significant confounding factor in the interpretation of results from such experiments.

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Section: Discussionmentioning

confidence: 86%

mentioning

confidence: 99%

The Mouse Model of Down Syndrome Ts65Dn Presents Visual Deficits as Assessed by Pattern Visual Evoked Potentials

Scott-McKean¹,

Chang

Hurd

et al. 2010

Invest. Ophthalmol. Vis. Sci.

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“…Intermediate-level visual processing abnormalities have also been reported, including abnormal visual evoked potentials (VEPs) in children 13,14,24,25 and adults with DS. 14,24,26 Mouse models of DS have been essential tools for investigating the molecular pathogenesis and pathophysiology of DS. In particular, the segmentally trisomic Ts65Dn mouse has been the most widely used mouse model of DS and is the most complete in terms of mimicking phenotypes seen in persons with DS, including impaired learning and memory.…”

mentioning

confidence: 99%

Quantitative Analysis of Retinal Structure and Function in Two Chromosomally Altered Mouse Models of Down Syndrome

Victorino

Scott-McKean

Johnson

et al. 2020

Invest. Ophthalmol. Vis. Sci.

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Ophthalmic disorders are among the most prevalent Down syndrome (DS) comorbidities. Therefore, when studying mouse models of DS, ignoring how vision is affected can lead to misinterpretation of results from assessments dependent on the integrity of the visual system. Here, we used imaging and electroretinography (ERG) to study eye structure and function in two important mouse models of DS: Ts65Dn and Dp(16)1Yey/+. METHODS. Cornea and anterior segment were examined with a slit-lamp. Thickness of retinal layers was quantified by optical coherence tomography (OCT). Eye and lens dimensions were measured by magnetic resonance imaging (MRI). Retinal vasculature parameters were assessed by bright field and fluorescent imaging, and by retinal flatmount preparations. Ganzfeld ERG responses to flash stimuli were used to assess retinal function in adult mice. RESULTS. Total retinal thickness is significantly increased in Ts65Dn and Dp(16)1Yey/+ compared with control mice, because of increased thickness of inner retinal layers, including the inner nuclear layer (INL). Increased retinal vessel caliber was found in both chromosomally altered mice when compared with controls. ERG responses in Ts65Dn and Dp(16)1Yey/+ mice showed subtle alterations compared with controls. These, however, seemed to be unrelated to the thickness of the INL, but instead dependent on the anesthetic agent used (ketamine, tribromoethanol, or urethane). CONCLUSIONS. We provide evidence of retinal alterations in Ts65Dn and Dp(16)1Yey/+ mice that are similar to those reported in persons with DS. Our ERG results are also a reminder that consideration should be given to the choice of anesthetic agents in such experiments.

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“…A literature review of children with Down syndrome age 0-16 years revealed that refractive error, strabismus, poor acuity, nystagmus, and blepharitis were common ophthalmologic findings whereas cataract and glaucoma were less common (Creavin & Brown, 2009). A pattern reversal VEP study demonstrated significantly longer P100 latency and smaller amplitude in DS patients (16/36 cases) as compared to agematched controls (Kakigi et al, 1993). By employing achromatic transient VEP, children with DS had small or undetectable N75 but normal latency as compared to normal developing children (Suttle & Turner, 2004).…”

Section: Information From Down Syndromementioning

confidence: 99%