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

Autor(es)

Victorino, Daniella B. ^{[1, 2]} ; Scott-McKean, Jonah J. ^{[1, 3]} ; Johnson, Mark W. ^{[1, 3]} ; Costa, Alberto C. S. ^{[1, 3, 4]}

Afiliação do(s) autor(es)

^[1] Case Western Reserve Univ, Dept Pediat, Div Pediat Neurol, 11100 Euclid Ave Mail Stop RBC 6090, Cleveland, OH 44106 - USA

^[2] Univ Fed Sao Paulo, Postgrad Program Neurol & Neurosci, Sao Paulo - Brazil

^[3] Costa, Alberto C. S., Case Western Reserve Univ, Dept Psychiat, Cleveland, OH 44106 USA.Victorino, Daniella B., Case Western Reserve Univ, Dept Pediat, Div Pediat Neurol, 11100 Euclid Ave Mail Stop RBC 6090, Cleveland, OH 44106 - USA

^[4] Case Western Reserve Univ, Dept Psychiat, Cleveland, OH 44106 - USA

• Tipo de documento: Artigo Científico
• Fonte: INVESTIGATIVE OPHTHALMOLOGY & VISUAL SCIENCE; v. 61, n. 5 MAY 2020.
• Citações Web of Science: 0

Resumo

PURPOSE. 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. (AU)

• Processo FAPESP: 16/17746-3 - Avaliação dos efeitos de benzodiazepínicos sobre a atividade eletrofisiológica cerebral in vivo e a viabilidade neuronal in vitro em camundongos da linhagem Ts65Dn
• Beneficiário: Daniella Balduino Victorino
• Modalidade de apoio: Bolsas no Brasil - Doutorado
• Processo FAPESP: 18/10131-9 - Efeitos da modulação alostérica positiva sobre as propriedades eletrofisiológicas da sinalização mediada por receptores GABAA em camundongos Ts65Dn e neurônios derivados de células-tronco pluripotentes induzidas de pessoas com a síndrome de Down
• Beneficiário: Daniella Balduino Victorino
• Modalidade de apoio: Bolsas no Exterior - Estágio de Pesquisa - Doutorado