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Stabilizing axin leads to optic nerve hypoplasia in a mouse model of autism.
Stabilizing axin leads to optic nerve hypoplasia in a mouse model of autism.
- Source :
-
Experimental eye research [Exp Eye Res] 2024 Aug; Vol. 245, pp. 109988. Date of Electronic Publication: 2024 Jul 02. - Publication Year :
- 2024
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Abstract
- Autism spectrum disorder (ASD) is a group of neurodevelopment disorders characterized by deficits in social interaction and communication, and repetitive or stereotyped behavior. Autistic children are more likely to have vision problems, and ASD is unusually common among blind people. However, the mechanisms behind the vision disorders in autism are unclear. Stabilizing WNT-targeted scaffold protein Axin2 by XAV939 during embryonic development causes overproduction of cortical neurons and leads to autistic-like behaviors in mice. In this study, we investigated the relationship between vision abnormality and autism using an XAV939-induced mouse model of autism. We found that the mice receiving XAV939 had decreased amplitude of bright light-adaptive ERG. The amplitudes and latency of flash visual evoked potential recorded from XAV939-treated mice were lower and longer, respectively than in the control mice, suggesting that XAV939 inhibits visual signal processing and conductance. Anatomically, the diameters of RGC axons were reduced when Axin2 was stabilized during the development, and the optic fibers had defective myelin sheaths and reduced oligodendrocytes. The results suggest that the WNT signaling pathway is crucial for optic nerve development. This study provides experimental evidence that conditions interfering with brain development may also lead to visual problems, which in turn might exaggerate the autistic features in humans.<br />Competing Interests: Declaration of competing interest The authors have no competing interests to declare.<br /> (Copyright © 2024 Elsevier Ltd. All rights reserved.)
- Subjects :
- Animals
Mice
Electroretinography
Mice, Inbred C57BL
Axons pathology
Retinal Ganglion Cells pathology
Retinal Ganglion Cells metabolism
Male
Wnt Signaling Pathway physiology
Autism Spectrum Disorder physiopathology
Autism Spectrum Disorder metabolism
Autistic Disorder physiopathology
Autistic Disorder metabolism
Axin Protein metabolism
Disease Models, Animal
Evoked Potentials, Visual physiology
Optic Nerve metabolism
Optic Nerve pathology
Subjects
Details
- Language :
- English
- ISSN :
- 1096-0007
- Volume :
- 245
- Database :
- MEDLINE
- Journal :
- Experimental eye research
- Publication Type :
- Academic Journal
- Accession number :
- 38964496
- Full Text :
- https://doi.org/10.1016/j.exer.2024.109988