ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2011
The Down syndrome critical region regulates retinogeniculate refinement.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.
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Who cites it
33 citing papers in PubMed, 50 citations in OpenAlex.
- Future Directions for Studying the Potential of Mammalian Dscam in Autism Spectrum Disorder and Alzheimer's Disease: Insights from Dose Sensitivity.Molecular neurobiology · 2025Review
- Prevalence and diversity of retinal disease in adults with Down syndrome.Eye (London, England) · 2025Review
- The role of Down syndrome cell adhesion molecule in Down syndrome.Medical review (2021) · 2024Review
- DSCAM is differentially patterned along the optic axon pathway in the developing Xenopus visual system and guides axon termination at the target.Neural development · 2022Article
- All Creatures Great and Small: New Approaches for Understanding Down Syndrome Genetics.Trends in genetics : TIG · 2021Review
- Suprachiasmatic lesions restore object recognition in down syndrome model mice.Neurobiology of sleep and circadian rhythms · 2020Article
- ZebrafishThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2020Article
- Nuclear import of the DSCAM-cytoplasmic domain drives signaling capable of inhibiting synapse formation.The EMBO journal · 2019Article
- Combination of Genetics and Nanotechnology for Down Syndrome Modification: A Potential Hypothesis and Review of the Literature.Iranian journal of public health · 2019Article
- Advances in understanding the association between Down syndrome and Hirschsprung disease (DS-HSCR).Pediatric surgery international · 2018Review
- DSCAM differentially modulates pre- and postsynaptic structural and functional central connectivity during visual system wiring.Neural development · 2018Article
- Age- and speed-dependent modulation of gaits in DSCAMJournal of neurophysiology · 2018Article
- A third copy of the Down syndrome cell adhesion molecule (Dscam) causes synaptic and locomotor dysfunction in Drosophila.Neurobiology of disease · 2018Article
- DSCAM-mediated control of dendritic and axonal arbor outgrowth enforces tiling and inhibits synaptic plasticity.Proceedings of the National Academy of Sciences of the United States of America · 2017Article
- Activity-Dependent Dysfunction in Visual and Olfactory Sensory Systems in Mouse Models of Down Syndrome.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2017Article
- DSCAM promotes axon fasciculation and growth in the developing optic pathway.Proceedings of the National Academy of Sciences of the United States of America · 2017Article
- Synaptopathies: synaptic dysfunction in neurological disorders - A review from students to students.Journal of neurochemistry · 2016Review
- Novel axon projection after stress and degeneration in the Dscam mutant retina.Molecular and cellular neurosciences · 2016Article
- Role of DSCAM in the development of the spinal locomotor and sensorimotor circuits.Journal of neurophysiology · 2016Article
- DSCAM promotes refinement in the mouse retina through cell death and restriction of exploring dendrites.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2015Article
Corrections and comments
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Authors and funding
11 authors at 5 institutions in 1 country.
Funding
Abstract
Down syndrome (DS) is a developmental disorder caused by a third chromosome 21 in humans (Trisomy 21), leading to neurological deficits and cognitive impairment. Studies in mouse models of DS suggest that cognitive deficits in the adult are associated with deficits in synaptic learning and memory mechanisms, but it is unclear whether alterations in the early wiring and refinement of neuronal circuits contribute to these deficits. Here, we show that early developmental refinement of visual circuits is perturbed in mouse models of Down syndrome. Specifically, we find excessive eye-specific segregation of retinal axons in the dorsal lateral geniculate nucleus. Indeed, the degree of refinement scales with defects in the "Down syndrome critical region" (DSCR) in a dose-dependent manner. We further identify Dscam (Down syndrome cell adhesion molecule), a gene within the DSCR, as a regulator of eye-specific segregation of retinogeniculate projections. Although Dscam is not the sole gene in the DSCR contributing to enhanced refinement in trisomy, Dscam dosage clearly regulates cell spacing and dendritic fasciculation in a specific class of retinal ganglion cells. Thus, altered developmental refinement of visual circuits that occurs before sensory experience is likely to contribute to visual impairment in individuals with Down syndrome.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.