ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2006
Central roles of the roof plate in telencephalic development and holoprosencephaly.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2006. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 58 papers.
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Who cites it
58 citing papers in PubMed, 102 citations in OpenAlex.
- Ophthalmologic Findings in an Induced Model of Holoprosencephaly in Zebrafish.The Journal of comparative neurology · 2025Article
- Sequential emergence and contraction of epithelial subtypes in the prenatal human choroid plexus revealed by a stem cell model.Nature communications · 2025Article
- Human endothelial cells promote a human neural stem cell type B phenotype via Notch signaling.Nature communications · 2025Article
- Holoprosencephaly spectrum: an up-to-date overview of classification, genetics and neuroimaging.Japanese journal of radiology · 2025Review
- Pax6 limits the competence of developing cerebral cortical cells to respond to inductive intercellular signals.PLoS biology · 2022Article
- From Neural Crest to Definitive Roof Plate: The Dynamic Behavior of the Dorsal Neural Tube.International journal of molecular sciences · 2021Review
- The Polycomb group protein Ring1 regulates dorsoventral patterning of the mouse telencephalon.Nature communications · 2020Article
- Early dorsomedial tissue interactions regulate gyrification of distal neocortex.Nature communications · 2019Article
- Cajal-Retzius neurons are required for the development of the human hippocampal fissure.Journal of anatomy · 2019Article
- Case report: a novel mutation in ZIC2 in an infant with microcephaly, holoprosencephaly, and arachnoid cyst.Medicine · 2019Article
- Extracephalic manifestations of nonchromosomal, nonsyndromic holoprosencephaly.American journal of medical genetics. Part C, Seminars in medical genetics · 2018Review
- Cell-Biological Requirements for the Generation of Dentate Gyrus Granule Neurons.Frontiers in cellular neuroscience · 2018Review
- TGF-β Family Signaling in Neural and Neuronal Differentiation, Development, and Function.Cold Spring Harbor perspectives in biology · 2017Review
- Brorin is required for neurogenesis, gliogenesis, and commissural axon guidance in the zebrafish forebrain.PloS one · 2017Article
- BMP4 acts as a dorsal telencephalic morphogen in a mouse embryonic stem cell culture system.Biology open · 2016Article
- Transcriptional and epigenetic mechanisms of early cortical development: An examination of how Pax6 coordinates cortical development.The Journal of comparative neurology · 2016Review
- Generation of functional hippocampal neurons from self-organizing human embryonic stem cell-derived dorsomedial telencephalic tissue.Nature communications · 2015Article
- Development and functions of the choroid plexus-cerebrospinal fluid system.Nature reviews. Neuroscience · 2015Review
- Semi-adaptive response and noise attenuation in bone morphogenetic protein signalling.Journal of the Royal Society, Interface · 2015Article
- A novel (paternally inherited) duplication 13q31.3q32.3 in a 12-year-old patient with facial dysmorphism and developmental delay.Molecular syndromology · 2014Article
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
Abstract
The roof plate is a well known signaling center in CNS development, but its roles in the developing telencephalon and the common holoprosencephaly (HPE) malformation have been uncertain. Using cellular ablations in mice, we show that roof plate cell loss causes failed midline induction and HPE in the dorsal telencephalon. This morphologic phenotype is accompanied by selective deficits in midline gene expression and a reduced activity gradient for bone morphogenetic proteins (Bmps), the major signals produced by the roof plate. In dissociated cells and mutant explants, exogenous Bmp4 is sufficient to mimic roof plate selectivity in midline gene regulation and to rescue roof plate-dependent midline patterning. Previously unrecognized neuroanatomical defects predicted by the mouse model are then confirmed in human HPE patients. These findings establish selective roles for roof plate-dependent Bmp signaling in dorsal telencephalic patterning and HPE and define novel candidate genes for the human disorder.
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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.