ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2013
A morpholino-based screen to identify novel genes involved in craniofacial morphogenesis.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 33 papers, 1 of them a synthesis that pooled it.
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33 citing papers in PubMed, 1 synthesis or guideline pooled it, 62 citations in OpenAlex.
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- Genetic insight into the putative causal proteins and druggable targets of osteoporosis: a large-scale proteome-wide mendelian randomization study.Frontiers in genetics · 2023Article
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- Satb2 acts as a gatekeeper for major developmental transitions during early vertebrate embryogenesis.Nature communications · 2021Article
- The Mandibular and Hyoid Arches-From Molecular Patterning to Shaping Bone and Cartilage.International journal of molecular sciences · 2021Review
- Suppression of Cell Tumorigenicity by Non-neural Pro-differentiation Factors via Inhibition of Neural Property in Tumorigenic Cells.Frontiers in cell and developmental biology · 2021Article
- Deficiency of the SMOC2 matricellular protein impairs bone healing and produces age-dependent bone loss.Scientific reports · 2020Article
- Neuron-Derived Neurotrophic Factor Is Mutated in Congenital Hypogonadotropic Hypogonadism.American journal of human genetics · 2020Article
- Secreted modular calcium-binding proteins in pathophysiological processes and embryonic development.Chinese medical journal · 2019Review
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- Gene silencing through RNAi and antisense Vivo-Morpholino increases the efficacy of pyrethroids on larvae of Anopheles stephensi.Malaria journal · 2019Article
- Heterozygous loss-of-function variants of MEIS2 cause a triad of palatal defects, congenital heart defects, and intellectual disability.European journal of human genetics : EJHG · 2019Article
- Master Regulators of Signaling Pathways: An Application to the Analysis of Gene Regulation in Breast Cancer.Frontiers in genetics · 2019Article
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- Identification of stiffness-induced signalling mechanisms in cells from patent and fused sutures associated with craniosynostosis.Scientific reports · 2017Article
Corrections and comments
- Erratum issued
Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
backgroundThe regulatory mechanisms underpinning facial development are conserved between diverse species. Therefore, results from model systems provide insight into the genetic causes of human craniofacial defects. Previously, we generated a comprehensive dataset examining gene expression during development and fusion of the mouse facial prominences. Here, we used this resource to identify genes that have dynamic expression patterns in the facial prominences, but for which only limited information exists concerning developmental function.
resultsThis set of ∼80 genes was used for a high-throughput functional analysis in the zebrafish system using Morpholino gene knockdown technology. This screen revealed three classes of cranial cartilage phenotypes depending upon whether knockdown of the gene affected the neurocranium, viscerocranium, or both. The targeted genes that produced consistent phenotypes encoded proteins linked to transcription (meis1, meis2a, tshz2, vgll4l), signaling (pkdcc, vlk, macc1, wu:fb16h09), and extracellular matrix function (smoc2). The majority of these phenotypes were not altered by reduction of p53 levels, demonstrating that both p53-dependent and -independent mechanisms were involved in the craniofacial abnormalities.
conclusionsThis Morpholino-based screen highlights new genes involved in development of the zebrafish craniofacial skeleton with wider relevance to formation of the face in other species, particularly mouse and human.
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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.