ArticlePloS one2012
The Onecut transcription factor HNF-6 regulates in motor neurons the formation of the neuromuscular junctions.
Article in PloS one, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed, 28 citations in OpenAlex.
- Postnatal relevance of HOXA5 transcription factor in cerebellum-associated behaviors and disorders.Behavioral and brain functions : BBF · 2026Article
- Apolipoprotein D neofunctionalization couples lipid allocation to wing evolution.The EMBO journal · 2026Article
- Neurotrophin-3 produced by motor neurons non-cell autonomously regulate the development of pre-motor interneurons in the developing spinal cord.bioRxiv : the preprint server for biology · 2026Article
- Regulation of motor neuron differentiation in thebioRxiv : the preprint server for biology · 2025Article
- Arid3c identifies an uncharacterized subpopulation of V2 interneurons during embryonic spinal cord development.Frontiers in cellular neuroscience · 2024Article
- A modERN resource: identification of Drosophila transcription factor candidate target genes using RNAi.Genetics · 2023Article
- CUT homeobox genes: transcriptional regulation of neuronal specification and beyond.Frontiers in cellular neuroscience · 2023Review
- microRNA-124 regulates Notch and NeuroD1 to mediate transition states of neuronal development.Developmental neurobiology · 2023Article
- Long-Term Treatment with Bortezomib Induces Specific Methylation Changes in Differentiated Neuronal Cells.Cancers · 2022Article
- Onecut Regulates Core Components of the Molecular Machinery for Neurotransmission in Photoreceptor Differentiation.Frontiers in cell and developmental biology · 2021Article
- How to Build and to Protect the Neuromuscular Junction: The Role of the Glial Cell Line-Derived Neurotrophic Factor.International journal of molecular sciences · 2020Review
- Onecut-dependent Nkx6.2 transcription factor expression is required for proper formation and activity of spinal locomotor circuits.Scientific reports · 2020Article
- Onecut Factors and Pou2f2 Regulate the Distribution of V2 Interneurons in the Mouse Developing Spinal Cord.Frontiers in cellular neuroscience · 2019Article
- A Multi-step Transcriptional and Chromatin State Cascade Underlies Motor Neuron Programming from Embryonic Stem Cells.Cell stem cell · 2017Article
- The Onecut Transcription Factors Regulate Differentiation and Distribution of Dorsal Interneurons during Spinal Cord Development.Frontiers in molecular neuroscience · 2017Article
- Expression of Terminal Effector Genes in Mammalian Neurons Is Maintained by a Dynamic Relay of Transient Enhancers.Neuron · 2016Article
- Onecut transcription factors in development and disease.Trends in developmental biology · 2016Article
- Overelaborated synaptic architecture and reduced synaptomatrix glycosylation in a Drosophila classic galactosemia disease model.Disease models & mechanisms · 2014Article
- Generating spinal motor neuron diversity: a long quest for neuronal identity.Cellular and molecular life sciences : CMLS · 2014Review
- Motor neurons and the generation of spinal motor neuron diversity.Frontiers in cellular neuroscience · 2014Review
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Authors and funding
8 authors at 3 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The neuromuscular junctions are the specialized synapses whereby spinal motor neurons control the contraction of skeletal muscles. The formation of the neuromuscular junctions is controlled by a complex interplay of multiple mechanisms coordinately activated in motor nerve terminals and in their target myotubes. However, the transcriptional regulators that control in motor neurons the genetic programs involved in neuromuscular junction development remain unknown. Here, we provide evidence that the Onecut transcription factor HNF-6 regulates in motor neurons the formation of the neuromuscular junctions. Indeed, adult Hnf6 mutant mice exhibit hindlimb muscle weakness and abnormal locomotion. This results from defects of hindlimb neuromuscular junctions characterized by an abnormal morphology and defective localization of the synaptic vesicle protein synaptophysin at the motor nerve terminals. These defects are consequences of altered and delayed formation of the neuromuscular junctions in newborn mutant animals. Furthermore, we show that the expression level of numerous regulators of neuromuscular junction formation, namely agrin, neuregulin-2 and TGF-ß receptor II, is downregulated in the spinal motor neurons of Hnf6 mutant newborn animals. Finally, altered formation of neuromuscular junction-like structures in a co-culture model of wildtype myotubes with mutant embryonic spinal cord slices is rescued by recombinant agrin and neuregulin, indicating that depletion in these factors contributes to defective neuromuscular junction development in the absence of HNF-6. Thus, HNF-6 controls in spinal motor neurons a genetic program that coordinates the formation of hindlimb neuromuscular junctions.
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