ArticleMolecular and cellular biology2005
Loss of the serine/threonine kinase fused results in postnatal growth defects and lethality due to progressive hydrocephalus.
Article in Molecular and cellular biology, 2005. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 54 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
54 citing papers in PubMed, 130 citations in OpenAlex.
- Illumination of understudied ciliary kinases.Frontiers in molecular biosciences · 2024Review
- Ulk4 promotes Shh signaling by regulating Stk36 ciliary localization and Gli2 phosphorylation.eLife · 2023Article
- ULK4 and Fused/STK36 interact to mediate assembly of a motile flagellum.Molecular biology of the cell · 2023Article
- Hedgehog signaling mechanism and role in cancer.Seminars in cancer biology · 2022Review
- Ptpn20 deletion in H-Tx rats enhances phosphorylation of the NKCC1 cotransporter in the choroid plexus: an evidence of genetic risk for hydrocephalus in an experimental study.Fluids and barriers of the CNS · 2022Article
- Gli Phosphorylation Code in Hedgehog Signal Transduction.Frontiers in cell and developmental biology · 2022Review
- ULK4 in Neurodevelopmental and Neuropsychiatric Disorders.Frontiers in cell and developmental biology · 2022Review
- Article
- Motile cilia genetics and cell biology: big results from little mice.Cellular and molecular life sciences : CMLS · 2021Review
- Genes causing congenital hydrocephalus: Their chromosomal characteristics of telomere proximity and DNA compositions.Experimental neurology · 2021Review
- Next generation DNA sequencing of atypical choroid plexus papilloma of brain: Identification of novel mutations in a female patient by Ion Proton.Oncology letters · 2019Article
- Phosphorylation of Ci/Gli by Fused Family Kinases Promotes Hedgehog Signaling.Developmental cell · 2019Article
- The emerging roles of phosphatases in Hedgehog pathway.Cell communication and signaling : CCS · 2017Review
- Ventricular Zone Disruption in Human Neonates With Intraventricular Hemorrhage.Journal of neuropathology and experimental neurology · 2017Article
- Ulk4 Is Essential for Ciliogenesis and CSF Flow.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2016Article
- Ubr3, a Novel Modulator of Hh Signaling Affects the Degradation of Costal-2 and Kif7 through Poly-ubiquitination.PLoS genetics · 2016Article
- RestrictedJournal of developmental biology · 2016Article
- Sonic hedgehog patterning during cerebellar development.Cellular and molecular life sciences : CMLS · 2016Review
- The medaka dhc2 mutant reveals conserved and distinct mechanisms of Hedgehog signaling in teleosts.BMC developmental biology · 2015Article
- The Hedgehog pathway: role in cell differentiation, polarity and proliferation.Archives of toxicology · 2015Review
Corrections and comments
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Authors and funding
13 authors at 3 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The Drosophila Fused (Fu) kinase is an integral component of the Hedgehog (Hh) pathway that helps promote Hh-dependent gene transcription. Vertebrate homologues of Fu function in the Hh pathway in vitro, suggesting that Fu is evolutionarily conserved. We have generated fused (stk36) knockout mice to address the in vivo function of the mouse Fu (mFu) homologue. fused knockouts develop normally, being born in Mendelian ratios, but fail to thrive within 2 weeks, displaying profound growth retardation with communicating hydrocephalus and early mortality. The fused gene is expressed highly in ependymal cells and the choroid plexus, tissues involved in the production and circulation of cerebral spinal fluid (CSF), suggesting that loss of mFu disrupts CSF homeostasis. Similarly, fused is highly expressed in the nasal epithelium, where fused knockouts display bilateral suppurative rhinitis. No obvious defects were observed in the development of organs where Hh signaling is required (limbs, face, bones, etc.). Specification of neuronal cell fates by Hh in the neural tube was normal in fused knockouts, and induction of Hh target genes in numerous tissues is not affected by the loss of mFu. Furthermore, stimulation of fused knockout cerebellar granule cells to proliferate with Sonic Hh revealed no defect in Hh signal transmission. These results show that the mFu homologue is not required for Hh signaling during embryonic development but is required for proper postnatal development, possibly by regulating the CSF homeostasis or ciliary function.
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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.