ArticleNeuroscience2020
Mutations in Ciliary Trafficking Genes affect Sonic Hedgehog-dependent Neural Tube Patterning Differentially along the Anterior-Posterior Axis.
Article in Neuroscience, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.
What it found
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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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Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
7 citing papers in PubMed, 11 citations in OpenAlex.
- Light- and chemical-induced ciliary signaling governs dorsal/ventral regionalization of human telencephalic organoids.Nature communications · 2026Article
- Tulp3 quantitative alleles titrate requirements for viability, brain development, and kidney homeostasis but do not suppress Zfp423 mutations in mice.PLoS genetics · 2025Article
- Article
- A defined tubby domain β-barrel surface region of TULP3 mediates ciliary trafficking of diverse cargoes.Molecular biology of the cell · 2025Article
- A pathogenic variant of TULP3 causes renal and hepatic fibrocystic disease.Frontiers in genetics · 2022Article
- Identification of new candidate genes for spina bifida through exome sequencing.Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery · 2021Article
- Patterns of cilia gene dysregulations in major psychiatric disorders.Progress in neuro-psychopharmacology & biological psychiatry · 2021Article
Corrections and comments
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Authors and funding
2 authors at 1 institution in 1 country.
Funding
Abstract
Cell specification in the ventral spinal cord is a well-studied model system to understand how tissue pattern develops in response to a morphogen gradient. Ventral cell types including motor neurons (MNs) are induced in the neural tube in response to graded Sonic Hedgehog (Shh) signaling. We performed a forward genetic screen in the mouse that incorporated a GFP-expressing transgene to visualize MNs to identify genes regulating ventral patterning. Here we contrast the neural patterning phenotypes of two mouse lines carrying induced mutations in ciliary trafficking genes. We show that a hypomorphic mutation in the gene Tubby-like protein 3 (Tulp3) resulted in a dorsal expansion of MNs consistent with an up-regulation of Shh signaling. Interestingly, patterning defects in Tulp3 mutants were restricted to posterior regions of the spinal cord as patterning was similar to WT in the anterior spinal cord. In contrast, a mutation in the ciliary trafficking gene cytoplasmic dynein 2 heavy chain 1 (Dync2h1), led to a complete loss of MNs in anterior regions of the spinal cord, indicating a strong down-regulation of Shh signaling. However, this severe phenotype was restricted to the cervical region as MNs developed posteriorly. Mutations in cilia trafficking genes affect Shh-dependent signaling in the neural tube differentially along the anterior-posterior (A-P) axis in a process that is not understood.
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What OpenQuestion holds
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.