ArticlePLoS genetics2005
The zebrafish mutants dre, uki, and lep encode negative regulators of the hedgehog signaling pathway.
Article in PLoS genetics, 2005. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
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.
The trial behind it
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.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
43 citing papers in PubMed, 86 citations in OpenAlex.
- Cannabidiol exerts teratogenic effects on developing zebrafish through the sonic hedgehog signaling pathway.Scientific reports · 2025Article
- Anterior-posterior constraint on Hedgehog signaling by hhip in teleost fin elaboration.Development (Cambridge, England) · 2024Article
- A simple and scalable zebrafish model of Sonic hedgehog medulloblastoma.Cell reports · 2024Article
- Understanding the Roles of the Hedgehog Signaling Pathway during T-Cell Lymphopoiesis and in T-Cell Acute Lymphoblastic Leukemia (T-ALL).International journal of molecular sciences · 2023Review
- Infection by the ParasiteMicroorganisms · 2022Article
- Insights Gained From Zebrafish Models for the Ciliopathy Joubert Syndrome.Frontiers in genetics · 2022Review
- Genetic Engineering of Zebrafish in Cancer Research.Cancers · 2020Review
- QDPR homologues in Danio rerio regulate melanin synthesis, early gliogenesis, and glutamine homeostasis.PloS one · 2019Article
- Hedgehog pathway mutations drive oncogenic transformation in high-risk T-cell acute lymphoblastic leukemia.Leukemia · 2018Article
- Gene expression profile of a selection of Polycomb Group genes during zebrafish embryonic and germ line development.PloS one · 2018Article
- Biophysical characterisation of the novel zinc binding property in Suppressor of Fused.Scientific reports · 2017Article
- Targeted resequencing identifies PTCH1 as a major contributor to ocular developmental anomalies and extends the SOX2 regulatory network.Genome research · 2016Article
- Sonic hedgehog functions upstream of disrupted-in-schizophrenia 1 (disc1): implications for mental illness.Biology open · 2015Article
- Secreted HHIP1 interacts with heparan sulfate and regulates Hedgehog ligand localization and function.The Journal of cell biology · 2015Article
- Regulation of Hedgehog signaling by ubiquitination.Frontiers in biology · 2015Article
- Endothelial cilia are essential for developmental vascular integrity in zebrafish.Journal of the American Society of Nephrology : JASN · 2015Article
- Article
- Positive and negative regulation of Gli activity by Kif7 in the zebrafish embryo.PLoS genetics · 2013Article
- An ENU mutagenesis screen in zebrafish for visual system mutants identifies a novel splice-acceptor site mutation in patched2 that results in Colobomas.Investigative ophthalmology & visual science · 2012Article
- Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
8 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Proliferation is one of the basic processes that control embryogenesis. To identify factors involved in the regulation of proliferation, we performed a zebrafish genetic screen in which we used proliferating cell nuclear antigen (PCNA) expression as a readout. Two mutants, hu418B and hu540A, show increased PCNA expression. Morphologically both mutants resembled the dre (dreumes), uki (ukkie), and lep (leprechaun) mutant class and both are shown to be additional uki alleles. Surprisingly, although an increased size is detected of multiple structures in these mutant embryos, adults become dwarfs. We show that these mutations disrupt repressors of the Hedgehog (Hh) signaling pathway. The dre, uki, and lep loci encode Su(fu) (suppressor of fused), Hip (Hedgehog interacting protein), and Ptc2 (Patched2) proteins, respectively. This class of mutants is therefore unique compared to previously described Hh mutants from zebrafish genetic screens, which mainly show loss of Hh signaling. Furthermore, su(fu) and ptc2 mutants have not been described in vertebrate model systems before. Inhibiting Hh activity by cyclopamine rescues uki and lep mutants and confirms the overactivation of the Hh signaling pathway in these mutants. Triple uki/dre/lep mutants show neither an additive increase in PCNA expression nor enhanced embryonic phenotypes, suggesting that other negative regulators, possibly Ptc1, prevent further activation of the Hh signaling pathway. The effects of increased Hh signaling resulting from the genetic alterations in the uki, dre, and lep mutants differ from phenotypes described as a result of Hh overexpression and therefore provide additional insight into the role of Hh signaling during vertebrate development.
Indexed as
Identifiers
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.