ArticleDevelopment (Cambridge, England)2009
Mutations in zebrafish leucine-rich repeat-containing six-like affect cilia motility and result in pronephric cysts, but have variable effects on left-right patterning.
Article in Development (Cambridge, England), 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 37 papers, 1 of them a synthesis that pooled it.
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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
37 citing papers in PubMed, 1 synthesis or guideline pooled it, 53 citations in OpenAlex.
- Laterality defects are influenced by timing of treatments and animal model.Differentiation; research in biological diversity · 2012Pooled it
- DynAPs and cytoplasmic assembly of axonemal dyneins.Journal of cell science · 2025Article
- Variable phenotypes and penetrance between and within different zebrafish ciliary transition zone mutants.Disease models & mechanisms · 2022Article
- The effect of a novel LRRC6 mutation on the flagellar ultrastructure in a primary ciliary dyskinesia patient.Journal of assisted reproduction and genetics · 2021Article
- Aquatic models of human ciliary diseases.Genesis (New York, N.Y. : 2000) · 2021Review
- Defects in the cytoplasmic assembly of axonemal dynein arms cause morphological abnormalities and dysmotility in sperm cells leading to male infertility.PLoS genetics · 2021Article
- Article
- Zebrafish as a Model for Human Ciliopathies.Journal of genetics and genomics = Yi chuan xue bao · 2016Review
- c21orf59/kurly Controls Both Cilia Motility and Polarization.Cell reports · 2016Article
- Ccdc11 is a novel centriolar satellite protein essential for ciliogenesis and establishment of left-right asymmetry.Molecular biology of the cell · 2016Article
- Cdc42 and sec10 Are Required for Normal Retinal Development in Zebrafish.Investigative ophthalmology & visual science · 2015Article
- Mechanisms of otoconia and otolith development.Developmental dynamics : an official publication of the American Association of Anatomists · 2015Review
- Unique among ciliopathies: primary ciliary dyskinesia, a motile cilia disorder.F1000prime reports · 2015Review
- Symmetry breakage in the vertebrate embryo: when does it happen and how does it work?Developmental biology · 2014Review
- Small heat shock proteins are necessary for heart migration and laterality determination in zebrafish.Developmental biology · 2013Article
- It's never too early to get it Right: A conserved role for the cytoskeleton in left-right asymmetry.Communicative & integrative biology · 2013Article
- Cdc42 deficiency causes ciliary abnormalities and cystic kidneys.Journal of the American Society of Nephrology : JASN · 2013Article
- Differential roles for 3-OSTs in the regulation of cilia length and motility.Development (Cambridge, England) · 2013Article
- ZMYND10 is mutated in primary ciliary dyskinesia and interacts with LRRC6.American journal of human genetics · 2013Article
- Reptin/Ruvbl2 is a Lrrc6/Seahorse interactor essential for cilia motility.Proceedings of the National Academy of Sciences of the United States of America · 2013Article
Corrections and comments
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Authors and funding
11 authors at 2 institutions in 1 country.
Funding
Abstract
Cilia defects have been implicated in a variety of human diseases and genetic disorders, but how cilia motility contributes to these phenotypes is still unknown. To further our understanding of how cilia function in development, we have cloned and characterized two alleles of seahorse, a zebrafish mutation that results in pronephric cysts. seahorse encodes Lrrc6l, a leucine-rich repeat-containing protein that is highly conserved in organisms that have motile cilia. seahorse is expressed in zebrafish tissues known to contain motile cilia. Although mutants do not affect cilia structure and retain the ability to interact with Disheveled, both alleles of seahorse strongly affect cilia motility in the zebrafish pronephros and neural tube. Intriguingly, although seahorse mutations variably affect fluid flow in Kupffer's vesicle, they can have very weak effects on left-right patterning. Combined with recently published results, our alleles suggest that the function of seahorse in cilia motility is separable from its function in other cilia-related phenotypes.
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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.