ArticlePloS one2011
Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior.
Article in PloS one, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 24 papers.
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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
24 citing papers in PubMed, 48 citations in OpenAlex.
- Diverse species of animal models in epilepsy research: Progress and perspectives.Zoological research · 2025Review
- Congenital hydrocephalus: a review of recent advances in genetic etiology and molecular mechanisms.Military Medical Research · 2024Review
- Genetic and molecular mechanisms of hydrocephalus.Frontiers in molecular neuroscience · 2024Review
- Dichloroacetate improves mitochondrial function, physiology, and morphology in FBXL4 disease models.JCI insight · 2022Article
- Aquatic Freshwater Vertebrate Models of Epilepsy Pathology: Past Discoveries and Future Directions for Therapeutic Discovery.International journal of molecular sciences · 2022Review
- Biallelic and monoallelic variants in PLXNA1 are implicated in a novel neurodevelopmental disorder with variable cerebral and eye anomalies.Genetics in medicine : official journal of the American College of Medical Genetics · 2021Article
- Imaging epilepsy in larval zebrafish.European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society · 2020Review
- Unveiling Tumor Microenvironment Interactions Using Zebrafish Models.Frontiers in molecular biosciences · 2020Review
- Pyridoxamine Supplementation Effectively Reverses the Abnormal Phenotypes of Zebrafish Larvae With PNPO Deficiency.Frontiers in pharmacology · 2019Article
- Depdc5 knockdown causes mTOR-dependent motor hyperactivity in zebrafish.Annals of clinical and translational neurology · 2018Article
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- Suppression of breast cancer metastasis through the inactivation of ADP-ribosylation factor 1.Oncotarget · 2016Article
- Toxicity of 3,5,6-trichloro-2-pyridinol tested at multiple stages of zebrafish (Danio rerio) development.Environmental science and pollution research international · 2016Article
- Developmental toxic effects of monocrotophos, an organophosphorous pesticide, on zebrafish (Danio rerio) embryos.Environmental science and pollution research international · 2015Article
- Zebrafish cytosolic carboxypeptidases 1 and 5 are essential for embryonic development.The Journal of biological chemistry · 2013Article
- Evaluating human cancer cell metastasis in zebrafish.BMC cancer · 2013Article
- In vivo modeling of the morbid human genome using Danio rerio.Journal of visualized experiments : JoVE · 2013Article
- LGI proteins in the nervous system.ASN neuro · 2013Review
- Mechanisms of prickle1a function in zebrafish epilepsy and retinal neurogenesis.Disease models & mechanisms · 2013Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
7 authors at 2 institutions in 1 country.
Funding
Abstract
Mutations in the LGI1 gene predispose to a hereditary epilepsy syndrome and is the first gene associated with this disease which does not encode an ion channel protein. In zebrafish, there are two paralogs of the LGI1 gene, lgi1a and lgi1b. Knockdown of lgi1a results in a seizure-like hyperactivity phenotype with associated developmental abnormalities characterized by cellular loss in the eyes and brain. We have now generated knockdown morphants for the lgi1b gene which also show developmental abnormalities but do not show a seizure-like behavior. Instead, the most striking phenotype involves significant enlargement of the ventricles (hydrocephalus). As shown for the lgi1a morphants, however, lgi1b morphants are also sensitized to PTZ-induced hyperactivity. The different phenotypes between the two lgi1 morphants support a subfunctionalization model for the two paralogs.
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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.