ArticlePloS one2010
Cleft palate is caused by CNS dysfunction in Gad1 and Viaat knockout mice.
Article in PloS one, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 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
17 citing papers in PubMed, 28 citations in OpenAlex.
- Epigenetic Mechanisms in Cleft Lip and Palate: A Comprehensive Review of Current Insights and Future Perspectives.Plastic and reconstructive surgery. Global open · 2026Article
- Developmental dynamics of catshark cranial neural crest cells provide insights into gnathostome facial evolution.Development (Cambridge, England) · 2026Article
- Ventral body wall closure: Mechanistic insights from mouse models and translation to human pathology.Developmental dynamics : an official publication of the American Association of Anatomists · 2025Review
- Decreased GABA levels during development result in increased connectivity in the larval zebrafish tectum.bioRxiv : the preprint server for biology · 2024Article
- Inhibitory Synaptic Influences on Developmental Motor Disorders.International journal of molecular sciences · 2023Review
- Ultra-Rare Variants Identify Biological Pathways and Candidate Genes in the Pathobiology of Non-Syndromic Cleft Palate Only.Biomolecules · 2023Article
- Review
- Regulation of GABATrends in neurosciences · 2021Review
- Early-infantile onset epilepsy and developmental delay caused by bi-allelic GAD1 variants.Brain : a journal of neurology · 2020Article
- Bi-allelic GAD1 variants cause a neonatal onset syndromic developmental and epileptic encephalopathy.Brain : a journal of neurology · 2020Article
- RDH10 function is necessary for spontaneous fetal mouth movement that facilitates palate shelf elevation.Disease models & mechanisms · 2019Article
- Determinants of orofacial clefting I: Effects of 5-Aza-2'-deoxycytidine on cellular processes and gene expression during development of the first branchial arch.Reproductive toxicology (Elmsford, N.Y.) · 2017Article
- Nucleotide Variants of the BH4 Biosynthesis Pathway Gene GCH1 and the Risk of Orofacial Clefts.Molecular neurobiology · 2016Article
- Molecular basis of cleft palates in mice.World journal of biological chemistry · 2015Review
- GABAergic neurons regulate lateral ventricular development via transcription factor Pax5.Genesis (New York, N.Y. : 2000) · 2013Article
- Synaptic inhibition and γ-aminobutyric acid in the mammalian central nervous system.Proceedings of the Japan Academy. Series B, Physical and biological sciences · 2013Review
- SDF and GABA interact to regulate axophilic migration of GnRH neurons.Journal of cell science · 2012Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
4 authors at 2 institutions in 1 country.
Funding
Abstract
backgroundPrevious studies have shown that disruption of GABA signaling in mice via mutations in the Gad1, Gabrb3 or Viaat genes leads to the development of non-neural developmental defects such as cleft palate. Studies of the Gabrb3 and Gad1 mutant mice have suggested that GABA function could be required either in the central nervous system or in the palate itself for normal palatogenesis. METHODOLOGY/PRINCIPAL
findingsTo further examine the role of GABA signaling in palatogenesis we used three independent experimental approaches to test whether Gad1 or Viaat function is required in the fetal CNS for normal palate development. We used oral explant cultures to demonstrate that the Gad1 and Viaat mutant palates were able to undergo palatogenesis in culture, suggesting that there is no defect in the palate tissue itself in these mice. In a second series of experiments we found that the GABA(A) receptor agonist muscimol could rescue the cleft palate phenotype in Gad1 and Viaat mutant embryos. This suggested that normal multimeric GABA(A) receptors in the CNS were necessary for normal palatogenesis. In addition, we showed that CNS-specific inactivation of Gad1 was sufficient to disrupt palate development. CONCLUSIONS/SIGNIFICANCE: Our results are consistent with a role for Gad1 and Viaat in the central nervous system for normal development of the palate. We suggest that the alterations in GABA signaling lead to non-neural defects such as cleft palate as a secondary effect due to alterations in or elimination of fetal movements.
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.