ArticleProceedings of the National Academy of Sciences of the United States of America2008
Ultrasonic vocalization impairment of Foxp2 (R552H) knockin mice related to speech-language disorder and abnormality of Purkinje cells.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 106 papers, 1 of them a synthesis that pooled it.
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106 citing papers in PubMed, 1 synthesis or guideline pooled it, 247 citations in OpenAlex.
- Genetic and molecular mechanisms of the FOXP2-CNTNAP2 pathway in Autism spectrum disorders and speech disorders.Journal of neural transmission (Vienna, Austria : 1996) · 2026Pooled it
- Activity-dependent development of vocal circuits in the neonatal rodent forebrain.EMBO reports · 2026Article
- Cerebellar pathology contributes to neurodevelopmental deficits in spinal muscular atrophy.Brain : a journal of neurology · 2026Article
- Cerebellar Abnormalities: A Component of Autism Pathophysiology.Medicina (Kaunas, Lithuania) · 2026Review
- Foxp2 mutations and abnormal brain and gastrointestinal development: insights from animal models of speech-language and autism spectrum disorders.Frontiers in neuroanatomy · 2026Review
- Cerebellar organoids model cell type-specific FOXP2 expression during human cerebellar development.Disease models & mechanisms · 2025Article
- FOXP genes regulate Purkinje cell diversity and cerebellar morphogenesis.Nature neuroscience · 2025Article
- Abnormal development of gastrointestinal system of homozygous Foxp2(R552H)-mutated mice.Communications biology · 2025Article
- DNA binding and mitotic phosphorylation protect polyglutamine proteins from assembly formation.Cell · 2025Article
- Mu-opioid receptor knockout on Foxp2-expressing neurons reduces aversion-resistant alcohol drinking.Pharmacology, biochemistry, and behavior · 2025Article
- TLX3 regulates CGN progenitor proliferation during cerebellum development and its dysfunction can lead to autism.iScience · 2024Article
- Activity of forkhead box P2-positive neurons is associated with tadpole begging behaviour.Biology letters · 2024Article
- Activity of FoxP2-positive neurons is associated with tadpole begging behavior.bioRxiv : the preprint server for biology · 2024Article
- Bayesian semiparametric Markov renewal mixed models for vocalization syntax.Biostatistics (Oxford, England) · 2024Article
- Neurodevelopmental deficits and cell-type-specific transcriptomic perturbations in a mouse model of HNRNPU haploinsufficiency.PLoS genetics · 2023Article
- Speech- and language-linked FOXP2 mutation targets protein motors in striatal neurons.Brain : a journal of neurology · 2023Article
- Identification and characterization of transcribed enhancers during cerebellar development through enhancer RNA analysis.BMC genomics · 2023Article
- High-precision spatial analysis of mouse courtship vocalization behavior reveals sex and strain differences.Scientific reports · 2023Article
- Article
- Modelling Autism Spectrum Disorder (ASD) and Attention-Deficit/Hyperactivity Disorder (ADHD) Using Mice and Zebrafish.International journal of molecular sciences · 2022Review
46 more citing papers are in PubMed but not listed here.
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7 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Previous studies have demonstrated that mutation in the forkhead domain of the forkhead box P2 (FOXP2) protein (R553H) causes speech-language disorders. To further analyze FOXP2 function in speech learning, we generated a knockin (KI) mouse for Foxp2 (R552H) [Foxp2 (R552H)-KI], corresponding to the human FOXP2 (R553H) mutation, by homologous recombination. Homozygous Foxp2 (R552H)-KI mice showed reduced weight, immature development of the cerebellum with incompletely folded folia, Purkinje cells with poor dendritic arbors and less synaptophysin immunoreactivity, and achieved crisis stage for survival 3 weeks after birth. At postnatal day 10, these mice also showed severe ultrasonic vocalization (USV) and motor impairment, whereas the heterozygous Foxp2 (R552H)-KI mice exhibited modest impairments. Similar to the wild-type protein, Foxp2 (R552H) localized in the nuclei of the Purkinje cells and the thalamus, striatum, cortex, and hippocampus (CA1) neurons of the homozygous Foxp2 (R552H)-KI mice (postnatal day 10), and some of the neurons showed nuclear aggregates of Foxp2 (R552H). In addition to the immature development of the cerebellum, Foxp2 (R552H) nuclear aggregates may further compromise the function of the Purkinje cells and cerebral neurons of the homozygous mice, resulting in their death. In contrast, heterozygous Foxp2 (R552H)-KI mice, which showed modest impairment of USVs with different USV qualities and which did not exhibit nuclear aggregates, should provide insights into the common molecular mechanisms between the mouse USV and human speech learning and the relationship between the USV and motor neural systems.
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