ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2015
A hypomorphic allele reveals an important role of inturned in mouse skeletal development.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers, 1 of them a synthesis that pooled it.
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Who cites it
13 citing papers in PubMed, 1 synthesis or guideline pooled it, 20 citations in OpenAlex.
- Combined genome-wide association study of 136 quantitative ear morphology traits in multiple populations reveal 8 novel loci.PLoS genetics · 2023Pooled it
- Moderate levels of folic acid benefit outcomes for cilia based neural tube defects.Developmental biology · 2025Article
- CPLANE protein INTU regulates growth and patterning of the mouse lungs through cilia-dependent Hh signaling.Developmental biology · 2024Article
- Increasing Sufu gene dosage reveals its unorthodox role in promoting polydactyly and medulloblastoma tumorigenesis.JCI insight · 2024Article
- Mecom mutation related to radioulnar synostosis with amegakaryocytic thrombocytopenia reduces HSPCs in mice.Blood advances · 2023Article
- A direct interaction between CENTLEIN and RABIN8 is required for primary cilium formation.Acta biochimica et biophysica Sinica · 2023Article
- TMEM132A regulates mouse hindgut morphogenesis and caudal development.Development (Cambridge, England) · 2023Article
- Wdpcp regulates cellular proliferation and differentiation in the developing limb via hedgehog signaling.BMC developmental biology · 2021Article
- Hexa-Longin domain scaffolds for inter-Rab signalling.Bioinformatics (Oxford, England) · 2020Article
- INTU is essential for oncogenic Hh signaling through regulating primary cilia formation in basal cell carcinoma.Oncogene · 2017Article
- Integration of over 9,000 mass spectrometry experiments builds a global map of human protein complexes.Molecular systems biology · 2017Article
- Genetic advances in craniosynostosis.American journal of medical genetics. Part A · 2017Review
- From Planar Cell Polarity to Ciliogenesis and Back: The Curious Tale of the PPE and CPLANE proteins.Trends in cell biology · 2017Review
Corrections and comments
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Authors and funding
4 authors at 2 institutions in 1 country.
Funding
Abstract
backgroundCilia are important for Hedgehog signaling in vertebrates and many genes that encode proteins involved in ciliogenesis have been studied for their roles in embryonic development. Null mutations in many of these genes cause early embryonic lethality, hence an understanding of their roles in postnatal development is limited.
resultsThe Inturned (Intu) gene is required for ciliogenesis and here we report a recessive hypomorphic mutation, resulting in substitution of a conserved hydrophobic residue (I813N) near the C-terminus, that sheds light on later functions of Intu. Mice homozygous for this Double-thumb (Intu(Dtm)) allele exhibit polydactyly, retarded growth, and reduced survival. There is a moderate loss of cilia in Intu(Dtm/Dtm) mutants, and Intu(I813N) exhibits compromised ability to increase ciliogenesis in cultured Intu null mutant cells. Intu(Dtm) mutants show rib defects and delay of endochondral ossification in long bones, digits, vertebrae, and the sternum. These skeletal defects correlate with a decrease in Hh signaling. However, patterning of the neural tube and planar cell polarity appear to be normal.
conclusionsThis hypomorphic Intu allele highlights an important role of Intu in mouse skeletal development.
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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.