ArticleDevelopmental biology2016
Novel α-tubulin mutation disrupts neural development and tubulin proteostasis.
Article in Developmental biology, 2016. 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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Who cites it
24 citing papers in PubMed, 44 citations in OpenAlex.
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- Methylome-wide and meQTL analysis helps to distinguish treatment response from non-response and pathogenesis markers in schizophrenia.Frontiers in psychiatry · 2024Article
- Culturing Primary Cortical Neurons for Live-Imaging.Methods in molecular biology (Clifton, N.J.) · 2024Article
- Article
- Quantifying Yeast Microtubules and Spindles Using the Toolkit for Automated Microtubule Tracking (TAMiT).Biomolecules · 2023Article
- Tubulin isotype regulation maintains asymmetric requirement for α-tubulin over β-tubulin.The Journal of cell biology · 2023Article
- Codon modification of Tuba1a alters mRNA levels and causes a severe neurodevelopmental phenotype in mice.Scientific reports · 2023Article
- Novel loss of function mutation inFrontiers in cellular neuroscience · 2023Article
- Article
- Microtubules in Microorganisms: How Tubulin Isotypes Contribute to Diverse Cytoskeletal Functions.Frontiers in cell and developmental biology · 2022Review
- The molecular biology of tubulinopathies: Understanding the impact of variants on tubulin structure and microtubule regulation.Frontiers in cellular neuroscience · 2022Review
- Tubulin isotypes optimize distinct spindle positioning mechanisms during yeast mitosis.The Journal of cell biology · 2021Article
- Modeling a disease-correlated tubulin mutation in budding yeast reveals insight into MAP-mediated dynein function.Molecular biology of the cell · 2021Article
- Loss-of-Function Plays a Major Role in Early Neurogenesis of Tubulin α-1 A (TUBA1A) Mutation-Related Brain Malformations.Molecular neurobiology · 2021Article
- Novel function ofProceedings of the National Academy of Sciences of the United States of America · 2021Article
- Bridging the Gap: The Importance of TUBA1A α-Tubulin in Forming Midline Commissures.Frontiers in cell and developmental biology · 2021Article
- Kinetically Stabilizing Mutations in Beta Tubulins Create Isotype-Specific Brain Malformations.Frontiers in cell and developmental biology · 2021Article
- Cranial irradiation impairs juvenile social memory and modulates hippocampal physiology.Brain research · 2020Article
- A proteomic survey of microtubule-associated proteins in a R402H TUBA1A mutant mouse.PLoS genetics · 2020Article
- Differential requirements of tubulin genes in mammalian forebrain development.PLoS genetics · 2019Article
Corrections and comments
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Authors and funding
7 authors at 3 institutions in 1 country.
Funding
Abstract
Mutations in the microtubule cytoskeleton are linked to cognitive and locomotor defects during development, and neurodegeneration in adults. How these mutations impact microtubules, and how this alters function at the level of neurons is an important area of investigation. Using a forward genetic screen in mice, we identified a missense mutation in Tuba1a α-tubulin that disrupts cortical and motor neuron development. Homozygous mutant mice exhibit cortical dysgenesis reminiscent of human tubulinopathies. Motor neurons fail to innervate target muscles in the limbs and show synapse defects at proximal targets. To directly examine effects on tubulin function, we created analogous mutations in the α-tubulin isotypes in budding yeast. These mutations sensitize yeast cells to microtubule stresses including depolymerizing drugs and low temperatures. Furthermore, we find that mutant α-tubulin is depleted from the cell lysate and from microtubules, thereby altering ratios of α-tubulin isotypes. Tubulin-binding cofactors suppress the effects of the mutation, indicating an important role for these cofactors in regulating the quality of the α-tubulin pool. Together, our results give new insights into the functions of Tuba1a, mechanisms for regulating tubulin proteostasis, and how compromising these may lead to neural defects.
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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.