ArticleNature communications2015
Developmental disruptions underlying brain abnormalities in ciliopathies.
Article in Nature communications, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 89 papers.
What it found
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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.
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Who cites it
89 citing papers in PubMed.
- Folic acid intervention during pregnancy alters DNA methylation, affecting neural target genes through two distinct mechanisms.Clinical epigenetics · 2022Trial
- Cilia SubQ: a modular suite of semi- and fully automated pipelines for analysis of primary cilia and ciliary subdomains.Methods (San Diego, Calif.) · 2026Article
- Primary cilia and neural computation.Journal of biomedical science · 2026Review
- Proximity labeling proteomics maps radial glial ciliary proteins across the developing telencephalon.Cell reports · 2026Article
- The multifaceted role of EXOC6A in ciliogenesis.eLife · 2026Article
- Cilia SubQ, a modular suite of pipelines for automated analysis of primary cilia and ciliary subdomains.bioRxiv : the preprint server for biology · 2026Article
- Bi-allelic variants in FSD1L cause a neurodevelopmental disorder overlapping with L1 syndrome.American journal of human genetics · 2026Article
- Primary cilia as a molecular convergence hub linking neurodevelopment and autism spectrum disorder.Frontiers in cellular neuroscience · 2026Review
- Non-transport functions of motor proteins in corticogenesis.Frontiers in cell and developmental biology · 2026Review
- CEP290 deficiency disrupts ciliary axonemal architecture in human iPSC-derived brain organoids.Journal of cell science · 2025Article
- Tackling ciliary specialization to understand phenotypic variability in human primary ciliopathies.Journal of cell science · 2025Review
- Article
- Identification of New Ciliary Signaling Pathways in the Brain and Insights into Neurological Disorders.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025Article
- GPR161 mechanosensitivity at the primary cilium drives neuronal saltatory migration.Science advances · 2025Article
- Centrosomes and cilia in neurodegeneration: main actors or mere spectators?Open biology · 2025Review
- Methods to Assess Neuronal Primary Cilia Electrochemical Signaling.Journal of cellular physiology · 2025Article
- Biallelic Variants in LRRC45 Impair Ciliogenesis and Cause a Severe Neurological Disorder.Clinical genetics · 2025Article
- Neuronal guidance behaviours: the primary cilium perspective.Frontiers in cell and developmental biology · 2025Review
- Inferring DNA methylation in non-skeletal tissues of ancient specimens.Nature ecology & evolution · 2025Article
- Primary cilia signaling in astrocytes mediates development and regional-specific functional specification.Nature neuroscience · 2024Article
29 more citing papers are in PubMed but not listed here.
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.
Funding
Abstract
Primary cilia are essential conveyors of signals underlying major cell functions. Cerebral cortical progenitors and neurons have a primary cilium. The significance of cilia function for brain development and function is evident in the plethora of developmental brain disorders associated with human ciliopathies. Nevertheless, the role of primary cilia function in corticogenesis remains largely unknown. Here we delineate the functions of primary cilia in the construction of cerebral cortex and their relevance to ciliopathies, using an shRNA library targeting ciliopathy genes known to cause brain disorders, but whose roles in brain development are unclear. We used the library to query how ciliopathy genes affect distinct stages of mouse cortical development, in particular neural progenitor development, neuronal migration, neuronal differentiation and early neuronal connectivity. Our results define the developmental functions of ciliopathy genes and delineate disrupted developmental events that are integrally related to the emergence of brain abnormalities in ciliopathies.
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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.