ArticleCell death and differentiation2022
Disruption of GMNC-MCIDAS multiciliogenesis program is critical in choroid plexus carcinoma development.
Article in Cell death and differentiation, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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Who cites it
14 citing papers in PubMed, 15 citations in OpenAlex.
- Overactivation of SHH signaling induces a cascade of dedifferentiation and oncogenesis in a new mouse model for choroid plexus carcinoma.Acta neuropathologica communications · 2026Article
- Choroid plexus carcinoma: state of the field and emerging directions.Oncogenesis · 2026Review
- Unique clinical features and transcriptomic profiling of carcinogenesis in patients with familial lung cancer in Yunnan Province, Wumeng mountains, China.Respiratory research · 2026Article
- Investigation of a global mouse methylome atlas reveals subtype-specific copy number alterations in pediatric cancer models.Nature genetics · 2026Article
- SOX2 commands LIM homeobox transcription factors in choroid plexus development and tumorigenesis.Neuro-oncology · 2025Article
- Sequential emergence and contraction of epithelial subtypes in the prenatal human choroid plexus revealed by a stem cell model.Nature communications · 2025Article
- Choroid Plexus Pathophysiology.Annual review of pathology · 2025Review
- Single-nucleus RNA-seq dissection of choroid plexus tumor cell heterogeneity.The EMBO journal · 2024Article
- Influence of Hypoxia on the Airway Epithelium.Physiological research · 2024Review
- Breakthroughs in choroid plexus and CSF biology from the first European Choroid plexus Scientific Forum (ECSF).Fluids and barriers of the CNS · 2024Review
- RBL2 represses the transcriptional activity of Multicilin to inhibit multiciliogenesis.Cell death & disease · 2024Article
- Multi-omics analyses of choroid plexus carcinoma cell lines reveal potential targetable pathways and alterations.Journal of neuro-oncology · 2024Article
- Cancer Stem Cells in Tumours of the Central Nervous System in Children: A Comprehensive Review.Cancers · 2023Review
- Preclinical validation of a novel therapeutic strategy for choroid plexus carcinoma.Journal of controlled release : official journal of the Controlled Release Society · 2023Article
Corrections and comments
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Authors and funding
25 authors at 13 institutions in 6 countries.
Funding
Abstract
Multiciliated cells (MCCs) in the brain reside in the ependyma and the choroid plexus (CP) epithelia. The CP secretes cerebrospinal fluid that circulates within the ventricular system, driven by ependymal cilia movement. Tumors of the CP are rare primary brain neoplasms mostly found in children. CP tumors exist in three forms: CP papilloma (CPP), atypical CPP, and CP carcinoma (CPC). Though CPP and atypical CPP are generally benign and can be resolved by surgery, CPC is a particularly aggressive and little understood cancer with a poor survival rate and a tendency for recurrence and metastasis. In contrast to MCCs in the CP epithelia, CPCs in humans are characterized by solitary cilia, frequent TP53 mutations, and disturbances to multiciliogenesis program directed by the GMNC-MCIDAS transcriptional network. GMNC and MCIDAS are early transcriptional regulators of MCC fate differentiation in diverse tissues. Consistently, components of the GMNC-MCIDAS transcriptional program are expressed during CP development and required for multiciliation in the CP, while CPC driven by deletion of Trp53 and Rb1 in mice exhibits multiciliation defects consequent to deficiencies in the GMNC-MCIDAS program. Previous studies revealed that abnormal NOTCH pathway activation leads to CPP. Here we show that combined defects in NOTCH and Sonic Hedgehog signaling in mice generates tumors that are similar to CPC in humans. NOTCH-driven CP tumors are monociliated, and disruption of the NOTCH complex restores multiciliation and decreases tumor growth. NOTCH suppresses multiciliation in tumor cells by inhibiting the expression of GMNC and MCIDAS, while Gmnc-Mcidas overexpression rescues multiciliation defects and suppresses tumor cell proliferation. Taken together, these findings indicate that reactivation of the GMNC-MCIDAS multiciliogenesis program is critical for inhibiting tumorigenesis in the CP, and it may have therapeutic implications for the treatment of CPC.
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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.