ArticleScientific reports2018
Strain-specific differences in brain gene expression in a hydrocephalic mouse model with motile cilia dysfunction.
Article in Scientific reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 26 citations in OpenAlex.
- Exploring Ciliary Mechanisms in the Causation of Hydrocephalus in Humans-Similarities and Differences from Animal Models.Journal of molecular neuroscience : MN · 2025Review
- The power of mouse models in the diagnostic odyssey of patients with rare congenital anomalies.Mammalian genome : official journal of the International Mammalian Genome Society · 2025Review
- Screening of SNP Loci Related to Leg Length Trait in Leizhou Goats Based on Whole-Genome Resequencing.International journal of molecular sciences · 2024Article
- Inbred Mouse Models inJournal of fungi (Basel, Switzerland) · 2024Review
- Article
- PCD Genes-From Patients to Model Organisms and Back to Humans.International journal of molecular sciences · 2022Review
- Ependymal Cilia: Physiology and Role in Hydrocephalus.Frontiers in molecular neuroscience · 2022Review
- A HomozygousGenes · 2021Article
- Microglia activated by microbial neuraminidase contributes to ependymal cell death.Fluids and barriers of the CNS · 2021Article
- Motile cilia genetics and cell biology: big results from little mice.Cellular and molecular life sciences : CMLS · 2021Review
- Variants in RABL2A causing male infertility and ciliopathy.Human molecular genetics · 2020Article
- Analysis of the sperm flagellar axoneme using gene-modified mice.Experimental animals · 2020Review
- An exome-first approach to aid in the diagnosis of primary ciliary dyskinesia.Human genetics · 2020Article
- CAMSAP3 facilitates basal body polarity and the formation of the central pair of microtubules in motile cilia.Proceedings of the National Academy of Sciences of the United States of America · 2020Article
- Identification and mapping of central pair proteins by proteomic analysis.Biophysics and physicobiology · 2020Article
- Ciliary Dysfunction: The Hairy Explanation of Normal Pressure Hydrocephalus?Movement disorders clinical practice · 2020Article
- Functional loss ofDisease models & mechanisms · 2019Article
- The year in review: progress in brain barriers and brain fluid research in 2018.Fluids and barriers of the CNS · 2019Article
Corrections and comments
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
Abstract
Congenital hydrocephalus results from cerebrospinal fluid accumulation in the ventricles of the brain and causes severe neurological damage, but the underlying causes are not well understood. It is associated with several syndromes, including primary ciliary dyskinesia (PCD), which is caused by dysfunction of motile cilia. We previously demonstrated that mouse models of PCD lacking ciliary proteins CFAP221, CFAP54 and SPEF2 all have hydrocephalus with a strain-dependent severity. While morphological defects are more severe on the C57BL/6J (B6) background than 129S6/SvEvTac (129), cerebrospinal fluid flow is perturbed on both backgrounds, suggesting that abnormal cilia-driven flow is not the only factor underlying the hydrocephalus phenotype. Here, we performed a microarray analysis on brains from wild type and nm1054 mice lacking CFAP221 on the B6 and 129 backgrounds. Expression differences were observed for a number of genes that cluster into distinct groups based on expression pattern and biological function, many of them implicated in cellular and biochemical processes essential for proper brain development. These include genes known to be functionally relevant to congenital hydrocephalus, as well as formation and function of both motile and sensory cilia. Identification of these genes provides important clues to mechanisms underlying congenital hydrocephalus severity.
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Registered trials
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