PUBLICATION

Functional and genetic analysis of choroid plexus development in zebrafish

Authors
Henson, H.E., Parupalli, C., Ju, B., Taylor, M.R.
ID
ZDB-PUB-141127-2
Date
2014
Source
Frontiers in neuroscience   8: 364 (Journal)
Registered Authors
Ju, Bensheng, Taylor, Michael R.
Keywords
Claudin 5, choroid plexus, fluorescein, forward genetic screen, genetic mapping, transporters, zebrafish
MeSH Terms
none
PubMed
25426018 Full text @ Front. Neurosci.
Abstract
The choroid plexus, an epithelial-based structure localized in the brain ventricle, is the major component of the blood-cerebrospinal fluid barrier. The choroid plexus produces the cerebrospinal fluid and regulates the components of the cerebrospinal fluid. Abnormal choroid plexus function is associated with neurodegenerative diseases, tumor formation in the choroid plexus epithelium, and hydrocephaly. In this study, we used zebrafish (Danio rerio) as a model system to understand the genetic components of choroid plexus development. We generated an enhancer trap line, Et(cp:EGFP) (sj2), that expresses enhanced green fluorescent protein (EGFP) in the choroid plexus epithelium. Using immunohistochemistry and fluorescent tracers, we demonstrated that the zebrafish choroid plexus possesses brain barrier properties such as tight junctions and transporter activity. Thus, we have established zebrafish as a functionally relevant model to study choroid plexus development. Using an unbiased approach, we performed a forward genetic dissection of the choroid plexus to identify genes essential for its formation and function. Using Et(cp:EGFP) (sj2), we isolated 10 recessive mutant lines with choroid plexus abnormalities, which were grouped into five classes based on GFP intensity, epithelial localization, and overall choroid plexus morphology. We also mapped the mutation for two mutant lines to chromosomes 4 and 21, respectively. The mutants generated in this study can be used to elucidate specific genes and signaling pathways essential for choroid plexus development, function, and/or maintenance and will provide important insights into how these genetic mutations contribute to disease.
Genes / Markers
Figures
No images available
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
a9
    Complex
    sj2TgTransgenic Insertion
      w2
        Point Mutation
        zf627
          Unknown
          zf628
            Unknown
            zf629
              Unknown
              zf630
                Unknown
                zf631
                  Unknown
                  zf632
                    Unknown
                    zf633
                      Point Mutation
                      1 - 10 of 13
                      Show
                      Human Disease / Model
                      No data available
                      Sequence Targeting Reagents
                      No data available
                      Fish
                      Antibodies
                      Orthology
                      Engineered Foreign Genes
                      Marker Marker Type Name
                      EGFPEFGEGFP
                      1 - 1 of 1
                      Show
                      Mapping