FAM19A5l Affects Mustard Oil-Induced Peripheral Nociception in Zebrafish

Jeong, Inyoung; Yun, Seongsik; Shahapal, Anu; Cho, Eun Bee; Hwang, Sun Wook; Seong, Jae Young; Park, Hae Chul

doi:10.1007/s12035-021-02449-z

Cited by 11 publications

(16 citation statements)

References 58 publications

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“…To validate efficiency of the sgRNA, fabp7b sgRNA and Cas9 mRNA was microinjected into one-cell wildtype embryos and genomic DNAs were extracted from circa 20 injected embryos at 24 hpf. Then, T7 endonuclease I assay was performed (Jao et al , 2013; Jeong et al , 2021). Next, homology arms of the target vector were designed by the GTagHD (http://www.genesculpt.org/gtaghd/) and the pGTag-2A-Gal4vp16-B-actin plasmid (Addgene) was used as a donor vector (Welker et al ., 2021; Wierson et al ., 2020).…”

Section: Star Methodsmentioning

confidence: 99%

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The evolutionary conserved choroid plexus sustains the homeostasis of brain ventricles in zebrafish

Jeong,

Andreassen,

Hoang

et al. 2023

Preprint

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SummaryThe choroid plexus produces cerebrospinal fluid (CSF) by transport of electrolytes and water from the vasculature to the brain ventricles. The choroid plexus plays additional roles in brain development and homeostasis by secreting neurotrophic molecules, and by serving as a CSF-blood barrier and immune interface. Prior studies have identified transporters on the epithelial cells that transport water and ions into the ventricles and tight junctions involved in the CSF-blood barrier. Yet, how the choroid plexus epithelial cells maintain the brain ventricle system and control brain physiology remain unresolved. To provide novel insights into the physiological roles of the choroid plexus, we use juvenile and adult zebrafish as model systems. Upon histological and transcriptomic analyses, we first identified that the zebrafish choroid plexus is highly conserved with the mammalian choroid plexus and that it expresses all transporters necessary for CSF secretion. Using novel genetic lines, we also identified that the choroid plexus secretes proteins into the CSF. Next, we generated a transgenic line allowing us to ablate specifically the epithelial cells in the choroid plexus. Using the ablation system, we identified a reduction of the ventricular sizes, but no alterations of the CSF-blood barrier. Altogether, our findings identified that the zebrafish choroid plexus is evolutionarily conserved and critical for maintaining the size and homeostasis of the brain ventricles.Graphical abstractHighlightsThe zebrafish choroid plexus has similar anatomical features with the mammalian choroid plexus.The expression of choroid plexus transporters involved in CSF secretion is evolutionarily conserved across vertebrates.Generation of a novel choroid plexus specific driver line shows that the choroid plexus epithelial cells secrete proteins into CSF.Ablation of the choroid plexus decreases the size of the brain ventricles.

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Section: Star Methodsmentioning

confidence: 99%

“…RNA in situ hybridization was performed by alkaline phosphatase-NBT/BCIP (Roche) chromogenic reaction, as described earlier (Jeong et al ., 2021; Thisse & Thisse, 2008). The brains were rehydrated gradually through 75%, 50%, 25% methanol in 0.2 % PBST solution steps for 5 min per each solution.…”

Section: Star Methodsmentioning

confidence: 99%

The evolutionary conserved choroid plexus sustains the homeostasis of brain ventricles in zebrafish

Jeong,

Andreassen,

Hoang

et al. 2023

Preprint

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“…First, the Cas9 target site in exon 1 of foxj1a gene for the single guide RNA (sgRNA) was selected using CHOPCHOP (http://chopchop.cbu.uib.no/). The sgRNA was synthesized by the cloning-free gRNA in vitro synthesis method (Wierson, Welker et al 2020, Jeong, Yun et al 2021, Welker, Wierson et al 2021, and Cas9 mRNA from the pT3TS-nCas9n vector (Addgene plasmid #46757) with mMESSAGE mMACHINE TM T3 transcription kit (Thermo Fisher). To validate the efficiency of the sgRNA, foxj1a sgRNA and Cas9 mRNA was injected into one-cell embryos and genomic DNA was extracted from the injected embryos at 24 hpf.…”

Section: Generation Of Gt(foxj1a:2a-tagrfp) Frzcc 1100 Knock-in Zebra...mentioning

confidence: 99%

Foxj1 controls olfactory ciliogenesis and differentiation program of the olfactory sensory neurons

Rayamajhi

Ege

Ukhanov

et al. 2023

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In vertebrates, olfactory receptors localize on multiple cilia elaborated on dendritic knobs of olfactory sensory neurons (OSNs). Although olfactory cilia dysfunction can cause anosmia, how their differentiation is programmed at the transcriptional level has remained largely unexplored. We discovered in zebrafish and mice that Foxj1, a forkhead domain-containing transcription factor linked with motile cilia biogenesis, is expressed in OSNs and required for olfactory epithelium (OE) formation. In keeping with the immotile nature of olfactory cilia, we observed that ciliary motility genes are repressed in zebrafish, mouse, and human OSNs. Strikingly, we also found that besides ciliogenesis, Foxj1 controls the differentiation of the OSNs themselves by regulating their cell type-specific gene expression, such as that ofolfactory marker protein(omp) involved in odor-evoked signal transduction. In line with this, response to bile acid, an odor detected by OMP-positive OSNs, was significantly diminished infoxj1mutant zebrafish. Taken together, our findings establish how the canonical Foxj1-mediated motile ciliogenic transcriptional program has been repurposed for the biogenesis of immotile olfactory cilia and for development of the OSNs.

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“…FAM19A5, a polypeptide encoded by the TAFA5 gene located on chromosome 22 (22q13.32) in humans and chromosome 15 in mice, is primarily expressed in the brain 1 . This restricted expression and the highly conserved amino acid sequence across vertebrates suggest a crucial role for FAM19A5 in brain development and function 2,3 . A study by Huang et al (2021) showed that loss of FAM19A5 led to reduced spine density, glutamate signaling and neuronal activity.…”

Section: Introductionmentioning

confidence: 99%

Partial FAM19A5 Deficiency in Mice Leads to Disrupted Spine Maturation, Hyperactivity, and an Altered Fear Response

Shahapal,

Park,

Yoo

et al. 2024

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The FAM19A5 polypeptide, encoded by the TAFA5 gene, is evolutionarily conserved among vertebral species. This protein is predominantly expressed in the brain, highlighting its crucial role in the central nervous system (CNS). Here, we investigated the potential roles of FAM19A5 in brain development and behavior using a FAM19A5-LacZ KI mouse model. This model exhibited a partial reduction in the FAM19A5 protein level. FAM19A5-LacZ KI mice displayed no significant alterations in gross brain structure but alterations in dendritic spine distribution, with a bias towards immature forms. These mice also showed lower body weights. Behavioral tests revealed that compared with their wild-type littermates, FAM19A5-LacZ KI mice displayed hyperactivity and a delayed innate fear response. These findings suggest that FAM19A5 plays a role in regulating spine formation and maintenance, thereby contributing to neural connectivity and behavior.

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FAM19A5l Affects Mustard Oil-Induced Peripheral Nociception in Zebrafish

Cited by 11 publications

References 58 publications

The evolutionary conserved choroid plexus sustains the homeostasis of brain ventricles in zebrafish

The evolutionary conserved choroid plexus sustains the homeostasis of brain ventricles in zebrafish

Foxj1 controls olfactory ciliogenesis and differentiation program of the olfactory sensory neurons

Partial FAM19A5 Deficiency in Mice Leads to Disrupted Spine Maturation, Hyperactivity, and an Altered Fear Response

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