<scp>FIBCD1</scp>
            is an endocytic
            <scp>GAG</scp>
            receptor associated with a novel neurodevelopmental disorder

Fell, Christopher W.; Hagelkrüys, Astrid; Cicvaric, Ana; Horrer, Marion; Liu, Lucy; Li, Joshua Shing Shun; Stadlmann, Johannes; Polyansky, Anton A.; Mereiter, Stefan; Tejada, Miguel; Kokotović, Tomislav; Achuta, Venkat Swaroop; Scaramuzza, Angelica; Twyman, Kimberly A.; Morrow, Michelle M.; Juusola, Jane; Yan, Huifang; Wang, Jingmin; Burmeister, Margit; Choudhury, Biswa; Andersen, Thomas Levin; Wirnsberger, Gerald; Holmskov, Uffe; Perrimon, Norbert; Žagrović, Bojan; Monje, Francisco J.; Møeller, Jesper Bonnet; Penninger, Josef; Nagy, Vanja

doi:10.15252/emmm.202215829

Cited by 12 publications

(11 citation statements)

References 83 publications

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“…The 2 most highly upregulated genes in the beneficial group are CG15263 (no human ortholog) and CG6788 ( FIBCD1 ) (Log 2 (fold change) = 8.27 and 4.04, respectively) ( Hu et al 2011 ). While the function of CG6788 is unknown, loss of function mutations in FIBCD1 is associated with neurodevelopmental disorders in humans ( Fell et al 2022 ). CG6788 may also play a role in neurodevelopment in Drosophila.…”

Section: Resultsmentioning

confidence: 99%

A Drosophila screen identifies a role for histone methylation in ER stress preconditioning

Owings,

Chow

2023

G3: Genes, Genomes, Genetics

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Stress preconditioning occurs when transient, sublethal stress events impact an organism's ability to counter future stresses. Although preconditioning effects are often noted in the literature, very little is known about the underlying mechanisms. To model preconditioning, we exposed a panel of genetically diverse Drosophila melanogaster to a sublethal heat shock and measured how well the flies survived subsequent exposure to endoplasmic reticulum (ER) stress. The impact of preconditioning varied with genetic background, ranging from dying half as fast to 4 and a half times faster with preconditioning compared to no preconditioning. Subsequent association and transcriptional analyses revealed that histone methylation, and transcriptional regulation are both candidate preconditioning modifier pathways. Strikingly, almost all subunits (7/8) in the Set1/COMPASS complex were identified as candidate modifiers of preconditioning. Functional analysis of Set1 knockdown flies demonstrated that loss of Set1 led to the transcriptional dysregulation of canonical ER stress genes during preconditioning. Based on these analyses, we propose a preconditioning model in which Set1 helps to establish an interim transcriptional “memory” of previous stress events, resulting in a preconditioned response to subsequent stress.

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Section: Resultsmentioning

confidence: 99%

A Drosophila screen identifies a role for histone methylation in ER stress preconditioning

Owings,

Chow

2023

G3: Genes, Genomes, Genetics

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“…The Ncan gene encodes for a CSPG neurocan, the main component of perineuronal nets stabilizing synapses. A very recent study has identified the fibrinogen C domain containing 1 (FIBCD1) as an endocytic receptor for CSPGs predominantly expressed in the CA1 area of the hippocampus within the brain [ 39 ]. Thus, the PSEN1 ΔE9 mutation in transplanted human glia dysregulated the expression of CSPG signaling molecules in the mouse hippocampus.…”

Section: Resultsmentioning

confidence: 99%

“…A genetic variant in the human NCAN gene increasing the level of neurocan expression was recently found to be associated with worsened hippocampal memory function in healthy humans [ 66 ]. Moreover, FIBCD1 has recently been demonstrated to be involved in hippocampal-dependent learning in mice [ 39 ]. Altogether, these gene expression changes are in line with better spatial memory function in PSEN1 ΔE9-mutant glia-transplanted mice observed in our study.…”

Section: Discussionmentioning

confidence: 99%

Human PSEN1 Mutant Glia Improve Spatial Learning and Memory in Aged Mice

Jäntti

Oksanen

Kettunen

et al. 2022

Cells

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The PSEN1 ΔE9 mutation causes a familial form of Alzheimer’s disease (AD) by shifting the processing of amyloid precursor protein (APP) towards the generation of highly amyloidogenic Aβ42 peptide. We have previously shown that the PSEN1 ΔE9 mutation in human-induced pluripotent stem cell (iPSC)-derived astrocytes increases Aβ42 production and impairs cellular responses. Here, we injected PSEN1 ΔE9 mutant astrosphere-derived glial progenitors into newborn mice and investigated mouse behavior at the ages of 8, 12, and 16 months. While we did not find significant behavioral changes in younger mice, spatial learning and memory were paradoxically improved in 16-month-old PSEN1 ΔE9 glia-transplanted male mice as compared to age-matched isogenic control-transplanted animals. Memory improvement was associated with lower levels of soluble, but not insoluble, human Aβ42 in the mouse brain. We also found a decreased engraftment of PSEN1 ΔE9 mutant cells in the cingulate cortex and significant transcriptional changes in both human and mouse genes in the hippocampus, including the extracellular matrix-related genes. Overall, the presence of PSEN1 ΔE9 mutant glia exerted a more beneficial effect on aged mouse brain than the isogenic control human cells likely as a combination of several factors.

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“…Fibcd1 encodes a receptor for endocytosed glycosaminoglycans in the brain, and plays a role in nervous system structure, function and plasticity. Knocking down Fibcd1 leads to idiopathic complex neurodevelopmental disorder (NDD) (Fell et al 2022). Eng2b is involved in the localization of diencephalon-mesencephalon boundary during brain development (Rampon et al 2015).…”

Section: Discussionmentioning

confidence: 99%

Multi-omics analysis unravels the underlying mechanisms of the escape response of the common strain black carp

Xu,

Dong,

Sewo

et al. 2024

Preprint

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Reducing the escape response of fish during the aquaculture has important economic value and ecological safety significance. This study takes the common strain black carp (Cyprinus carpio var. baisenensis), which is known for its low-escape response, as the main research object. Through a simulated flood experiment, the common strain black carps were divided into the low-escape (BL) group and the high-escape (BH) group. Multi-omics techniques:transcriptome sequencing, LS-MS/MS detection, and 16s sequencing were used to analyze the differences in brain gene transcription levels, liver metabolites, and intestinal microbiota composition between the two groups. In addition, in order to reduce false positives generated by transcriptome experiments, Jian carps (Cyprinus carpio var. Jian) were also grouped into the low-escape (JL) group and the high-escape (JH) group and subjected to transcriptome analysis. According to the differentially expressed genes (DEGs) analysis, 18 DEGs were obtained from both the common strain black carp and Jian carp. Except for the MAP6 gene, which was significantly highly expressed, the remaining 17 genes showed significantly low expression in the BL group, which were almost related to signal transduction, and brain tissue and neuronal development. The results of KEGG signaling pathway annotation, KOG functional annotation, and topGO enrichment analysis showed that there were significant differences in signal transduction between the BL and BH groups, especially in the gama aminobutyric acid (GABA) signaling pathway. The detection of liver metabolites showed that there were differences in tryptophan metabolism between the BL and BH groups of the common strain black carp, with higher tryptophan content in the liver of the BL group fish. This study suggests that the brain activity and development of low-escape fish may be lower than that of high-escape fish. Meanwhile, differences in the brain's GABA signaling pathway and the liver's tryptophan metabolismmay also affect fish's escape response. This study accumulates experimental foundation of fish escape response, and provides a new insight into breeding low-escape fish and developing novel strategies to reduce escape responses during aquaculture.

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FIBCD1 is an endocytic GAG receptor associated with a novel neurodevelopmental disorder

Cited by 12 publications

References 83 publications

A Drosophila screen identifies a role for histone methylation in ER stress preconditioning

A Drosophila screen identifies a role for histone methylation in ER stress preconditioning

Human PSEN1 Mutant Glia Improve Spatial Learning and Memory in Aged Mice

Multi-omics analysis unravels the underlying mechanisms of the escape response of the common strain black carp

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