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White matter abnormalities in the Hdc knockout mouse, a model of tic and OCD pathophysiology

ABSTRACT: White matter abnormalities in the Hdc knockout mouse, a model of tic and OCD pathophysiology

PROVIDER: PRJNA676035 | ENA |

REPOSITORIES: ENA

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			Action	DRS
	SRR13024003.fastq.gz	Fastqsanger.gz
	SRR13024004.fastq.gz	Fastqsanger.gz
	SRR13024005.fastq.gz	Fastqsanger.gz
	SRR13024006.fastq.gz	Fastqsanger.gz
	SRR13024007.fastq.gz	Fastqsanger.gz

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White matter abnormalities in the Hdc knockout mouse, a model of tic and OCD pathophysiology

Project description:An inactivating mutation in the histidine decarboxylase gene (Hdc) has been identified as a rare but high-penetrance genetic cause of Tourette syndrome (TS). TS is a neurodevelopmental syndrome characterized by recurrent motor and vocal tics; it is accompanied by structural and functional abnormalities in the cortico-basal ganglia circuitry. Hdc, which is expressed both in the posterior hypothalamus and peripherally, encodes an enzyme required for the biosynthesis of histamine. Hdc knockout mice (Hdc-KO) recapitulate this mutation and exhibit behavioral and neurochemical abnormalities that parallel those seen in patients with TS. Exploratory RNA-Seq analysis revealed, unexpectedly, that genes associated with oligodendrocytes and with myelin production are upregulated in the dorsal striatum of these mice. This was confirmed by qPCR and immunoblotting. These results suggest an abnormality in myelination in the striatum. To test this in an intact brain, we performed whole-brain ex vivo diffusion tensor imaging (DTI), which revealed reduced fractional anisotropy (FA) in the mid-dorsal striatum. While the DTI literature in individuals with TS is sparse, these results are consistent with findings of disrupted descending cortical projections in patients with tics. The Hdc-KO model may represent a powerful system in which to examine the developmental mechanisms underlying this abnormality.

2023-01-04 | GSE161252 | GEO

White matter abnormalities in the <i>Hdc</i> knockout mouse, a model of tic and OCD pathophysiology.

Project description:IntroductionAn inactivating mutation in the histidine decarboxylase gene (Hdc) has been identified as a rare but high-penetrance genetic cause of Tourette syndrome (TS). TS is a neurodevelopmental syndrome characterized by recurrent motor and vocal tics; it is accompanied by structural and functional abnormalities in the cortico-basal ganglia circuitry. Hdc, which is expressed both in the posterior hypothalamus and peripherally, encodes an enzyme required for the biosynthesis of histamine. Hdc knockout mice (Hdc-KO) functionally recapitulate this mutation and exhibit behavioral and neurochemical abnormalities that parallel those seen in patients with TS.Materials and methodsWe performed exploratory RNA-seq to identify pathological alterations in several brain regions in Hdc-KO mice. Findings were corroborated with RNA and protein quantification, immunohistochemistry, and ex vivo brain imaging using MRI.ResultsExploratory RNA-Seq analysis revealed, unexpectedly, that genes associated with oligodendrocytes and with myelin production are upregulated in the dorsal striatum of these mice. This was confirmed by qPCR, immunostaining, and immunoblotting. These results suggest an abnormality in myelination in the striatum. To test this in an intact mouse brain, we performed whole-brain ex vivo diffusion tensor imaging (DTI), which revealed reduced fractional anisotropy (FA) in the dorsal striatum.DiscussionWhile the DTI literature in individuals with TS is sparse, these results are consistent with findings of disrupted descending cortical projections in patients with tics. The Hdc-KO model may represent a powerful system in which to examine the developmental mechanisms underlying this abnormality.

| S-EPMC9731796 | biostudies-literature

Differential expression of Hdc-GFP+/hiCD11b+Gr1+ vs Hdc-GFP-/loCD11b+Gr1+ myeloid cells from mouse bone marrow

Project description:Bone marrow Hdc-GFP+/hi and Hdc-GFP-/loCD11b+Gr1+ cells were isolated from bones from histidine decarboxylase (Hdc) green fluorescent protein (Hdc-GFP) mice Hdc-GFP+/hiCD11b+Gr1+ cells and Hdc-GFP-/loCD11b+Gr1+ cells were sorted by combinations of GFP and myeloid cell surface markers CD11b and Gr1 and their differential mRNA expression compared with Affymetrix microarrays.

2017-08-22 | GSE79728 | GEO

Differential expression of Hdc-GFP+/hiCD11b+Gr1lo vs Hdc-GFP+/hiCD11b+Gr1hi myeloid cells from mouse bone marrow

Project description:Bone marrow Hdc-GFP+/hiCD11b+Gr1lo vs Hdc-GFP+/hiCD11b+Gr1hi myeloid cells were isolated from bones from histidine decarboxylase (Hdc) green fluorescent protein (Hdc-GFP) mice. Hdc-GFP+/hiCD11b+Gr1hi cells and Hdc-GFP+/hiCD11b+Gr1/lo cells were sorted by combinations of GFP and myeloid cell surface markers CD11b and Gr1 and their differential mRNA expression compared with Affymetrix microarrays.

2019-04-01 | GSE80143 | GEO

Gene expression data from 1 year old Hdc+/+ and Hdc-/- stomach.

Project description:Histamine defeciency result to hypertrophic gastropathy in the stomach. We use gene array to distover the major cause of stomach lesion in Hdc-/-. Tesult shows that chronic inflammation and dysregulation of macrophage functions are the major trigger for hypertrophic gastropathy in Hdc--/- stomach.

2022-09-14 | GSE213293 | GEO

The Hdc GC box is critical for Hdc gene transcription and histamine-mediated anaphylaxis [ATAC-Seq]

Project description:Histamine is a critical mediator of anaphylaxis, a neurotransmitter, and a regulator of gastric acid secretion. Histidine decarboxylase is a rate-limiting enzyme for histamine synthesis. However, in vivo regulation of Hdc, the gene that encodes histidine decarboxylase is poorly understood. We sought to investigate how enhancers regulate Hdc gene transcription and histamine synthesis in resting conditions and in a mouse model of anaphylaxis. H3K27 acetylation histone modification and chromatin accessibility were used to identify candidate enhancers; The enhancer activity of candidate enhancers was measured in a reporter gene assay; and the function enhancers were validated using CRISPR deletion. Deletion of the GC box, which binds to zinc finger transcription factors, in the proximal Hdc enhancer, reduced Hdc gene transcription and histamine synthesis in the mouse and human mast cell lines. Mast cells, basophils, brain cells, and stomach cells from GC box-deficient mice transcribed the Hdc gene much less than similar cells from wild-type mice and Hdc GC box-deficient mice failed to develop anaphylaxis. Our results demonstrate that the HDC GC box within the proximal enhancer in the mouse and human HDC gene is essential for Hdc gene transcription, histamine synthesis, and histamine-mediated anaphylaxis in vitro and in vivo.

2023-02-23 | GSE223387 | GEO

The Hdc GC box is critical for Hdc gene transcription and histamine-mediated anaphylaxis [CUT&Tag]

2023-02-23 | GSE223386 | GEO

Differential expression of Hdc-GFP+/hi HSC VS Hdc-GFP-/lo hematopoetic stem cells (HSCs) from bone marrow.

Project description:Bone marrow Hdc-GFPhi and Hdc-GFPlo HSPC (SLAM-LSK, Lin-c-kit+Sca-1+CD150+CD48-) HSCs were isolated from mouse femur and tibia from histidine decarboxylase (Hdc) green fluorescent protein (Hdc-GFP) mice. Hdc-GFPhi HSC and Hdc-GFPlo HSC cells were sorted by combinations of GFP and the cell surface markers of HSC. total RNA was isolated from sorted 2,500 HSPCs using ARCTURUS PicoPure RNA isolation kit (Life Technologies). cDNA was amplified and libraries were constructed by using SMARTer Ultra Low Input RNA kit (Clontech Laboratories) and Nextera XT DNA Library Preparation kit (Illumina) according to manufacturer’s instructions respectively. Sequencing was performed on the Illumina HiSeq2000 platform.

2019-11-20 | GSE90787 | GEO

Differential expression of Hdc-GFPhi HSPC VS Hdc-GFPlo HSPC hematopoetic stem and progenitor cells from mouse bone marrow

Project description:Differential expression of Hdc-GFPhi HSPC VS Hdc-GFPlo HSPC hematopoetic stem and progenitor cells from mouse bone marrow

2019-04-01 | GSE80092 | GEO

Gene expression data from 1 year old Hdc+/+ and Hdc-/- stomach.

Project description:Gene expression data from 1 year old Hdc+/+ and Hdc-/- stomach.

| PRJNA880342 | ENA