htt huntingtin [Drosophila melanogaster (fruit fly)] - Gene

Summary

Official Symbol: httprovided by FlyBase
Official Full Name: huntingtinprovided by FlyBase
Primary source: FLYBASE:FBgn0027655
Locus tag: Dmel_CG9995
See related: AllianceGenome:FB:FBgn0027655
Gene type: protein coding
RefSeq status: REVIEWED
Organism: Drosophila melanogaster
Lineage: Eukaryota; Metazoa; Ecdysozoa; Arthropoda; Hexapoda; Insecta; Pterygota; Neoptera; Endopterygota; Diptera; Brachycera; Muscomorpha; Ephydroidea; Drosophilidae; Drosophila; Sophophora
Also known as: CG9995; dhtt; dHtt; Dmel\CG9995; DmHtt; HD; Htt; HTT
Summary: Involved in several processes, including establishment of mitotic spindle orientation; heterochromatin boundary formation; and synaptic vesicle transport. Located in cytoplasm. Is expressed in imaginal disc; larval central nervous system; and organism. Used to study Huntington's disease and neurodegenerative disease. Human ortholog(s) of this gene implicated in Huntington's disease and Parkinson's disease. Orthologous to human HTT (huntingtin). [provided by Alliance of Genome Resources, Dec 2024]
Orthologs: all
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Genomic context

Location:: 98E2-98E2; 3-97 cM

Exon count:: 29

Annotation release	Status	Assembly	Chr	Location
Release 6.54	current	Release 6 plus ISO1 MT (GCF_000001215.4)	3R	NT_033777.3 (28696242..28734816)
Release 5.57	previous assembly	Release 5 (GCF_000001215.2)	3R	NT_033777.2 (24521964..24560411)

Chromosome 3R - NT_033777.3 Genomic Context describing neighboring genes

Genomic regions, transcripts, and products

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Go to reference sequence details

Genomic Sequence:

Go to nucleotide: Graphics FASTA GenBank

Bibliography

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GeneRIFs: Gene References Into Functions

What's a GeneRIF?

The ubiquitin thioesterase YOD1 ameliorates mutant Huntingtin induced pathology in Drosophila.
Title: The ubiquitin thioesterase YOD1 ameliorates mutant Huntingtin induced pathology in Drosophila.
Huntingtin Plays a Role in the Physiological Response to Ethanol in Drosophila.
Title: Huntingtin Plays a Role in the Physiological Response to Ethanol in Drosophila.
Htt is a repressor of Abl activity required for APP-induced axonal growth.
Title: Htt is a repressor of Abl activity required for APP-induced axonal growth.
Application of a behavioral platform to screen for clock-regulated Huntington's Disease suppressors, identifies a role for Heat Shock Protein 70/90 Organizing Protein (Hop). Hop knockdown paradoxically reduces mutant Htt aggregation and toxicity. This reveals a clock-regulated molecular and cellular pathway that links clock function to neurodegenerative disease.
Title: Circadian Clocks Function in Concert with Heat Shock Organizing Protein to Modulate Mutant Huntingtin Aggregation and Toxicity.
Loss of CrebA also partially suppresses mHtt effects on behavior and cell loss and restoration of CrebA can suppress Atx2 effects. Our results indicate a prominent role of Atx2 in mediating mHtt pathology, specifically via its regulation of CrebA, defining a novel molecular pathway in Huntington's disease pathogenesis.
Title: Ataxin2 functions via CrebA to mediate Huntingtin toxicity in circadian clock neurons.
Jp modifies the neuronal degeneration in a Drosophila model of Huntington's disease, and it has uncoveedr an unsuspected functional relationship with the Notch pathway.
Title: The Drosophila junctophilin gene is functionally equivalent to its four mammalian counterparts and is a modifier of a Huntingtin poly-Q expansion and the Notch pathway.
Data strongly suggest that mutant Htt expression exclusively in all the CNS and PNS neurons leads to fluctuation of body weight, carbohydrate and protein stores, global lipid levels along with the intracellular lipid deposits through its effect on the integrated process of metabolic homeostasis. These results also support the idea that lipid metabolism remains centrally affected in HD leading to altered body weight.
Title: Altered lipid metabolism in Drosophila model of Huntington's disease.
These results therefore identify native Htt as a regulator of synaptic capture and neuropeptide storage.
Title: Loss of Huntingtin stimulates capture of retrograde dense-core vesicles to increase synaptic neuropeptide stores.
Mutant HTT causes severe mislocalization and aggregation of nucleoporins and defective nucleocytoplasmic transport.
Title: Mutant Huntingtin Disrupts the Nuclear Pore Complex.
Early-onset sleep defects in mutated HTT Drosophila models of Huntington's disease reflect alterations of PKA/CREB signaling.
Title: Early-onset sleep defects in Drosophila models of Huntington's disease reflect alterations of PKA/CREB signaling.

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Interactions

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Products	Interactant	Other Gene	Complex	Source	Pubs	Description

General gene information

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Homology

NCBI Orthologs

Orthologs from OrthoDB

Gene Ontology Provided by FlyBase

Process	Evidence Code	Pubs
involved_in axo-dendritic transport	IMP Inferred from Mutant Phenotype more info	PubMed
involved_in establishment of mitotic spindle orientation	IMP Inferred from Mutant Phenotype more info	PubMed
involved_in heterochromatin boundary formation	IMP Inferred from Mutant Phenotype more info	PubMed
involved_in synaptic vesicle transport	IMP Inferred from Mutant Phenotype more info	PubMed