Structural basis of BBSome-mediated ciliary exit
BBSome介导的纤毛退出的结构基础
基本信息
- 批准号:10624912
- 负责人:
- 金额:$ 70.87万
- 依托单位:
- 依托单位国家:美国
- 项目类别:
- 财政年份:2020
- 资助国家:美国
- 起止时间:2020-06-01 至 2024-05-31
- 项目状态:已结题
- 来源:
- 关键词:AddressAffectBardet-Biedl SyndromeBindingBiochemicalBiological AssayBlindnessCattleCellsCiliaClathrin AdaptorsComplexComputer softwareCryoelectron MicroscopyDataDefectDiseaseErinaceidaeEukaryotic CellExcisionFunctional disorderFutureG-Protein-Coupled ReceptorsGTP BindingGoalsGuanosine TriphosphateGuanosine Triphosphate PhosphohydrolasesHairHealthHereditary DiseaseHumanIceImageKnowledgeLightMapsMediatingMembraneMembrane ProteinsModelingMolecularMolecular ConformationMovementMutationObesityPalliative CarePathogenicityPathway interactionsPatientsPeptidesPhotoreceptorsPolydactylyProteinsReceptor SignalingRegulationResolutionRetinaRetinal DegenerationRetinal DystrophySSTR3 geneSideSignal PathwaySignal TransductionSignaling MoleculeSmell PerceptionSolidStructural ModelsStructureTFAP2A geneTestingTherapeutic InterventionTrainingTranscription Factor AP-1VariantVertebral columnVisionWorkciliopathyconformergene replacement therapygene therapyinsightkidney malformationmorphogensnovel therapeutic interventionparticlepre-clinicalpreclinical trialprotein complexrecruitsmoothened signaling pathwaytherapeutically effectivetrafficking
项目摘要
PROJECT SUMMARY
Primary cilia organize signaling pathways such as vision, olfaction and Hedgehog signaling. Proper functioning
of these pathways is critically dependent on the movements of molecules into, inside and out of cilia, yet our
understanding of the basic mechanisms governing trafficking through cilia remains fragmentary. Past work
from the lab identified and characterized the BBSome, a protein complex that ferries signaling receptors out of
cilia and clears photoreceptor outer segments of unwanted proteins. The relevance of the BBSome to human
health and disease is evidence by the fact that BBSome dysfunction causes Bardet-Biedl Syndrome (BBS), a
hereditary disease characterized by obesity, retinal degeneration, polydactyly and kidney malformations.
The major goal of this proposal is to determine the structure and function of the molecular cogs and levers
within the BBSome that enable selective removal of proteins from cilia. The proposed studies will cast new light
on ciliary trafficking and lay the basis of future therapeutic interventions.
项目摘要
初级纤毛组织信号通路,如视觉,嗅觉和刺猬信号。正常运作
这些途径的关键是依赖于运动的分子进入,内部和外部的纤毛,但我们的
对通过纤毛进行贩运的基本机制的了解仍然是零碎的。过去的工作
来自实验室的研究人员鉴定并表征了BBSome,这是一种蛋白质复合物,
纤毛和清除感光细胞外节的不需要的蛋白质。BBSome与人类的相关性
BBSome功能障碍导致Bardet-Biedl综合征(BBS),一种
以肥胖、视网膜退化、多指和肾畸形为特征的遗传性疾病。
这项建议的主要目标是确定分子齿轮和杠杆的结构和功能
在BBSome中,能够从纤毛中选择性地去除蛋白质。拟议中的研究将为我们提供新的视角,
纤毛贩运,并奠定了未来的治疗干预措施的基础。
项目成果
期刊论文数量(0)
专著数量(0)
科研奖励数量(0)
会议论文数量(0)
专利数量(0)
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Maxence V Nachury其他文献
Xenopus Cdc14α/β are localized to the nucleolus and centrosome and are required for embryonic cell division
- DOI:
10.1186/1471-2121-5-27 - 发表时间:
2004-07-13 - 期刊:
- 影响因子:2.700
- 作者:
Brett K Kaiser;Maxence V Nachury;Bryan E Gardner;Peter K Jackson - 通讯作者:
Peter K Jackson
Maxence V Nachury的其他文献
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{{ truncateString('Maxence V Nachury', 18)}}的其他基金
Structural basis of BBSome-mediated ciliary exit
BBSome介导的纤毛退出的结构基础
- 批准号:
10409687 - 财政年份:2020
- 资助金额:
$ 70.87万 - 项目类别:
Structural basis of BBSome-mediated ciliary exit
BBSome介导的纤毛退出的结构基础
- 批准号:
10161785 - 财政年份:2020
- 资助金额:
$ 70.87万 - 项目类别:
Proteomics of Primary Cilia through Proximity Labeling
通过邻近标记研究初级纤毛的蛋白质组学
- 批准号:
9590675 - 财政年份:2015
- 资助金额:
$ 70.87万 - 项目类别:
Quality control of the primary cilium proteome
初级纤毛蛋白质组的质量控制
- 批准号:
10551228 - 财政年份:2010
- 资助金额:
$ 70.87万 - 项目类别:
Quality control of the primary cilium proteome
初级纤毛蛋白质组的质量控制
- 批准号:
10546935 - 财政年份:2010
- 资助金额:
$ 70.87万 - 项目类别:
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