Proceedings of the National Academy of Sciences of the United States of America · 2000
A mutation in Rab27a causes the vesicle transport defects observed in ashen mice.
СтатьяПоддержка правительства СШАПоддержка службы здравоохранения СШАНазвание и аннотация: Перевод пока не подготовлен
Обработка статьи · этапы, ошибки и управление
Загрузка состояния…
Ошибки и ограничения
История · было → стало
Состояние обновляется каждые 5 секунд, пока блок открыт. Процент показывается только там, где известен объём работы. Пауза останавливает продолжение этапа; текущий запрос к источнику может завершиться.
Wilson S M, Yip R, Swing D A, O'Sullivan T N, Zhang Y, Novak E K, Swank R T, Russell L B, Copeland N G, Jenkins N A
Аннотация
The dilute (d), leaden (ln), and ashen (ash) mutations provide a unique model system for studying vesicle transport in mammals. All three mutations produce a lightened coat color because of defects in pigment granule transport. In addition, all three mutations are suppressed by the semidominant dilute-suppressor (dsu), providing genetic evidence that these mutations function in the same or overlapping transport pathways. Previous studies showed that d encodes a major vesicle transport motor, myosin-VA, which is mutated in Griscelli syndrome patients. Here, using positional cloning and bacterial artificial chromosome rescue, we show that ash encodes Rab27a. Rab GTPases represent the largest branch of the p21 Ras superfamily and are recognized as key players in vesicular transport and organelle dynamics in eukaryotic cells. We also show that ash mice have platelet defects resulting in increased bleeding times and a reduction in the number of platelet dense granules. These defects have not been reported for d and ln mice. Collectively, our studies identify Rab27a as a critical gene for organelle-specific protein trafficking in melanocytes and platelets and suggest that Rab27a functions in both MyoVa dependent and independent pathways.
Предметные рубрики MeSH
Albinism, OculocutaneousAnimalsBiological TransportBlood PlateletsChromosome MappingCytoplasmic GranulesDisease Models, AnimalGene LibraryGenetic Complementation TestHair ColorIntermediate Filament ProteinsIntracellular MembranesMelanocytesMiceMice, Inbred C3HMice, Mutant StrainsMuridaeMyosin Heavy ChainsMyosin Type VProtein BindingRNA SplicingSkinSyndromerab GTP-Binding Proteinsrab27 GTP-Binding Proteins