IHB OpenIR  > 藻类生物学及应用研究中心  > 期刊论文
Development of the Swimbladder Surfactant System and Biogenesis of Lysosome-Related Organelles Is Regulated by BLOS1 in Zebrafish
Chen, Tianbing1,2; Song, Guili1; Yang, Huihui1,2; Mao, Lin1,2; Cui, Zongbin1; Huang, Kaiyao1
2018-03-01
Source PublicationGENETICS
ISSN0016-6731
Volume208Issue:3Pages:1131-1146
Abstract

Hermansky-Pudlak syndrome (HPS) is a human autosomal recessive disorder that is characterized by oculocutaneous albinism and a deficiency of the platelet storage pool resulting from defective biogenesis of lysosome-related organelles (LROs). To date, 10 HPS genes have been identified, three of which belong to the octamer complex BLOC-1 (biogenesis of lysosome-related organelles complex 1). One subunit of the BLOC-1 complex, BLOS1, also participates in the BLOC-1-related complex (BORC). Due to lethality at the early embryo stage in BLOS1 knockout mice, the function of BLOS1 in the above two complexes and whether it has a novel function are unclear. Here, we generated three zebrafish mutant lines with a BLOC-1 deficiency, in which melanin and silver pigment formation was attenuated as a result of mutation of bloc1s1, bloc1s2, and dtnbp1a, suggesting that they function in the same complex. In addition, mutations of bloc1s1 and bloc1s2 caused an accumulation of clusters of lysosomal vesicles at the posterior part of the tectum, representing a BORC-specific function in zebrafish. Moreover, bloc1s1 is highly expressed in the swimbladder during postembryonic stages and is required for positively regulating the expression of the genes, which is known to govern surfactant production and lung development in mammals. Our study identified BLOS1 as a crucial regulator of the surfactant system. Thus, the zebrafish swimbladder might be an easy system to screen and study genetic modifiers that control surfactant production and homeostasis.

Keywordlysosome-related organelles Hermansky-Pudlak syndrome surfactant system swimbladder
DOI10.1534/genetics.117.300621
Funding OrganizationChinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China
Indexed BySCI
Language英语
Funding ProjectChinese Academy of Sciences[KJZD-EW-L08] ; Hi-Tech Research and Development Program of China (863 Program)[2012AA022402-Y312021603] ; National Natural Science Foundation of China[31371354] ; National Natural Science Foundation of China[31400654]
WOS Research AreaGenetics & Heredity
WOS SubjectGenetics & Heredity
WOS IDWOS:000426219600019
WOS KeywordHERMANSKY-PUDLAK-SYNDROME ; MUTATION SCREEN REVEALS ; PULMONARY SURFACTANT ; PROTEIN TRAFFICKING ; LUNG DEVELOPMENT ; BLOC-1 ; COMPLEX ; MODEL ; SNAPIN ; TRANSPORT
PublisherGENETICS SOCIETY AMERICA
Funding OrganizationChinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Chinese Academy of Sciences ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; Hi-Tech Research and Development Program of China (863 Program) ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China ; National Natural Science Foundation of China
Citation statistics
Cited Times:1[WOS]   [WOS Record]     [Related Records in WOS]
Document Type期刊论文
Identifierhttp://ir.ihb.ac.cn/handle/342005/29923
Collection藻类生物学及应用研究中心_期刊论文
Corresponding AuthorCui, Zongbin; Huang, Kaiyao
Affiliation1.Chinese Acad Sci, Inst Hydrobiol, Wuchang Donghu Nanlu 7, Wuhan 430072, Hubei, Peoples R China
2.Univ Chinese Acad Sci, Beijing 100049, Peoples R China
Recommended Citation
GB/T 7714
Chen, Tianbing,Song, Guili,Yang, Huihui,et al. Development of the Swimbladder Surfactant System and Biogenesis of Lysosome-Related Organelles Is Regulated by BLOS1 in Zebrafish[J]. GENETICS,2018,208(3):1131-1146.
APA Chen, Tianbing,Song, Guili,Yang, Huihui,Mao, Lin,Cui, Zongbin,&Huang, Kaiyao.(2018).Development of the Swimbladder Surfactant System and Biogenesis of Lysosome-Related Organelles Is Regulated by BLOS1 in Zebrafish.GENETICS,208(3),1131-1146.
MLA Chen, Tianbing,et al."Development of the Swimbladder Surfactant System and Biogenesis of Lysosome-Related Organelles Is Regulated by BLOS1 in Zebrafish".GENETICS 208.3(2018):1131-1146.
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