Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality

Karlsson P, Droce A, Moser JM, Cuhlmann S, Padilla CO, Heimann P, Bartsch JW, Fuchtbauer A, Fuchtbauer E-M, Schmitt-John T (2013)
International Journal Of Molecular Sciences 14(6): 10908-10925.

Download
Es wurde kein Volltext hochgeladen. Nur Publikationsnachweis!
Zeitschriftenaufsatz | Veröffentlicht | Englisch
Autor
; ; ; ; ; ; ; ; ;
Abstract / Bemerkung
The identification of the mutation causing the phenotype of the amyotrophic lateral sclerosis (ALS) model mouse, wobbler, has linked motor neuron degeneration with retrograde vesicle traffic. The wobbler mutation affects protein stability of Vps54, a ubiquitously expressed vesicle-tethering factor and leads to partial loss of Vps54 function. Moreover, the Vps54 null mutation causes embryonic lethality, which is associated with extensive membrane blebbing in the neural tube and is most likely a consequence of impaired vesicle transport. Investigation of cells derived from wobbler and Vps54 null mutant embryos demonstrates impaired retrograde transport of the Cholera-toxin B subunit to the trans-Golgi network and mis-sorting of mannose-6-phosphate receptors and cargo proteins dependent on retrograde vesicle transport. Endocytosis assays demonstrate no difference between wobbler and wild type cells, indicating that the retrograde vesicle traffic to the trans-Golgi network, but not endocytosis, is affected in Vps54 mutant cells. The results obtained on wobbler cells were extended to test the use of cultured skin fibroblasts from human ALS patients to investigate the retrograde vesicle traffic. Analysis of skin fibroblasts of ALS patients will support the investigation of the critical role of the retrograde vesicle transport in ALS pathogenesis and might yield a diagnostic prospect.
Erscheinungsjahr
Zeitschriftentitel
International Journal Of Molecular Sciences
Band
14
Zeitschriftennummer
6
Seite
10908-10925
ISSN
eISSN
PUB-ID

Zitieren

Karlsson P, Droce A, Moser JM, et al. Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality. International Journal Of Molecular Sciences. 2013;14(6):10908-10925.
Karlsson, P., Droce, A., Moser, J. M., Cuhlmann, S., Padilla, C. O., Heimann, P., Bartsch, J. W., et al. (2013). Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality. International Journal Of Molecular Sciences, 14(6), 10908-10925. doi:10.3390/ijms140610908
Karlsson, P., Droce, A., Moser, J. M., Cuhlmann, S., Padilla, C. O., Heimann, P., Bartsch, J. W., Fuchtbauer, A., Fuchtbauer, E. - M., and Schmitt-John, T. (2013). Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality. International Journal Of Molecular Sciences 14, 10908-10925.
Karlsson, P., et al., 2013. Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality. International Journal Of Molecular Sciences, 14(6), p 10908-10925.
P. Karlsson, et al., “Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality”, International Journal Of Molecular Sciences, vol. 14, 2013, pp. 10908-10925.
Karlsson, P., Droce, A., Moser, J.M., Cuhlmann, S., Padilla, C.O., Heimann, P., Bartsch, J.W., Fuchtbauer, A., Fuchtbauer, E.-M., Schmitt-John, T.: Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality. International Journal Of Molecular Sciences. 14, 10908-10925 (2013).
Karlsson, Pall, Droce, Aida, Moser, Jakob M., Cuhlmann, Simon, Padilla, Carolina Ortiz, Heimann, Peter, Bartsch, Joerg W., Fuchtbauer, Annette, Fuchtbauer, Ernst-Martin, and Schmitt-John, Thomas. “Loss of Vps54 Function Leads to Vesicle Traffic Impairment, Protein Mis-Sorting and Embryonic Lethality”. International Journal Of Molecular Sciences 14.6 (2013): 10908-10925.

13 Zitationen in Europe PMC

Daten bereitgestellt von Europe PubMed Central.

Traffic from the endosome towards trans-Golgi network.
Saimani U, Kim K., Eur J Cell Biol 96(2), 2017
PMID: 28256269
The GARP Complex Is Involved in Intracellular Cholesterol Transport via Targeting NPC2 to Lysosomes.
Wei J, Zhang YY, Luo J, Wang JQ, Zhou YX, Miao HH, Shi XJ, Qu YX, Xu J, Li BL, Song BL., Cell Rep 19(13), 2017
PMID: 28658628
Endocytic membrane trafficking and neurodegenerative disease.
Schreij AM, Fon EA, McPherson PS., Cell Mol Life Sci 73(8), 2016
PMID: 26721251
Membrane dynamics in mammalian embryogenesis: Implication in signal regulation.
Wada Y, Sun-Wada GH, Kawamura N, Yasukawa J., Birth Defects Res C Embryo Today 108(1), 2016
PMID: 26992153
Vaccinia virus uses retromer-independent cellular retrograde transport pathways to facilitate the wrapping of intracellular mature virions during viral morphogenesis.
Harrison K, Haga IR, Pechenick Jowers T, Jasim S, Cintrat JC, Gillet D, Schmitt-John T, Digard P, Beard PM., J Virol (), 2016
PMID: 27581988
Comparative profiling of the sperm proteome.
Holland A, Ohlendieck K., Proteomics 15(4), 2015
PMID: 24909132
Implementation of a manual for working with wobbler mice and criteria for discontinuation of the experiment.
Ott B, Dahlke C, Meller K, Napirei M, Schmitt-John T, Brand-Saberi B, Theiss C, Saberi D., Ann Anat 200(), 2015
PMID: 25929815
Post-Golgi anterograde transport requires GARP-dependent endosome-to-TGN retrograde transport.
Hirata T, Fujita M, Nakamura S, Gotoh K, Motooka D, Murakami Y, Maeda Y, Kinoshita T., Mol Biol Cell 26(17), 2015
PMID: 26157166
VPS54 and the wobbler mouse.
Schmitt-John T., Front Neurosci 9(), 2015
PMID: 26539077
Inflammation and neuronal death in the motor cortex of the wobbler mouse, an ALS animal model.
Dahlke C, Saberi D, Ott B, Brand-Saberi B, Schmitt-John T, Theiss C., J Neuroinflammation 12(), 2015
PMID: 26597538
Intricate effects of primary motor neuronopathy on contractile proteins and metabolic muscle enzymes as revealed by label-free mass spectrometry.
Holland A, Schmitt-John T, Dowling P, Meleady P, Henry M, Clynes M, Ohlendieck K., Biosci Rep 34(4), 2014
PMID: 24895011
TPC1 has two variant isoforms, and their removal has different effects on endo-lysosomal functions compared to loss of TPC2.
Ruas M, Chuang KT, Davis LC, Al-Douri A, Tynan PW, Tunn R, Teboul L, Galione A, Parrington J., Mol Cell Biol 34(21), 2014
PMID: 25135478

30 References

Daten bereitgestellt von Europe PubMed Central.


Nagy A., Gertsenstein M., Vintersten K., Behringer R., 2003
Trafficking of Na,K-ATPase fused to enhanced green fluorescent protein is mediated by protein kinase A or C.
Kristensen B, Birkelund S, Jorgensen PL., J. Membr. Biol. 191(1), 2003
PMID: 12532274
Occurrence of an anomalous endocytic compartment in fibroblasts from Sandhoff disease patients.
Tancini B, Magini A, Latterini L, Urbanelli L, Ciccarone V, Elisei F, Emiliani C., Mol. Cell. Biochem. 335(1-2), 2010
PMID: 19823769
Neuronal sorting protein-related receptor sorLA/LR11 regulates processing of the amyloid precursor protein.
Andersen OM, Reiche J, Schmidt V, Gotthardt M, Spoelgen R, Behlke J, von Arnim CA, Breiderhoff T, Jansen P, Wu X, Bales KR, Cappai R, Masters CL, Gliemann J, Mufson EJ, Hyman BT, Paul SM, Nykjaer A, Willnow TE., Proc. Natl. Acad. Sci. U.S.A. 102(38), 2005
PMID: 16174740

Export

Markieren/ Markierung löschen
Markierte Publikationen

Open Data PUB

Web of Science

Dieser Datensatz im Web of Science®

Quellen

PMID: 23708095
PubMed | Europe PMC

Suchen in

Google Scholar