scholarly journals Tissue‐specific variation in nonsense mutant transcript level and drug‐induced read‐through efficiency in the Cln1 R151X mouse model of INCL

2016 ◽  
Vol 20 (2) ◽  
pp. 381-385 ◽  
Author(s):  
Vaughn Thada ◽  
Jake N. Miller ◽  
Attila D. Kovács ◽  
David A. Pearce
2010 ◽  
Vol 39 (3) ◽  
pp. 283-291 ◽  
Author(s):  
Richard M. Gustin ◽  
Terry Jo Bichell ◽  
Michael Bubser ◽  
Jennifer Daily ◽  
Irina Filonova ◽  
...  

2021 ◽  
Author(s):  
Elisabeth Holzer ◽  
Cornelia Rumpf-Kienzl ◽  
Sebastian Falk ◽  
Alexander Dammermann

Proximity-dependent labeling approaches such as BioID have been a great boon to studies of protein-protein interactions in the context of cytoskeletal structures such as centrosomes which are poorly amenable to traditional biochemical approaches like immunoprecipitation and tandem affinity purification. Yet, these methods have so far not been applied extensively to invertebrate experimental models such as C. elegans given the long labeling times required for the original promiscuous biotin ligase variant BirA*. Here, we show that the recently developed variant TurboID successfully probes the interactomes of both stably associated (SPD-5) and dynamically localized (PLK-1) centrosomal components. We further develop an indirect proximity labeling method employing a GFP nanobody- TurboID fusion, which allows the identification of protein interactors in a tissue-specific manner in the context of the whole animal. Critically, this approach utilizes available endogenous GFP fusions, avoiding the need to generate multiple additional strains for each target protein and the potential complications associated with overexpressing the protein from transgenes. Using this method, we identify homologs of two highly conserved centriolar components, Cep97 and Bld10/Cep135, which are present in various somatic tissues of the worm. Surprisingly, neither protein is expressed in early embryos, likely explaining why these proteins have escaped attention until now. Our work expands the experimental repertoire for C. elegans and opens the door for further studies of tissue-specific variation in centrosome architecture.


2018 ◽  
Vol 8 (1) ◽  
Author(s):  
Hyeongseok Kim ◽  
Yeong Gi Kim ◽  
Wonsuk Choi ◽  
Joon Ho Moon ◽  
Inseon Hwang ◽  
...  

2018 ◽  
Vol 115 (52) ◽  
pp. E12305-E12312 ◽  
Author(s):  
Meng Qu ◽  
Tomas Duffy ◽  
Tsuyoshi Hirota ◽  
Steve A. Kay

Either expression level or transcriptional activity of various nuclear receptors (NRs) have been demonstrated to be under circadian control. With a few exceptions, little is known about the roles of NRs as direct regulators of the circadian circuitry. Here we show that the nuclear receptor HNF4A strongly transrepresses the transcriptional activity of the CLOCK:BMAL1 heterodimer. We define a central role for HNF4A in maintaining cell-autonomous circadian oscillations in a tissue-specific manner in liver and colon cells. Not only transcript level but also genome-wide chromosome binding of HNF4A is rhythmically regulated in the mouse liver. ChIP-seq analyses revealed cooccupancy of HNF4A and CLOCK:BMAL1 at a wide array of metabolic genes involved in lipid, glucose, and amino acid homeostasis. Taken together, we establish that HNF4A defines a feedback loop in tissue-specific mammalian oscillators and demonstrate its recruitment in the circadian regulation of metabolic pathways.


Pancreas ◽  
2005 ◽  
Vol 31 (4) ◽  
pp. 478
Author(s):  
C Wei ◽  
A Rashid ◽  
C Amos ◽  
M Gannon ◽  
M L Frazier

1995 ◽  
Vol 40 (1) ◽  
pp. 1-8 ◽  
Author(s):  
J. S. Fleming ◽  
S. M. Galloway ◽  
R. J. Crawford ◽  
D. J. Tisdall ◽  
P. J. Greenwood

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