Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.

Activity of separase, a cysteine protease that cleaves sister chromatid cohesin at the onset of anaphase, is tightly regulated to ensure faithful chromosome segregation and genome stability. Two mechanisms negatively regulate separase: inhibition by securin and phosphorylation on serine 1121. To gau...

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Main Authors: Xingxu Huang, Claudia V Andreu-Vieyra, J Philippe York, Rashieda Hatcher, Tao Lu, Martin M Matzuk, Pumin Zhang
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2008-01-01
Series:PLoS Biology
Online Access:https://journals.plos.org/plosbiology/article/file?id=10.1371/journal.pbio.0060015&type=printable
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author Xingxu Huang
Claudia V Andreu-Vieyra
J Philippe York
Rashieda Hatcher
Tao Lu
Martin M Matzuk
Pumin Zhang
author_facet Xingxu Huang
Claudia V Andreu-Vieyra
J Philippe York
Rashieda Hatcher
Tao Lu
Martin M Matzuk
Pumin Zhang
author_sort Xingxu Huang
collection DOAJ
description Activity of separase, a cysteine protease that cleaves sister chromatid cohesin at the onset of anaphase, is tightly regulated to ensure faithful chromosome segregation and genome stability. Two mechanisms negatively regulate separase: inhibition by securin and phosphorylation on serine 1121. To gauge the physiological significance of the inhibitory phosphorylation, we created a mouse strain in which Ser1121 was mutated to Ala (S1121A). Here we report that this S1121A point mutation causes infertility in mice. We show that germ cells in the mutants are depleted during development. We further demonstrate that S1121A causes chromosome misalignment during proliferation of the postmigratory primordial germ cells, resulting in mitotic arrest, aneuploidy, and eventual cell death. Our results indicate that inhibitory phosphorylation of separase plays a critical role in the maintenance of sister chromatid cohesion and genome stability in proliferating postmigratory primordial germ cells.
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language English
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spelling doaj-art-d85fe5d4661f4440a5cf9952d83a82082025-08-20T03:22:35ZengPublic Library of Science (PLoS)PLoS Biology1544-91731545-78852008-01-0161e1510.1371/journal.pbio.0060015Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.Xingxu HuangClaudia V Andreu-VieyraJ Philippe YorkRashieda HatcherTao LuMartin M MatzukPumin ZhangActivity of separase, a cysteine protease that cleaves sister chromatid cohesin at the onset of anaphase, is tightly regulated to ensure faithful chromosome segregation and genome stability. Two mechanisms negatively regulate separase: inhibition by securin and phosphorylation on serine 1121. To gauge the physiological significance of the inhibitory phosphorylation, we created a mouse strain in which Ser1121 was mutated to Ala (S1121A). Here we report that this S1121A point mutation causes infertility in mice. We show that germ cells in the mutants are depleted during development. We further demonstrate that S1121A causes chromosome misalignment during proliferation of the postmigratory primordial germ cells, resulting in mitotic arrest, aneuploidy, and eventual cell death. Our results indicate that inhibitory phosphorylation of separase plays a critical role in the maintenance of sister chromatid cohesion and genome stability in proliferating postmigratory primordial germ cells.https://journals.plos.org/plosbiology/article/file?id=10.1371/journal.pbio.0060015&type=printable
spellingShingle Xingxu Huang
Claudia V Andreu-Vieyra
J Philippe York
Rashieda Hatcher
Tao Lu
Martin M Matzuk
Pumin Zhang
Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.
PLoS Biology
title Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.
title_full Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.
title_fullStr Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.
title_full_unstemmed Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.
title_short Inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells.
title_sort inhibitory phosphorylation of separase is essential for genome stability and viability of murine embryonic germ cells
url https://journals.plos.org/plosbiology/article/file?id=10.1371/journal.pbio.0060015&type=printable
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