CPAP insufficiency leads to incomplete centrioles that duplicate but fragment.
Journal
The Journal of cell biology
ISSN: 1540-8140
Titre abrégé: J Cell Biol
Pays: United States
ID NLM: 0375356
Informations de publication
Date de publication:
02 05 2022
02 05 2022
Historique:
received:
12
08
2021
revised:
13
01
2022
accepted:
28
02
2022
entrez:
11
4
2022
pubmed:
12
4
2022
medline:
19
4
2022
Statut:
ppublish
Résumé
Centrioles are structures that assemble centrosomes. CPAP is critical for centrosome assembly, and its mutations are found in patients with diseases such as primary microcephaly. CPAP's centrosomal localization, its dynamics, and the consequences of its insufficiency in human cells are poorly understood. Here we use human cells genetically engineered for fast degradation of CPAP, in combination with superresolution microscopy, to address these uncertainties. We show that three independent centrosomal CPAP populations are dynamically regulated during the cell cycle. We confirm that CPAP is critical for assembly of human centrioles, but not for recruitment of pericentriolar material on already assembled centrioles. Further, we reveal that CPAP insufficiency leads to centrioles with incomplete microtubule triplets that can convert to centrosomes, duplicate, and form mitotic spindle poles, but fragment owing to loss of cohesion between microtubule blades. These findings further our basic understanding of the role of CPAP in centrosome biogenesis and help understand how CPAP aberrations can lead to human diseases.
Identifiants
pubmed: 35404385
pii: 213119
doi: 10.1083/jcb.202108018
pmc: PMC9007748
pii:
doi:
Substances chimiques
CENPJ protein, human
0
Microtubule-Associated Proteins
0
Types de publication
Journal Article
Research Support, N.I.H., Intramural
Langues
eng
Sous-ensembles de citation
IM
Subventions
Organisme : NIH HHS
Pays : United States
Informations de copyright
© 2022 Vásquez Limeta et al.
Références
Nat Commun. 2019 Mar 1;10(1):993
pubmed: 30824690
Biochem Biophys Res Commun. 2014 Feb 21;444(4):644-50
pubmed: 24491538
J Cell Biol. 2013 Jul 22;202(2):211-9
pubmed: 23857771
Nat Methods. 2009 Dec;6(12):917-22
pubmed: 19915560
Mol Biol Cell. 2008 Jan;19(1):115-25
pubmed: 17959831
Biol Open. 2012 Oct 15;1(10):965-76
pubmed: 23213374
J Cell Biochem. 2013 Oct;114(10):2258-72
pubmed: 23606375
Cancer Metastasis Rev. 2009 Jun;28(1-2):85-98
pubmed: 19156503
Dev Cell. 2016 May 23;37(4):362-376
pubmed: 27219064
Biochem Biophys Res Commun. 2006 Jan 20;339(3):742-7
pubmed: 16316625
J Cell Sci. 2010 May 1;123(Pt 9):1414-26
pubmed: 20392737
Science. 2015 Jun 5;348(6239):1155-60
pubmed: 25931445
Methods Mol Biol. 2021;2329:249-263
pubmed: 34085228
Exp Cell Res. 2008 Aug 15;314(14):2591-602
pubmed: 18586240
J Cell Sci. 2010 May 1;123(Pt 9):1407-13
pubmed: 20388734
Elife. 2013 Sep 17;2:e01071
pubmed: 24052813
Curr Biol. 2009 Jun 23;19(12):1005-11
pubmed: 19481458
Cell Rep. 2016 Mar 29;14(12):2975-87
pubmed: 26997271
Philos Trans R Soc Lond B Biol Sci. 2014 Sep 5;369(1650):
pubmed: 25047613
Endocrinol Metab (Seoul). 2015 Mar 27;30(1):53-7
pubmed: 25827458
Dev Biol. 2011 Jan 15;349(2):427-39
pubmed: 20974123
Dev Cell. 2007 Aug;13(2):190-202
pubmed: 17681131
J Cell Sci. 2019 Feb 20;132(4):
pubmed: 30787112
Dev Cell. 2019 May 6;49(3):325-331
pubmed: 31063752
J Cell Sci. 2011 Nov 15;124(Pt 22):3884-93
pubmed: 22100914
J Biol Chem. 2015 Mar 13;290(11):6890-902
pubmed: 25616662
Nat Commun. 2020 Feb 14;11(1):903
pubmed: 32060285
J Biol Chem. 2009 Mar 13;284(11):6909-17
pubmed: 19131341
EMBO J. 2020 Nov 16;39(22):e106246
pubmed: 32954513
J Cell Sci. 2013 Jan 1;126(Pt 1):163-75
pubmed: 23015594
J Cell Sci. 2016 Jul 1;129(13):2501-13
pubmed: 27185865
Chromosome Res. 2017 Oct;25(3-4):253-260
pubmed: 28589221
Sci Adv. 2020 Feb 14;6(7):eaaz4137
pubmed: 32110738
Mol Biol Cell. 2011 Apr;22(7):1035-44
pubmed: 21289083
Dev Cell. 2003 Mar;4(3):431-9
pubmed: 12636923
J Cell Biol. 2009 Apr 6;185(1):101-14
pubmed: 19349582
Dev Cell. 2008 Aug;15(2):187-97
pubmed: 18694559
J Cell Biol. 1982 Jun;93(3):938-49
pubmed: 7119006
Curr Opin Struct Biol. 2021 Feb;66:58-65
pubmed: 33176264
J Cell Biol. 1998 Apr 6;141(1):163-74
pubmed: 9531556
J Pathol. 2017 Jan;241(2):294-309
pubmed: 27859258
Elife. 2019 May 22;8:
pubmed: 31115335
J Hum Genet. 2006;51(9):760-764
pubmed: 16900296
Cell. 2003 Feb 21;112(4):575-87
pubmed: 12600319
Mol Biol Cell. 2004 Jun;15(6):2697-706
pubmed: 15047868
J Cell Biol. 2020 Jun 1;219(6):
pubmed: 32271878
Curr Biol. 2007 Sep 4;17(17):1498-503
pubmed: 17716897
Curr Biol. 2014 Jun 2;24(11):1276-82
pubmed: 24835456
J Cell Biol. 2007 Oct 22;179(2):321-30
pubmed: 17954613
Curr Biol. 2014 Mar 17;24(6):664-70
pubmed: 24613305
J Cell Biol. 2000 Apr 17;149(2):317-30
pubmed: 10769025
J Cell Biol. 2010 Nov 15;191(4):731-9
pubmed: 21059844
J Cell Biol. 2011 May 16;193(4):727-39
pubmed: 21576395
Cells. 2020 Jun 09;9(6):
pubmed: 32526902
EMBO J. 2011 Oct 21;30(23):4790-804
pubmed: 22020124
J Cell Biol. 2007 Jan 15;176(2):173-82
pubmed: 17227892
Curr Biol. 2009 Jun 23;19(12):1012-8
pubmed: 19481460
J Cell Sci. 2012 Mar 1;125(Pt 5):1353-62
pubmed: 22349705
EMBO J. 2020 Nov 16;39(22):e106249
pubmed: 32954505
J Cell Biol. 2002 Jan 7;156(1):87-99
pubmed: 11781336
Elife. 2020 Sep 18;9:
pubmed: 32946374
J Cell Sci. 2020 Jun 24;133(12):
pubmed: 32501282
Cell Res. 2015 May;25(5):642-4
pubmed: 25906994
Dev Cell. 2012 Aug 14;23(2):412-24
pubmed: 22898782
J Microsc. 2019 Dec;276(3):145-159
pubmed: 31691972
Cell Rep. 2018 Dec 26;25(13):3618-3630.e6
pubmed: 30590037
Biol Cell. 2020 Jun;112(6):153-172
pubmed: 32170757
Cell. 2006 Jun 30;125(7):1375-86
pubmed: 16814722
Nature. 2006 Nov 30;444(7119):619-23
pubmed: 17136092
Nat Cell Biol. 2013 Jul;15(7):731-40
pubmed: 23666084
Curr Biol. 2013 Jul 22;23(14):1360-6
pubmed: 23810536
J Cell Biol. 2020 Dec 7;219(12):
pubmed: 33170211
Proc Natl Acad Sci U S A. 2014 Jan 21;111(3):E354-63
pubmed: 24385583
Cell. 2008 Jun 13;133(6):1032-42
pubmed: 18555779
Semin Cell Dev Biol. 2021 Sep;117:30-41
pubmed: 33836946
Development. 2018 Sep 17;145(18):
pubmed: 30224385
BMC Cell Biol. 2001;2:4
pubmed: 11255590
Mol Biol Cell. 2003 Jul;14(7):2999-3012
pubmed: 12857881
Nat Protoc. 2013 Nov;8(11):2281-2308
pubmed: 24157548
J Cell Sci. 2013 Jul 15;126(Pt 14):3223-33
pubmed: 23641073
Nat Commun. 2016 Jun 16;7:11874
pubmed: 27306797
Elife. 2017 Sep 14;6:
pubmed: 28906251
Nat Cell Biol. 2009 Jul;11(7):825-31
pubmed: 19503075
Curr Biol. 2007 Nov 20;17(22):1960-6
pubmed: 17980596
Mol Cell Biol. 2000 Oct;20(20):7813-25
pubmed: 11003675
J Cell Biol. 2008 Feb 25;180(4):771-85
pubmed: 18299348
FEBS Lett. 2014 Aug 1;588(15):2366-72
pubmed: 24951839
Nat Commun. 2011 Jun 21;2:359
pubmed: 21694707
Cold Spring Harb Symp Quant Biol. 2017;82:137-144
pubmed: 29610243
Elife. 2015 Nov 04;4:
pubmed: 26530814
Nature. 2010 Oct 7;467(7316):714-8
pubmed: 20852615
EMBO J. 2013 Apr 17;32(8):1141-54
pubmed: 23511974
Nat Med. 2014 Jun;20(6):616-23
pubmed: 24813252
J Cell Biol. 2009 Dec 14;187(6):905-20
pubmed: 20008567
Curr Opin Struct Biol. 2021 Feb;66:74-82
pubmed: 33186811
Nat Genet. 2005 Apr;37(4):353-5
pubmed: 15793586
Dev Cell. 2017 Feb 6;40(3):313-322.e5
pubmed: 28132847