Biogenesis and function of centriolar
and ciliary structures
Publications
3888256
BIOCIL
chicago-author-date
50
date
desc
year
14250
https://www.i2bc.paris-saclay.fr/wp-content/plugins/zotpress/
%7B%22status%22%3A%22success%22%2C%22updateneeded%22%3Afalse%2C%22instance%22%3A%22zotpress-23c7ef93c86a6d7b1e594a79106ba064%22%2C%22meta%22%3A%7B%22request_last%22%3A0%2C%22request_next%22%3A0%2C%22used_cache%22%3Atrue%7D%2C%22data%22%3A%5B%7B%22key%22%3A%2256JEGQL5%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Carvalhal%20et%20al.%22%2C%22parsedDate%22%3A%222024%22%2C%22numChildren%22%3A1%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3ECarvalhal%2C%20Sara%2C%20Bruno%20Carmona%2C%20Anne-Marie%20Tassin%2C%20and%20Jo%26%23xE3%3Bo%20Gon%26%23xE7%3Balves.%202024.%20%26%23x201C%3BEditorial%3A%20Molecular%20Mechanisms%20of%20Cilia%20Related%20Diseases.%26%23x201D%3B%20%3Ci%3EFrontiers%20in%20Molecular%20Biosciences%3C%5C%2Fi%3E%2011%3A1421419.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.3389%5C%2Ffmolb.2024.1421419%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.3389%5C%2Ffmolb.2024.1421419%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Editorial%3A%20Molecular%20mechanisms%20of%20cilia%20related%20diseases%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Sara%22%2C%22lastName%22%3A%22Carvalhal%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Bruno%22%2C%22lastName%22%3A%22Carmona%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jo%5Cu00e3o%22%2C%22lastName%22%3A%22Gon%5Cu00e7alves%22%7D%5D%2C%22abstractNote%22%3A%22%22%2C%22date%22%3A%222024%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.3389%5C%2Ffmolb.2024.1421419%22%2C%22ISSN%22%3A%222296-889X%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%5D%2C%22dateModified%22%3A%222024-06-07T14%3A05%3A36Z%22%7D%7D%2C%7B%22key%22%3A%22SD5372YN%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Bodin%20et%20al.%22%2C%22parsedDate%22%3A%222023-07-06%22%2C%22numChildren%22%3A1%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EBodin%2C%20Alexia%2C%20Logan%20Greibill%2C%20Julien%20Gouju%2C%20Franck%20Letournel%2C%20Silvia%20Pozzi%2C%20Jean-Pierre%20Julien%2C%20Laurence%20Renaud%2C%20et%20al.%202023.%20%26%23x201C%3BTransactive%20Response%20DNA-Binding%20Protein%2043%20Is%20Enriched%20at%20the%20Centrosome%20in%20Human%20Cells.%26%23x201D%3B%20%3Ci%3EBrain%3A%20A%20Journal%20of%20Neurology%3C%5C%2Fi%3E%2C%20July%2C%20awad228.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1093%5C%2Fbrain%5C%2Fawad228%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1093%5C%2Fbrain%5C%2Fawad228%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Transactive%20response%20DNA-binding%20protein%2043%20is%20enriched%20at%20the%20centrosome%20in%20human%20cells%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Alexia%22%2C%22lastName%22%3A%22Bodin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Logan%22%2C%22lastName%22%3A%22Greibill%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Julien%22%2C%22lastName%22%3A%22Gouju%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Franck%22%2C%22lastName%22%3A%22Letournel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Silvia%22%2C%22lastName%22%3A%22Pozzi%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jean-Pierre%22%2C%22lastName%22%3A%22Julien%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Laurence%22%2C%22lastName%22%3A%22Renaud%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Delphine%22%2C%22lastName%22%3A%22Bohl%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22St%5Cu00e9phanie%22%2C%22lastName%22%3A%22Millecamps%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Christophe%22%2C%22lastName%22%3A%22Verny%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Julien%22%2C%22lastName%22%3A%22Cassereau%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Guy%22%2C%22lastName%22%3A%22Lenaers%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Arnaud%22%2C%22lastName%22%3A%22Chevrollier%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Philippe%22%2C%22lastName%22%3A%22Codron%22%7D%5D%2C%22abstractNote%22%3A%22The%20centrosome%2C%20as%20the%20main%20microtubule%20organizing%20center%2C%20plays%20key%20roles%20in%20cell%20polarity%2C%20genome%20stability%20and%20ciliogenesis.%20The%20recent%20identification%20of%20ribosomes%2C%20RNA-binding%20proteins%20and%20transcripts%20at%20the%20centrosome%20suggests%20local%20protein%20synthesis.%20In%20this%20context%2C%20we%20hypothesized%20that%20TDP-43%2C%20a%20highly%20conserved%20RNA%20binding%20protein%20involved%20in%20the%20pathophysiology%20of%20amyotrophic%20lateral%20sclerosis%20and%20frontotemporal%20lobar%20degeneration%2C%20could%20be%20enriched%20at%20this%20organelle.%20Using%20dedicated%20high-magnification%20sub-diffraction%20microscopy%20on%20human%20cells%2C%20we%20discovered%20a%20novel%20localization%20of%20TDP-43%20at%20the%20centrosome%20during%20all%20phases%20of%20the%20cell%20cycle.%20These%20results%20were%20confirmed%20on%20purified%20centrosomes%20by%20western%20blot%20and%20immunofluorescence%20microscopy.%20In%20addition%2C%20the%20colocalization%20of%20TDP-43%20and%20pericentrin%20suggested%20a%20pericentriolar%20enrichment%20of%20the%20protein%2C%20leading%20us%20to%20hypothesize%20that%20TDP-43%20might%20interact%20with%20local%20mRNAs%20and%20proteins.%20Supporting%20this%20hypothesis%2C%20we%20found%204%20conserved%20centrosomal%20mRNAs%20and%2016%20centrosomal%20proteins%20identified%20as%20direct%20TDP-43%20interactors.%20More%20strikingly%2C%20all%20the%2016%20proteins%20are%20implicated%20in%20the%20pathophysiology%20of%20TDP-43%20proteinopathies%2C%20suggesting%20that%20TDP-43%20dysfunction%20in%20this%20organelle%20contributes%20to%20neurodegeneration.%20This%20first%20description%20of%20TDP-43%20centrosomal%20enrichment%20paves%20the%20way%20for%20a%20more%20comprehensive%20understanding%20of%20TDP-43%20physiology%20and%20pathology.%22%2C%22date%22%3A%222023-07-06%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.1093%5C%2Fbrain%5C%2Fawad228%22%2C%22ISSN%22%3A%221460-2156%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%5D%2C%22dateModified%22%3A%222023-07-10T08%3A56%3A35Z%22%7D%7D%2C%7B%22key%22%3A%22RG6FZAEZ%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Borgne%20et%20al.%22%2C%22parsedDate%22%3A%222022-09-07%22%2C%22numChildren%22%3A6%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EBorgne%2C%20Pierrick%20Le%2C%20Logan%20Greibill%2C%20Marine%20H%26%23xE9%3Bl%26%23xE8%3Bne%20Laporte%2C%20Michel%20Lemullois%2C%20Khaled%20Bouhouche%2C%20Mebarek%20Temagoult%2C%20Olivier%20Rosnet%2C%20et%20al.%202022.%20%26%23x201C%3BThe%20Evolutionary%20Conserved%20Proteins%20CEP90%2C%20FOPNL%2C%20and%20OFD1%20Recruit%20Centriolar%20Distal%20Appendage%20Proteins%20to%20Initiate%20Their%20Assembly.%26%23x201D%3B%20%3Ci%3EPLOS%20Biology%3C%5C%2Fi%3E%2020%20%289%29%3A%20e3001782.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1371%5C%2Fjournal.pbio.3001782%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1371%5C%2Fjournal.pbio.3001782%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22The%20evolutionary%20conserved%20proteins%20CEP90%2C%20FOPNL%2C%20and%20OFD1%20recruit%20centriolar%20distal%20appendage%20proteins%20to%20initiate%20their%20assembly%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Pierrick%20Le%22%2C%22lastName%22%3A%22Borgne%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Logan%22%2C%22lastName%22%3A%22Greibill%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Marine%20H%5Cu00e9l%5Cu00e8ne%22%2C%22lastName%22%3A%22Laporte%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Khaled%22%2C%22lastName%22%3A%22Bouhouche%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Mebarek%22%2C%22lastName%22%3A%22Temagoult%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Olivier%22%2C%22lastName%22%3A%22Rosnet%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Maeva%20Le%22%2C%22lastName%22%3A%22Guennec%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Laurent%22%2C%22lastName%22%3A%22Ligni%5Cu00e8res%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Guillaume%22%2C%22lastName%22%3A%22Chevreux%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Virginie%22%2C%22lastName%22%3A%22Hamel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Paul%22%2C%22lastName%22%3A%22Guichard%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%5D%2C%22abstractNote%22%3A%22In%20metazoa%2C%20cilia%20assembly%20is%20a%20cellular%20process%20that%20starts%20with%20centriole%20to%20basal%20body%20maturation%2C%20migration%20to%20the%20cell%20surface%2C%20and%20docking%20to%20the%20plasma%20membrane.%20Basal%20body%20docking%20involves%20the%20interaction%20of%20both%20the%20distal%20end%20of%20the%20basal%20body%20and%20the%20transition%20fibers%5C%2Fdistal%20appendages%2C%20with%20the%20plasma%20membrane.%20Mutations%20in%20numerous%20genes%20involved%20in%20basal%20body%20docking%20and%20transition%20zone%20assembly%20are%20associated%20with%20the%20most%20severe%20ciliopathies%2C%20highlighting%20the%20importance%20of%20these%20events%20in%20cilium%20biogenesis.%20In%20this%20context%2C%20the%20ciliate%20Paramecium%20has%20been%20widely%20used%20as%20a%20model%20system%20to%20study%20basal%20body%20and%20cilia%20assembly.%20However%2C%20despite%20the%20evolutionary%20conservation%20of%20cilia%20assembly%20events%20across%20phyla%2C%20whether%20the%20same%20molecular%20players%20are%20functionally%20conserved%2C%20is%20not%20fully%20known.%20Here%2C%20we%20demonstrated%20that%20CEP90%2C%20FOPNL%2C%20and%20OFD1%20are%20evolutionary%20conserved%20proteins%20crucial%20for%20ciliogenesis.%20Using%20ultrastructure%20expansion%20microscopy%2C%20we%20unveiled%20that%20these%20proteins%20localize%20at%20the%20distal%20end%20of%20both%20centrioles%5C%2Fbasal%20bodies%20in%20Paramecium%20and%20mammalian%20cells.%20Moreover%2C%20we%20found%20that%20these%20proteins%20are%20recruited%20early%20during%20centriole%20duplication%20on%20the%20external%20surface%20of%20the%20procentriole.%20Functional%20analysis%20performed%20both%20in%20Paramecium%20and%20mammalian%20cells%20demonstrate%20the%20requirement%20of%20these%20proteins%20for%20distal%20appendage%20assembly%20and%20basal%20body%20docking.%20Finally%2C%20we%20show%20that%20mammalian%20centrioles%20require%20another%20component%2C%20Moonraker%20%28MNR%29%2C%20to%20recruit%20OFD1%2C%20FOPNL%2C%20and%20CEP90%2C%20which%20will%20then%20recruit%20the%20distal%20appendage%20proteins%20CEP83%2C%20CEP89%2C%20and%20CEP164.%20Altogether%2C%20we%20propose%20that%20this%20OFD1%2C%20FOPNL%2C%20and%20CEP90%20functional%20module%20is%20required%20to%20determine%20in%20mammalian%20cells%20the%20future%20position%20of%20distal%20appendage%20proteins.%22%2C%22date%22%3A%222022-09-07%22%2C%22language%22%3A%22en%22%2C%22DOI%22%3A%2210.1371%5C%2Fjournal.pbio.3001782%22%2C%22ISSN%22%3A%221545-7885%22%2C%22url%22%3A%22https%3A%5C%2F%5C%2Fjournals.plos.org%5C%2Fplosbiology%5C%2Farticle%3Fid%3D10.1371%5C%2Fjournal.pbio.3001782%22%2C%22collections%22%3A%5B%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222023-12-14T15%3A36%3A22Z%22%7D%7D%2C%7B%22key%22%3A%22ZI9DM8KW%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Bouhouche%20et%20al.%22%2C%22parsedDate%22%3A%222022-04-14%22%2C%22numChildren%22%3A0%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EBouhouche%2C%20K.%2C%20M.%20S.%20Valentine%2C%20P.%20Le%20Borgne%2C%20M.%20Lemullois%2C%20J.%20Yano%2C%20S.%20Lodh%2C%20A.%20Nabi%2C%20A.%20M.%20Tassin%2C%20and%20J.%20L.%20Van%20Houten.%202022.%20%26%23x201C%3BParamecium%2C%20a%20Model%20to%20Study%20Ciliary%20Beating%20and%20Ciliogenesis%3A%20Insights%20From%20Cutting-Edge%20Approaches.%26%23x201D%3B%20%3Ci%3EFrontiers%20in%20Cell%20and%20Developmental%20Biology%3C%5C%2Fi%3E%2010%20%28April%29.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fwww.frontiersin.org%5C%2Farticle%5C%2F10.3389%5C%2Ffcell.2022.847908%27%3Ehttps%3A%5C%2F%5C%2Fwww.frontiersin.org%5C%2Farticle%5C%2F10.3389%5C%2Ffcell.2022.847908%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Paramecium%2C%20a%20Model%20to%20Study%20Ciliary%20Beating%20and%20Ciliogenesis%3A%20Insights%20From%20Cutting-Edge%20Approaches%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22K.%22%2C%22lastName%22%3A%22Bouhouche%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22M.%20S.%22%2C%22lastName%22%3A%22Valentine%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22P.%22%2C%22lastName%22%3A%22Le%20Borgne%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22M.%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22J.%22%2C%22lastName%22%3A%22Yano%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22S.%22%2C%22lastName%22%3A%22Lodh%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22A.%22%2C%22lastName%22%3A%22Nabi%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22A.%20M.%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22J.%20L.%22%2C%22lastName%22%3A%22Van%20Houten%22%7D%5D%2C%22abstractNote%22%3A%22Cilia%20are%20ubiquitous%20and%20highly%20conserved%20extensions%20that%20endow%20the%20cell%20with%20motility%20and%20sensory%20functions.%20They%20were%20present%20in%20the%20first%20eukaryotes%20and%20conserved%20throughout%20evolution%20%28Carvalho-Santos%20et%20al.%2C%202011%29.%20Paramecium%20has%20around%204%2C000%20motile%20cilia%20on%20its%20surface%20arranged%20in%20longitudinal%20rows%2C%20beating%20in%20waves%20to%20ensure%20movement%20and%20feeding.%20As%20with%20cilia%20in%20other%20model%20organisms%2C%20direction%20and%20speed%20of%20Paramecium%20ciliary%20beating%20is%20under%20bioelectric%20control%20of%20ciliary%20ion%20channels.%20In%20multiciliated%20cells%20of%20metazoans%20as%20well%20as%20paramecia%2C%20the%20cilia%20become%20physically%20entrained%20to%20beat%20in%20metachronal%20waves.%20This%20ciliated%20organism%2C%20Paramecium%2C%20is%20an%20attractive%20model%20for%20multidisciplinary%20approaches%20to%20dissect%20the%20location%2C%20structure%20and%20function%20of%20ciliary%20ion%20channels%20and%20other%20proteins%20involved%20in%20ciliary%20beating.%20Swimming%20behavior%20also%20can%20be%20a%20read-out%20of%20the%20role%20of%20cilia%20in%20sensory%20signal%20transduction.%20A%20cilium%20emanates%20from%20a%20BB%2C%20structurally%20equivalent%20to%20the%20centriole%20anchored%20at%20the%20cell%20surface%2C%20and%20elongates%20an%20axoneme%20composed%20of%20microtubule%20doublets%20enclosed%20in%20a%20ciliary%20membrane%20contiguous%20with%20the%20plasma%20membrane.%20The%20connection%20between%20the%20BB%20and%20the%20axoneme%20constitutes%20the%20transition%20zone%2C%20which%20serves%20as%20a%20diffusion%20barrier%20between%20the%20intracellular%20space%20and%20the%20cilium%2C%20defining%20the%20ciliary%20compartment.%20Human%20pathologies%20affecting%20cilia%20structure%20or%20function%2C%20are%20called%20ciliopathies%2C%20which%20are%20caused%20by%20gene%20mutations.%20For%20that%20reason%2C%20the%20molecular%20mechanisms%20and%20structural%20aspects%20of%20cilia%20assembly%20and%20function%20are%20actively%20studied%20using%20a%20variety%20of%20model%20systems%2C%20ranging%20from%20unicellular%20organisms%20to%20metazoa.%20In%20this%20review%2C%20we%20will%20highlight%20the%20use%20of%20Paramecium%20as%20a%20model%20to%20decipher%20ciliary%20beating%20mechanisms%20as%20well%20as%20high%20resolution%20insights%20into%20BB%20structure%20and%20anchoring.%20We%20will%20show%20that%20study%20of%20cilia%20in%20Paramecium%20promotes%20our%20understanding%20of%20cilia%20formation%20and%20function.%20In%20addition%2C%20we%20demonstrate%20that%20Paramecium%20could%20be%20a%20useful%20tool%20to%20validate%20candidate%20genes%20for%20ciliopathies.%22%2C%22date%22%3A%222022-04-14%22%2C%22language%22%3A%22%22%2C%22DOI%22%3A%22%22%2C%22ISSN%22%3A%222296-634X%22%2C%22url%22%3A%22https%3A%5C%2F%5C%2Fwww.frontiersin.org%5C%2Farticle%5C%2F10.3389%5C%2Ffcell.2022.847908%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222022-04-04T09%3A39%3A13Z%22%7D%7D%2C%7B%22key%22%3A%22FGD2QD9J%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Klena%20et%20al.%22%2C%22parsedDate%22%3A%222020-11-16%22%2C%22numChildren%22%3A6%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EKlena%2C%20Nikolai%2C%20Maeva%20Le%20Guennec%2C%20Anne-Marie%20Tassin%2C%20Hugo%20van%20den%20Hoek%2C%20Philipp%20S%20Erdmann%2C%20Miroslava%20Schaffer%2C%20Stefan%20Geimer%2C%20et%20al.%202020.%20%26%23x201C%3BArchitecture%20of%20the%20Centriole%20Cartwheel-Containing%20Region%20Revealed%20by%20Cryo-Electron%20Tomography.%26%23x201D%3B%20%3Ci%3EThe%20EMBO%20Journal%3C%5C%2Fi%3E%2039%20%2822%29%3A%20e106246.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.15252%5C%2Fembj.2020106246%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.15252%5C%2Fembj.2020106246%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Architecture%20of%20the%20centriole%20cartwheel-containing%20region%20revealed%20by%20cryo-electron%20tomography%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Nikolai%22%2C%22lastName%22%3A%22Klena%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Maeva%22%2C%22lastName%22%3A%22Le%20Guennec%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Hugo%22%2C%22lastName%22%3A%22van%20den%20Hoek%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Philipp%20S%22%2C%22lastName%22%3A%22Erdmann%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Miroslava%22%2C%22lastName%22%3A%22Schaffer%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Stefan%22%2C%22lastName%22%3A%22Geimer%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Gabriel%22%2C%22lastName%22%3A%22Aeschlimann%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lubomir%22%2C%22lastName%22%3A%22Kovacik%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Yashar%22%2C%22lastName%22%3A%22Sadian%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Kenneth%20N%22%2C%22lastName%22%3A%22Goldie%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Henning%22%2C%22lastName%22%3A%22Stahlberg%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Benjamin%20D%22%2C%22lastName%22%3A%22Engel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Virginie%22%2C%22lastName%22%3A%22Hamel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Paul%22%2C%22lastName%22%3A%22Guichard%22%7D%5D%2C%22abstractNote%22%3A%22Abstract%20Centrioles%20are%20evolutionarily%20conserved%20barrels%20of%20microtubule%20triplets%20that%20form%20the%20core%20of%20the%20centrosome%20and%20the%20base%20of%20the%20cilium.%20While%20the%20crucial%20role%20of%20the%20proximal%20region%20in%20centriole%20biogenesis%20has%20been%20well%20documented%2C%20its%20native%20architecture%20and%20evolutionary%20conservation%20remain%20relatively%20unexplored.%20Here%2C%20using%20cryo-electron%20tomography%20of%20centrioles%20from%20four%20evolutionarily%20distant%20species%2C%20we%20report%20on%20the%20architectural%20diversity%20of%20the%20centriole%27s%20proximal%20cartwheel-bearing%20region.%20Our%20work%20reveals%20that%20the%20cartwheel%20central%20hub%20is%20constructed%20from%20a%20stack%20of%20paired%20rings%20with%20cartwheel%20inner%20densities%20inside.%20In%20both%20Paramecium%20and%20Chlamydomonas%2C%20the%20repeating%20structural%20unit%20of%20the%20cartwheel%20has%20a%20periodicity%20of%2025%5Cu00a0nm%20and%20consists%20of%20three%20ring%20pairs%2C%20with%206%20radial%20spokes%20emanating%20and%20merging%20into%20a%20single%20bundle%20that%20connects%20to%20the%20microtubule%20triplet%20via%20the%20D2-rod%20and%20the%20pinhead.%20Finally%2C%20we%20identified%20that%20the%20cartwheel%20is%20indirectly%20connected%20to%20the%20A-C%20linker%20through%20the%20triplet%20base%20structure%20extending%20from%20the%20pinhead.%20Together%2C%20our%20work%20provides%20unprecedented%20evolutionary%20insights%20into%20the%20architecture%20of%20the%20centriole%20proximal%20region%2C%20which%20underlies%20centriole%20biogenesis.%22%2C%22date%22%3A%222020-11-16%22%2C%22language%22%3A%22English%22%2C%22DOI%22%3A%2210.15252%5C%2Fembj.2020106246%22%2C%22ISSN%22%3A%220261-4189%22%2C%22url%22%3A%22https%3A%5C%2F%5C%2Fwww.embopress.org%5C%2Fdoi%5C%2Ffull%5C%2F10.15252%5C%2Fembj.2020106246%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222022-09-12T06%3A57%3A25Z%22%7D%7D%2C%7B%22key%22%3A%22S8BDMEMI%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Steib%20et%20al.%22%2C%22parsedDate%22%3A%222020-09-18%22%2C%22numChildren%22%3A2%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3ESteib%2C%20Emmanuelle%2C%20Marine%20H.%20Laporte%2C%20Davide%20Gambarotto%2C%20Natacha%20Olieric%2C%20Celine%20Zheng%2C%20Susanne%20Borgers%2C%20Vincent%20Olieric%2C%20et%20al.%202020.%20%26%23x201C%3BWDR90%20Is%20a%20Centriolar%20Microtubule%20Wall%20Protein%20Important%20for%20Centriole%20Architecture%20Integrity.%26%23x201D%3B%20%3Ci%3EElife%3C%5C%2Fi%3E%209%20%28September%29%3Ae57205.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.7554%5C%2FeLife.57205%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.7554%5C%2FeLife.57205%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22WDR90%20is%20a%20centriolar%20microtubule%20wall%20protein%20important%20for%20centriole%20architecture%20integrity%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Emmanuelle%22%2C%22lastName%22%3A%22Steib%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Marine%20H.%22%2C%22lastName%22%3A%22Laporte%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Davide%22%2C%22lastName%22%3A%22Gambarotto%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Natacha%22%2C%22lastName%22%3A%22Olieric%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Celine%22%2C%22lastName%22%3A%22Zheng%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Susanne%22%2C%22lastName%22%3A%22Borgers%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Vincent%22%2C%22lastName%22%3A%22Olieric%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Maeva%22%2C%22lastName%22%3A%22Le%20Guennec%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%20O.%22%2C%22lastName%22%3A%22Steinmetz%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Paul%22%2C%22lastName%22%3A%22Guichard%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Virginie%22%2C%22lastName%22%3A%22Hamel%22%7D%5D%2C%22abstractNote%22%3A%22Centrioles%20are%20characterized%20by%20a%20nine-fold%20arrangement%20of%20microtubule%20triplets%20held%20together%20by%20an%20inner%20protein%20scaffold.%20These%20structurally%20robust%20organelles%20experience%20strenuous%20cellular%20processes%20such%20as%20cell%20division%20or%20ciliary%20beating%20while%20performing%20their%20function.%20However%2C%20the%20molecular%20mechanisms%20underlying%20the%20stability%20of%20microtubule%20triplets%2C%20as%20well%20as%20centriole%20architectural%20integrity%20remain%20poorly%20understood.%20Here%2C%20using%20ultrastructure%20expansion%20microscopy%20for%20nanoscale%20protein%20mapping%2C%20we%20reveal%20that%20POC16%20and%20its%20human%20homolog%20WDR90%20are%20components%20of%20the%20microtubule%20wall%20along%20the%20central%20core%20region%20of%20the%20centriole.%20We%20further%20found%20that%20WDR90%20is%20an%20evolutionary%20microtubule%20associated%20protein.%20Finally%2C%20we%20demonstrate%20that%20WDR90%20depletion%20impairs%20the%20localization%20of%20inner%20scaffold%20components%2C%20leading%20to%20centriole%20structural%20abnormalities%20in%20human%20cells.%20Altogether%2C%20this%20work%20highlights%20that%20WDR90%20is%20an%20evolutionary%20conserved%20molecular%20player%20participating%20in%20centriole%20architecture%20integrity.%22%2C%22date%22%3A%222020-09-18%22%2C%22language%22%3A%22English%22%2C%22DOI%22%3A%2210.7554%5C%2FeLife.57205%22%2C%22ISSN%22%3A%222050-084X%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222022-09-12T06%3A58%3A50Z%22%7D%7D%2C%7B%22key%22%3A%22KAPUW2EG%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Gogendeau%20et%20al.%22%2C%22parsedDate%22%3A%222020-03-12%22%2C%22numChildren%22%3A2%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EGogendeau%2C%20Delphine%2C%20Michel%20Lemullois%2C%20Pierrick%20Le%20Borgne%2C%20Manon%20Castelli%2C%20Anne%20Aubusson-Fleury%2C%20Olivier%20Arnaiz%2C%20Jean%20Cohen%2C%20et%20al.%202020.%20%26%23x201C%3BMKS-NPHP%20Module%20Proteins%20Control%20Ciliary%20Shedding%20at%20the%20Transition%20Zone.%26%23x201D%3B%20%3Ci%3EPLoS%20Biology%3C%5C%2Fi%3E%2018%20%283%29%3A%20e3000640.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1371%5C%2Fjournal.pbio.3000640%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1371%5C%2Fjournal.pbio.3000640%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22MKS-NPHP%20module%20proteins%20control%20ciliary%20shedding%20at%20the%20transition%20zone%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Delphine%22%2C%22lastName%22%3A%22Gogendeau%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Pierrick%22%2C%22lastName%22%3A%22Le%20Borgne%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Manon%22%2C%22lastName%22%3A%22Castelli%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne%22%2C%22lastName%22%3A%22Aubusson-Fleury%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Olivier%22%2C%22lastName%22%3A%22Arnaiz%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jean%22%2C%22lastName%22%3A%22Cohen%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Christine%22%2C%22lastName%22%3A%22Vesque%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Sylvie%22%2C%22lastName%22%3A%22Schneider-Maunoury%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Khaled%22%2C%22lastName%22%3A%22Bouhouche%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%5D%2C%22abstractNote%22%3A%22Ciliary%20shedding%20occurs%20from%20unicellular%20organisms%20to%20metazoans.%20Although%20required%20during%20the%20cell%20cycle%20and%20during%20neurogenesis%2C%20the%20process%20remains%20poorly%20understood.%20In%20all%20cellular%20models%2C%20this%20phenomenon%20occurs%20distal%20to%20the%20transition%20zone%20%28TZ%29%2C%20suggesting%20conserved%20molecular%20mechanisms.%20The%20TZ%20module%20proteins%20%28Meckel%20Gruber%20syndrome%20%5BMKS%5D%5C%2FNephronophtysis%20%5BNPHP%5D%5C%2FCentrosomal%20protein%20of%20290%20kDa%20%5BCEP290%5D%5C%2FRetinitis%20pigmentosa%20GTPase%20regulator-Interacting%20Protein%201-Like%20Protein%20%5BRPGRIP1L%5D%29%20are%20known%20to%20cooperate%20to%20establish%20TZ%20formation%20and%20function.%20To%20determine%20whether%20they%20control%20deciliation%2C%20we%20studied%20the%20function%20of%205%20of%20them%20%28Transmembrane%20protein%20107%20%5BTMEM107%5D%2C%20Transmembrane%20protein%20216%20%5BTMEM216%5D%2C%20CEP290%2C%20RPGRIP1L%2C%20and%20NPHP4%29%20in%20Paramecium.%20All%20proteins%20are%20recruited%20to%20the%20TZ%20of%20growing%20cilia%20and%20localize%20with%209-fold%20symmetry%20at%20the%20level%20of%20the%20most%20distal%20part%20of%20the%20TZ.%20We%20demonstrate%20that%20depletion%20of%20the%20MKS2%5C%2FTMEM216%20and%20TMEM107%20proteins%20induces%20constant%20deciliation%20of%20some%20cilia%2C%20while%20depletion%20of%20either%20NPHP4%2C%20CEP290%2C%20or%20RPGRIP1L%20prevents%20Ca2%2B%5C%2FEtOH%20deciliation.%20Our%20results%20constitute%20the%20first%20evidence%20for%20a%20role%20of%20conserved%20TZ%20proteins%20in%20deciliation%20and%20open%20new%20directions%20for%20understanding%20motile%20cilia%20physiology.%22%2C%22date%22%3A%222020-03-12%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.1371%5C%2Fjournal.pbio.3000640%22%2C%22ISSN%22%3A%221545-7885%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%2C%222FBUFWW8%22%5D%2C%22dateModified%22%3A%222022-09-12T07%3A02%3A09Z%22%7D%7D%2C%7B%22key%22%3A%22WEGCXB6G%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Le%20Guennec%20et%20al.%22%2C%22parsedDate%22%3A%222020-02-14%22%2C%22numChildren%22%3A5%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3ELe%20Guennec%2C%20Maeva%2C%20Nikolai%20Klena%2C%20Davide%20Gambarotto%2C%20Marine%20H.%20Laporte%2C%20Anne-Marie%20Tassin%2C%20Hugo%20van%20den%20Hoek%2C%20Philipp%20S.%20Erdmann%2C%20et%20al.%202020.%20%26%23x201C%3BA%20Helical%20Inner%20Scaffold%20Provides%20a%20Structural%20Basis%20for%20Centriole%20Cohesion.%26%23x201D%3B%20%3Ci%3EScience%20Advances%3C%5C%2Fi%3E%206%20%287%29%3A%20eaaz4137.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1126%5C%2Fsciadv.aaz4137%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1126%5C%2Fsciadv.aaz4137%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22A%20helical%20inner%20scaffold%20provides%20a%20structural%20basis%20for%20centriole%20cohesion%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Maeva%22%2C%22lastName%22%3A%22Le%20Guennec%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Nikolai%22%2C%22lastName%22%3A%22Klena%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Davide%22%2C%22lastName%22%3A%22Gambarotto%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Marine%20H.%22%2C%22lastName%22%3A%22Laporte%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Hugo%22%2C%22lastName%22%3A%22van%20den%20Hoek%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Philipp%20S.%22%2C%22lastName%22%3A%22Erdmann%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Miroslava%22%2C%22lastName%22%3A%22Schaffer%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lubomir%22%2C%22lastName%22%3A%22Kovacik%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Susanne%22%2C%22lastName%22%3A%22Borgers%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Kenneth%20N.%22%2C%22lastName%22%3A%22Goldie%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Henning%22%2C%22lastName%22%3A%22Stahlberg%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Bornens%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Juliette%22%2C%22lastName%22%3A%22Azimzadeh%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Benjamin%20D.%22%2C%22lastName%22%3A%22Engel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Virginie%22%2C%22lastName%22%3A%22Hamel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Paul%22%2C%22lastName%22%3A%22Guichard%22%7D%5D%2C%22abstractNote%22%3A%22The%20ninefold%20radial%20arrangement%20of%20microtubule%20triplets%20%28MTTs%29%20is%20the%20hallmark%20of%20the%20centriole%2C%20a%20conserved%20organelle%20crucial%20for%20the%20formation%20of%20centrosomes%20and%20cilia.%20Although%20strong%20cohesion%20between%20MTTs%20is%20critical%20to%20resist%20forces%20applied%20by%20ciliary%20beating%20and%20the%20mitotic%20spindle%2C%20how%20the%20centriole%20maintains%20its%20structural%20integrity%20is%20not%20known.%20Using%20cryo-electron%20tomography%20and%20subtomogram%20averaging%20of%20centrioles%20from%20four%20evolutionarily%20distant%20species%2C%20we%20found%20that%20MTTs%20are%20bound%20together%20by%20a%20helical%20inner%20scaffold%20covering%20~70%25%20of%20the%20centriole%20length%20that%20maintains%20MTTs%20cohesion%20under%20compressive%20forces.%20Ultrastructure%20Expansion%20Microscopy%20%28U-ExM%29%20indicated%20that%20POC5%2C%20POC1B%2C%20FAM161A%2C%20and%20Centrin-2%20localize%20to%20the%20scaffold%20structure%20along%20the%20inner%20wall%20of%20the%20centriole%20MTTs.%20Moreover%2C%20we%20established%20that%20these%20four%20proteins%20interact%20with%20each%20other%20to%20form%20a%20complex%20that%20binds%20microtubules.%20Together%2C%20our%20results%20provide%20a%20structural%20and%20molecular%20basis%20for%20centriole%20cohesion%20and%20geometry.%22%2C%22date%22%3A%222020-02-14%22%2C%22language%22%3A%22en%22%2C%22DOI%22%3A%2210.1126%5C%2Fsciadv.aaz4137%22%2C%22ISSN%22%3A%222375-2548%22%2C%22url%22%3A%22https%3A%5C%2F%5C%2Fwww.ncbi.nlm.nih.gov%5C%2Fpmc%5C%2Farticles%5C%2FPMC7021493%5C%2F%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222022-09-12T06%3A59%3A21Z%22%7D%7D%2C%7B%22key%22%3A%22SEUFRLG4%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Thomas%20et%20al.%22%2C%22parsedDate%22%3A%222020-02-06%22%2C%22numChildren%22%3A1%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EThomas%2C%20Lucie%2C%20Khaled%20Bouhouche%2C%20Marjorie%20Whitfield%2C%20Guillaume%20Thouvenin%2C%20Andre%20Coste%2C%20Bruno%20Louis%2C%20Claire%20Szymanski%2C%20et%20al.%202020.%20%26%23x201C%3BTTC12%20Loss-of-Function%20Mutations%20Cause%20Primary%20Ciliary%20Dyskinesia%20and%20Unveil%20Distinct%20Dynein%20Assembly%20Mechanisms%20in%20Motile%20Cilia%20Versus%20Flagella.%26%23x201D%3B%20%3Ci%3EAmerican%20Journal%20of%20Human%20Genetics%3C%5C%2Fi%3E%20106%20%282%29%3A%20153%26%23x2013%3B69.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.ajhg.2019.12.010%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.ajhg.2019.12.010%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22TTC12%20Loss-of-Function%20Mutations%20Cause%20Primary%20Ciliary%20Dyskinesia%20and%20Unveil%20Distinct%20Dynein%20Assembly%20Mechanisms%20in%20Motile%20Cilia%20Versus%20Flagella%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lucie%22%2C%22lastName%22%3A%22Thomas%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Khaled%22%2C%22lastName%22%3A%22Bouhouche%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Marjorie%22%2C%22lastName%22%3A%22Whitfield%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Guillaume%22%2C%22lastName%22%3A%22Thouvenin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Andre%22%2C%22lastName%22%3A%22Coste%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Bruno%22%2C%22lastName%22%3A%22Louis%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Claire%22%2C%22lastName%22%3A%22Szymanski%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Emilie%22%2C%22lastName%22%3A%22Bequignon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jean-Fran%5Cu00e7ois%22%2C%22lastName%22%3A%22Papon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Manon%22%2C%22lastName%22%3A%22Castelli%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Xavier%22%2C%22lastName%22%3A%22Dhalluin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Val%5Cu00e9rie%22%2C%22lastName%22%3A%22Drouin-Garraud%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Guy%22%2C%22lastName%22%3A%22Montantin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Sylvie%22%2C%22lastName%22%3A%22Tissier%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Philippe%22%2C%22lastName%22%3A%22Duquesnoy%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Bruno%22%2C%22lastName%22%3A%22Copin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Florence%22%2C%22lastName%22%3A%22Dastot%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Sandrine%22%2C%22lastName%22%3A%22Couvet%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Laure%22%2C%22lastName%22%3A%22Barbotin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Catherine%22%2C%22lastName%22%3A%22Faucon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Isabelle%22%2C%22lastName%22%3A%22Honore%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Bernard%22%2C%22lastName%22%3A%22Maitre%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Nicole%22%2C%22lastName%22%3A%22Beydon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Aline%22%2C%22lastName%22%3A%22Tamalet%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Nathalie%22%2C%22lastName%22%3A%22Rives%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Estelle%22%2C%22lastName%22%3A%22Escudier%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Aminata%22%2C%22lastName%22%3A%22Tour%5Cu00e9%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Val%5Cu00e9rie%22%2C%22lastName%22%3A%22Mitchell%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Serge%22%2C%22lastName%22%3A%22Amselem%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Marie%22%2C%22lastName%22%3A%22Legendre%22%7D%5D%2C%22abstractNote%22%3A%22Cilia%20and%20flagella%20are%20evolutionarily%20conserved%20organelles%20whose%20motility%20relies%20on%20the%20outer%20and%20inner%20dynein%20arm%20complexes%20%28ODAs%20and%20IDAs%29.%20Defects%20in%20ODAs%20and%20IDAs%20result%20in%20primary%20ciliary%20dyskinesia%20%28PCD%29%2C%20a%20disease%20characterized%20by%20recurrent%20airway%20infections%20and%20male%20infertility.%20PCD%20mutations%20in%20assembly%20factors%20have%20been%20shown%20to%20cause%20a%20combined%20ODA-IDA%20defect%2C%20affecting%20both%20cilia%20and%20flagella.%20We%20identified%20four%20loss-of-function%20mutations%20in%20TTC12%2C%20which%20encodes%20a%20cytoplasmic%20protein%2C%20in%20four%20independent%20families%20in%20which%20affected%20individuals%20displayed%20a%20peculiar%20PCD%20phenotype%20characterized%20by%20the%20absence%20of%20ODAs%20and%20IDAs%20in%20sperm%20flagella%2C%20contrasting%20with%20the%20absence%20of%20only%20IDAs%20in%20respiratory%20cilia.%20Analyses%20of%20both%20primary%20cells%20from%20individuals%20carrying%20TTC12%20mutations%20and%20human%20differentiated%20airway%20cells%20invalidated%20for%20TTC12%20by%20a%20CRISPR-Cas9%20approach%20revealed%20an%20IDA%20defect%20restricted%20to%20a%20subset%20of%20single-headed%20IDAs%20that%20are%20different%20in%20flagella%20and%20cilia%2C%20whereas%20TTC12%20depletion%20in%20the%20ciliate%20Paramecium%20tetraurelia%20recapitulated%20the%20sperm%20phenotype.%20Overall%2C%20our%20study%2C%20which%20identifies%20TTC12%20as%20a%20gene%20involved%20in%20PCD%2C%20unveils%20distinct%20dynein%20assembly%20mechanisms%20in%20human%20motile%20cilia%20versus%20flagella.%22%2C%22date%22%3A%222020-02-06%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.1016%5C%2Fj.ajhg.2019.12.010%22%2C%22ISSN%22%3A%221537-6605%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222022-09-12T06%3A58%3A30Z%22%7D%7D%2C%7B%22key%22%3A%22NSZ49XH3%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Carmona%20et%20al.%22%2C%22parsedDate%22%3A%222020-02%22%2C%22numChildren%22%3A0%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3ECarmona%2C%20Bruno%2C%20Carolina%20Camelo%2C%20M.%20Mehraz%2C%20M.%20Lemullois%2C%20David%20C.%20Ferreira%2C%20Sofia%20Nolasco%2C%20M.%20Lince-Faria%2C%20et%20al.%202020.%20%26%23x201C%3BCentrosome%20Positioning%20and%20Development%20of%20Ciliopathies%3A%20Role%20of%20the%20Human%20Centrosomal%20Protein%20TBCCD1.%26%23x201D%3B%20%3Ci%3EMedicine%3C%5C%2Fi%3E%2099%20%289%29.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Centrosome%20positioning%20and%20development%20of%20ciliopathies%3A%20role%20of%20the%20human%20centrosomal%20protein%20TBCCD1%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Bruno%22%2C%22lastName%22%3A%22Carmona%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Carolina%22%2C%22lastName%22%3A%22Camelo%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22M.%22%2C%22lastName%22%3A%22Mehraz%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22M.%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22David%20C.%22%2C%22lastName%22%3A%22Ferreira%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Sofia%22%2C%22lastName%22%3A%22Nolasco%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22M.%22%2C%22lastName%22%3A%22Lince-Faria%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22H.%22%2C%22lastName%22%3A%22Susana%20Marinho%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Monica%22%2C%22lastName%22%3A%22Bettencourt-Dias%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Helena%22%2C%22lastName%22%3A%22Soares%22%7D%5D%2C%22abstractNote%22%3A%22%22%2C%22date%22%3A%22FEB%202020%22%2C%22language%22%3A%22English%22%2C%22DOI%22%3A%22%22%2C%22ISSN%22%3A%220025-7974%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222020-12-16T13%3A52%3A35Z%22%7D%7D%2C%7B%22key%22%3A%222Z23UVZS%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Fassad%20et%20al.%22%2C%22parsedDate%22%3A%222018-12-06%22%2C%22numChildren%22%3A1%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EFassad%2C%20Mahmoud%20R.%2C%20Amelia%20Shoemark%2C%20Marie%20Legendre%2C%20Robert%20A.%20Hirst%2C%20France%20Koll%2C%20Pierrick%20le%20Borgne%2C%20Bruno%20Louis%2C%20et%20al.%202018.%20%26%23x201C%3BMutations%20in%20Outer%20Dynein%20Arm%20Heavy%20Chain%20DNAH9%20Cause%20Motile%20Cilia%20Defects%20and%20Situs%20Inversus.%26%23x201D%3B%20%3Ci%3EAmerican%20Journal%20of%20Human%20Genetics%3C%5C%2Fi%3E%20103%20%286%29%3A%20984%26%23x2013%3B94.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.ajhg.2018.10.016%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.ajhg.2018.10.016%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Mutations%20in%20Outer%20Dynein%20Arm%20Heavy%20Chain%20DNAH9%20Cause%20Motile%20Cilia%20Defects%20and%20Situs%20Inversus%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Mahmoud%20R.%22%2C%22lastName%22%3A%22Fassad%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Amelia%22%2C%22lastName%22%3A%22Shoemark%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Marie%22%2C%22lastName%22%3A%22Legendre%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Robert%20A.%22%2C%22lastName%22%3A%22Hirst%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Pierrick%22%2C%22lastName%22%3A%22le%20Borgne%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Bruno%22%2C%22lastName%22%3A%22Louis%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Farheen%22%2C%22lastName%22%3A%22Daudvohra%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Mitali%20P.%22%2C%22lastName%22%3A%22Patel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lucie%22%2C%22lastName%22%3A%22Thomas%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Mellisa%22%2C%22lastName%22%3A%22Dixon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Thomas%22%2C%22lastName%22%3A%22Burgoyne%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Joseph%22%2C%22lastName%22%3A%22Hayes%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Andrew%20G.%22%2C%22lastName%22%3A%22Nicholson%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Thomas%22%2C%22lastName%22%3A%22Cullup%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lucy%22%2C%22lastName%22%3A%22Jenkins%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Siobhan%20B.%22%2C%22lastName%22%3A%22Carr%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Paul%22%2C%22lastName%22%3A%22Aurora%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne%22%2C%22lastName%22%3A%22Aubusson-Fleury%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jean-Francois%22%2C%22lastName%22%3A%22Papon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Christopher%22%2C%22lastName%22%3A%22O%27Callaghan%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Serge%22%2C%22lastName%22%3A%22Amselem%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Claire%22%2C%22lastName%22%3A%22Hogg%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Estelle%22%2C%22lastName%22%3A%22Escudier%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Hannah%20M.%22%2C%22lastName%22%3A%22Mitchison%22%7D%5D%2C%22abstractNote%22%3A%22Motile%20cilia%20move%20body%20fluids%20and%20gametes%20and%20the%20beating%20of%20cilia%20lining%20the%20airway%20epithelial%20surfaces%20ensures%20that%20they%20are%20kept%20clear%20and%20protected%20from%20inhaled%20pathogens%20and%20consequent%20respiratory%20infections.%20Dynein%20motor%20proteins%20provide%20mechanical%20force%20for%20cilia%20beating.%20Dynein%20mutations%20are%20a%20common%20cause%20of%20primary%20ciliary%20dyskinesia%20%28PCD%29%2C%20an%20inherited%20condition%20characterized%20by%20deficient%20mucociliary%20clearance%20and%20chronic%20respiratory%20disease%20coupled%20with%20laterality%20disturbances%20and%20subfertility.%20Using%20next-generation%20sequencing%2C%20we%20detected%20mutations%20in%20the%20ciliary%20outer%20dynein%20arm%20%28ODA%29%20heavy%20chain%20gene%20DNAH9%20in%20individuals%20from%20PCD%20clinics%20with%20situs%20inversus%20and%20in%20one%20case%20male%20infertility.%20DNAH9%20and%20its%20partner%20heavy%20chain%20DNAH5%20localize%20to%20type%202%20ODAs%20of%20the%20distal%20cilium%20and%20in%20DNAH9-mutated%20nasal%20respiratory%20epithelial%20cilia%20we%20found%20a%20loss%20of%20DNAH9%5C%2FDNAH5-containing%20type%202%20ODAs%20that%20was%20restricted%20to%20the%20distal%20cilia%20region.%20This%20confers%20a%20reduced%20beating%20frequency%20with%20a%20subtle%20beating%20pattern%20defect%20affecting%20the%20motility%20of%20the%20distal%20cilia%20portion.%203D%20electron%20tomography%20ultrastructural%20studies%20confirmed%20regional%20loss%20of%20ODAs%20from%20the%20distal%20cilium%2C%20manifesting%20as%20either%20loss%20of%20whole%20ODA%20or%20partial%20loss%20of%20ODA%20volume.%20Paramecium%20DNAH9%20knockdown%20confirms%20an%20evolutionarily%20conserved%20function%20for%20DNAH9%20in%20cilia%20motility%20and%20ODA%20stability.%20We%20find%20that%20DNAH9%20is%20widely%20expressed%20in%20the%20airways%2C%20despite%20DNAH9%20mutations%20appearing%20to%20confer%20symptoms%20restricted%20to%20the%20upper%20respiratory%20tract.%20In%20summary%2C%20DNAH9%20mutations%20reduce%20cilia%20function%20but%20some%20respiratory%20mucociliary%20clearance%20potential%20may%20be%20retained%2C%20widening%20the%20PCD%20disease%20spectrum.%22%2C%22date%22%3A%22DEC%206%202018%22%2C%22language%22%3A%22English%22%2C%22DOI%22%3A%2210.1016%5C%2Fj.ajhg.2018.10.016%22%2C%22ISSN%22%3A%220002-9297%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222018-12-20T07%3A44%3A27Z%22%7D%7D%2C%7B%22key%22%3A%22PVULK5V2%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Fassad%20et%20al.%22%2C%22parsedDate%22%3A%222018-05-03%22%2C%22numChildren%22%3A1%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EFassad%2C%20Mahmoud%20R.%2C%20Amelia%20Shoemark%2C%20Pierrick%20le%20Borgne%2C%20France%20Koll%2C%20Mitali%20Patel%2C%20Mellisa%20Dixon%2C%20Jane%20Hayward%2C%20et%20al.%202018.%20%26%23x201C%3BC11orf70%20Mutations%20Disrupting%20the%20Intraflagellar%20Transport-Dependent%20Assembly%20of%20Multiple%20Axonemal%20Dyneins%20Cause%20Primary%20Ciliary%20Dyskinesia.%26%23x201D%3B%20%3Ci%3EAmerican%20Journal%20of%20Human%20Genetics%3C%5C%2Fi%3E%20102%20%285%29%3A%20956%26%23x2013%3B72.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.ajhg.2018.03.024%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.ajhg.2018.03.024%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22C11orf70%20Mutations%20Disrupting%20the%20Intraflagellar%20Transport-Dependent%20Assembly%20of%20Multiple%20Axonemal%20Dyneins%20Cause%20Primary%20Ciliary%20Dyskinesia%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Mahmoud%20R.%22%2C%22lastName%22%3A%22Fassad%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Amelia%22%2C%22lastName%22%3A%22Shoemark%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Pierrick%22%2C%22lastName%22%3A%22le%20Borgne%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Mitali%22%2C%22lastName%22%3A%22Patel%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Mellisa%22%2C%22lastName%22%3A%22Dixon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jane%22%2C%22lastName%22%3A%22Hayward%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Charlotte%22%2C%22lastName%22%3A%22Richardson%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Emily%22%2C%22lastName%22%3A%22Frost%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lucy%22%2C%22lastName%22%3A%22Jenkins%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Thomas%22%2C%22lastName%22%3A%22Cullup%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Eddie%20M.%20K.%22%2C%22lastName%22%3A%22Chung%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne%22%2C%22lastName%22%3A%22Aubusson-Fleury%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Claire%22%2C%22lastName%22%3A%22Hogg%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22David%20R.%22%2C%22lastName%22%3A%22Mitchell%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Hannah%20M.%22%2C%22lastName%22%3A%22Mitchison%22%7D%5D%2C%22abstractNote%22%3A%22Primary%20ciliary%20dyskinesia%20%28PCD%29%20is%20a%20genetically%20and%20phenotypically%20heterogeneous%20disorder%20characterized%20by%20destructive%20respiratory%20disease%20and%20laterality%20abnormalities%20due%20to%20randomized%20left-right%20body%20asymmetry.%20PCD%20is%20mostly%20caused%20by%20mutations%20affecting%20the%20core%20axoneme%20structure%20of%20motile%20cilia%20that%20is%20essential%20for%20movement.%20Genes%20that%20cause%20PCD%20when%20mutated%20include%20a%20group%20that%20encode%20proteins%20essential%20for%20the%20assembly%20of%20the%20ciliary%20dynein%20motors%20and%20the%20active%20transport%20process%20that%20delivers%20them%20from%20their%20cytoplasmic%20assembly%20site%20into%20the%20axoneme.%20We%20screened%20a%20cohort%20of%20affected%20individuals%20for%20disease-causing%20mutations%20using%20a%20targeted%20next%20generation%20sequencing%20panel%20and%20identified%20two%20unrelated%20families%20%28three%20affected%20children%29%20with%20mutations%20in%20the%20uncharacterized%20C11orf70%20gene%20%28official%20gene%20name%20CFAP300%29.%20The%20affected%20children%20share%20a%20consistent%20PCD%20phenotype%20from%20early%20life%20with%20laterality%20defects%20and%20immotile%20respiratory%20cilia%20displaying%20combined%20loss%20of%20inner%20and%20outer%20dynein%20arms%20%28IDA%2BODA%29.%20Phylogenetic%20analysis%20shows%20C11orf70%20is%20highly%20conserved%2C%20distributed%20across%20species%20similarly%20to%20proteins%20involved%20in%20the%20intraflagellar%20transport%20%28IFT%29-dependant%20assembly%20of%20axonemal%20dyneins.%20Paramecium%20C11orf70%20RNAi%20knockdown%20led%20to%20combined%20loss%20of%20ciliary%20IDA%2BODA%20with%20reduced%20cilia%20beating%20and%20swim%20velocity.%20Tagged%20C11orf70%20in%20Paramecium%20and%20Chlamydomonas%20localizes%20mainly%20in%20the%20cytoplasm%20with%20a%20small%20amount%20in%20the%20ciliary%20component.%20IFT139%5C%2FTTC21B%20%28IFT-A%20protein%29%20and%20FLA10%20%28IFT%20kinesin%29%20depletion%20experiments%20show%20that%20its%20transport%20within%20cilia%20is%20IFT%20dependent.%20During%20ciliogenesis%2C%20C11orf70%20accumulates%20at%20the%20ciliary%20tips%20in%20a%20similar%20distribution%20to%20the%20IFT-B%20protein%20IFT46.%20In%20summary%2C%20C11orf70%20is%20essential%20for%20assembly%20of%20dynein%20arms%20and%20C11orf70%20mutations%20cause%20defective%20cilia%20motility%20and%20PCD.%22%2C%22date%22%3A%222018-05-03%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.1016%5C%2Fj.ajhg.2018.03.024%22%2C%22ISSN%22%3A%221537-6605%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222022-09-12T07%3A03%3A08Z%22%7D%7D%2C%7B%22key%22%3A%22IZ7I5QZH%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Tr%5Cu00e9pout%20et%20al.%22%2C%22parsedDate%22%3A%222018-04-01%22%2C%22numChildren%22%3A4%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3ETr%26%23xE9%3Bpout%2C%20Sylvain%2C%20Anne-Marie%20Tassin%2C%20Sergio%20Marco%2C%20and%20Philippe%20Bastin.%202018.%20%26%23x201C%3BSTEM%20Tomography%20Analysis%20of%20the%20Trypanosome%20Transition%20Zone.%26%23x201D%3B%20%3Ci%3EJournal%20of%20Structural%20Biology%3C%5C%2Fi%3E%20202%20%281%29%3A%2051%26%23x2013%3B60.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.jsb.2017.12.005%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fj.jsb.2017.12.005%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22STEM%20tomography%20analysis%20of%20the%20trypanosome%20transition%20zone%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Sylvain%22%2C%22lastName%22%3A%22Tr%5Cu00e9pout%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Sergio%22%2C%22lastName%22%3A%22Marco%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Philippe%22%2C%22lastName%22%3A%22Bastin%22%7D%5D%2C%22abstractNote%22%3A%22The%20protist%20Trypanosoma%20brucei%20is%20an%20emerging%20model%20for%20the%20study%20of%20cilia%20and%20flagella.%20Here%2C%20we%20used%20scanning%20transmission%20electron%20microscopy%20%28STEM%29%20tomography%20to%20describe%20the%20structure%20of%20the%20trypanosome%20transition%20zone%20%28TZ%29.%20At%20the%20base%20of%20the%20TZ%2C%20nine%20transition%20fibres%20irradiate%20from%20the%20B%20microtubule%20of%20each%20doublet%20towards%20the%20membrane.%20The%20TZ%20adopts%20a%209%5Cu202f%2B%5Cu202f0%20structure%20throughout%20its%20length%20of%20%5Cu223c300%5Cu202fnm%20and%20its%20lumen%20contains%20an%20electron-dense%20structure.%20The%20proximal%20portion%20of%20the%20TZ%20has%20an%20invariant%20length%20of%20150%5Cu202fnm%20and%20is%20characterised%20by%20a%20collarette%20surrounding%20the%20membrane%20and%20the%20presence%20of%20electron-dense%20material%20between%20the%20membrane%20and%20the%20doublets.%20The%20distal%20portion%20exhibits%20more%20length%20variation%20%28from%2055%20to%20235%5Cu202fnm%29%20and%20contains%20typical%20Y-links.%20STEM%20analysis%20revealed%20a%20more%20complex%20organisation%20of%20the%20Y-links%20compared%20to%20what%20was%20reported%20by%20conventional%20transmission%20electron%20microscopy.%20Observation%20of%20the%20very%20early%20phase%20of%20flagellum%20assembly%20demonstrated%20that%20the%20proximal%20portion%20and%20the%20collarette%20are%20assembled%20early%20during%20construction.%20The%20presence%20of%20the%20flagella%20connector%20that%20maintains%20the%20tip%20of%20the%20new%20flagellum%20to%20the%20side%20of%20the%20old%20was%20confirmed%20and%20additional%20filamentous%20structures%20making%20contact%20with%20the%20membrane%20of%20the%20flagellar%20pocket%20were%20also%20detected.%20The%20structure%20and%20potential%20functions%20of%20the%20TZ%20in%20trypanosomes%20are%20discussed%2C%20as%20well%20as%20its%20mode%20of%20assembly.%22%2C%22date%22%3A%222018-04-01%22%2C%22language%22%3A%22en%22%2C%22DOI%22%3A%2210.1016%5C%2Fj.jsb.2017.12.005%22%2C%22ISSN%22%3A%221047-8477%22%2C%22url%22%3A%22http%3A%5C%2F%5C%2Fwww.sciencedirect.com%5C%2Fscience%5C%2Farticle%5C%2Fpii%5C%2FS1047847717302289%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%5D%2C%22dateModified%22%3A%222022-09-12T07%3A03%3A24Z%22%7D%7D%2C%7B%22key%22%3A%226I7258UK%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Shi%20et%20al.%22%2C%22parsedDate%22%3A%222018%22%2C%22numChildren%22%3A4%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EShi%2C%20Lei%2C%20France%20Koll%2C%20Olivier%20Arnaiz%2C%20and%20Jean%20Cohen.%202018.%20%26%23x201C%3BThe%20Ciliary%20Protein%20IFT57%20in%20the%20Macronucleus%20of%20Paramecium.%26%23x201D%3B%20%3Ci%3EThe%20Journal%20of%20Eukaryotic%20Microbiology%3C%5C%2Fi%3E%2065%20%281%29%3A%2012%26%23x2013%3B27.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1111%5C%2Fjeu.12423%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1111%5C%2Fjeu.12423%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22The%20Ciliary%20Protein%20IFT57%20in%20the%20Macronucleus%20of%20Paramecium%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lei%22%2C%22lastName%22%3A%22Shi%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Olivier%22%2C%22lastName%22%3A%22Arnaiz%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jean%22%2C%22lastName%22%3A%22Cohen%22%7D%5D%2C%22abstractNote%22%3A%22The%20intraflagellar%20transport%20IFT57%20protein%20is%20essential%20for%20ciliary%20growth%20and%20maintenance.%20Also%20known%20as%20HIPPI%2C%20human%20IFT57%20can%20be%20translocated%20to%20the%20nucleus%20via%20a%20molecular%20partner%20of%20the%20Huntingtin%2C%20Hip1%2C%20inducing%20gene%20expression%20changes.%20In%20Paramecium%20tetraurelia%2C%20we%20identified%20four%20IFT57%20genes%20forming%20two%20subfamilies%20IFT57A%5C%2FB%20and%20IFT57C%5C%2FD%20arising%20from%20whole%20genome%20duplications.%20The%20depletion%20of%20proteins%20of%20the%20two%20subfamilies%20induced%20ciliary%20defects%20and%20IFT57A%20and%20IFT57C%20localized%20in%20basal%20bodies%20and%20cilia.%20We%20observed%20that%20IFT57A%2C%20but%20not%20IFT57C%2C%20is%20also%20present%20in%20the%20macronucleus%20and%20able%20to%20traffic%20toward%20the%20developing%20anlage%20during%20autogamy.%20Analysis%20of%20chimeric%20IFT57A-IFT57C-GFP-tagged%20proteins%20allowed%20us%20to%20identify%20a%20region%20of%20IFT57A%20necessary%20for%20nuclear%20localization.%20We%20studied%20the%20localization%20of%20the%20unique%20IFT57%20protein%20of%20Paramecium%20caudatum%2C%20a%20species%2C%20which%20diverged%20from%20P.%5Cu00a0tetraurelia%20before%20the%20whole%20genome%20duplications.%20The%20P.%5Cu00a0caudatumIFT57C%20protein%20was%20excluded%20from%20the%20nucleus.%20We%20also%20analyzed%20whether%20the%20overexpression%20of%20IFT57A%20in%20Paramecium%20could%20affect%20gene%20transcription%20as%20the%20human%20protein%20does%20in%20HeLa%20cells.%20The%20expression%20of%20some%20genes%20was%20indeed%20affected%20by%20overexpression%20of%20IFT57A%2C%20but%20the%20set%20of%20affected%20genes%20poorly%20overlaps%20the%20set%20of%20genes%20affected%20in%20human%20cells.%22%2C%22date%22%3A%222018%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.1111%5C%2Fjeu.12423%22%2C%22ISSN%22%3A%221550-7408%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%2C%2283ZRAMIC%22%2C%222FBUFWW8%22%5D%2C%22dateModified%22%3A%222021-03-16T16%3A00%3A30Z%22%7D%7D%2C%7B%22key%22%3A%225C3RDS3P%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Aubusson-Fleury%20et%20al.%22%2C%22parsedDate%22%3A%222017-04-21%22%2C%22numChildren%22%3A5%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EAubusson-Fleury%2C%20Anne%2C%20Guillaume%20Balavoine%2C%20Michel%20Lemullois%2C%20Khaled%20Bouhouche%2C%20Janine%20Beisson%2C%20and%20France%20Koll.%202017.%20%26%23x201C%3BCentrin%20Diversity%20and%20Basal%20Body%20Patterning%20across%20Evolution%3A%20New%20Insights%20from%20Paramecium.%26%23x201D%3B%20%3Ci%3EBiology%20Open%3C%5C%2Fi%3E%206%20%286%29%3A%20765%26%23x2013%3B76.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1242%5C%2Fbio.024273%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1242%5C%2Fbio.024273%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Centrin%20diversity%20and%20basal%20body%20patterning%20across%20evolution%3A%20new%20insights%20from%20Paramecium%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne%22%2C%22lastName%22%3A%22Aubusson-Fleury%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Guillaume%22%2C%22lastName%22%3A%22Balavoine%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Khaled%22%2C%22lastName%22%3A%22Bouhouche%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Janine%22%2C%22lastName%22%3A%22Beisson%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%5D%2C%22abstractNote%22%3A%22First%20discovered%20in%20unicellular%20eukaryotes%2C%20centrins%20play%20crucial%20roles%20in%20basal%20body%20duplication%20and%20anchoring%20mechanisms.%20While%20the%20evolutionary%20status%20of%20the%20founding%20members%20of%20the%20family%2C%20Centrin2%5C%2FVfl2%20and%20Centrin3%5C%2Fcdc31%20has%20long%20been%20investigated%2C%20the%20evolutionary%20origin%20of%20other%20members%20of%20the%20family%20has%20received%20less%20attention.%20Using%20a%20phylogeny%20of%20ciliate%20centrins%2C%20we%20identify%20two%20other%20centrin%20families%2C%20the%20ciliary%20centrins%20and%20the%20centrins%20present%20in%20the%20contractile%20filaments%20%28ICL%20centrins%29.%20In%20this%20paper%2C%20we%20carry%20on%20the%20functional%20analysis%20of%20still%20not%20well%20known%20centrins%2C%20the%20ICL1e%20subfamily%20identified%20in%20Paramecium%2C%20and%20show%20their%20requirement%20for%20correct%20basal%20body%20anchoring%20through%20interactions%20with%20Centrin2%20and%20Centrin3.%20Using%20Paramecium%20as%20well%20as%20an%20Eukaryote-wide%20sampling%20of%20centrins%20from%20completely%20sequenced%20genomes%2C%20we%20revisited%20the%20evolutionary%20story%20of%20centrins.%20Their%20phylogeny%20shows%20that%20the%20centrins%20associated%20with%20the%20ciliate%20contractile%20filaments%20are%20widespread%20in%20eukaryotic%20lineages%20and%20could%20be%20as%20ancient%20as%20Centrin2%20and%20Centrin3.%22%2C%22date%22%3A%22Apr%2021%2C%202017%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.1242%5C%2Fbio.024273%22%2C%22ISSN%22%3A%22%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%5D%2C%22dateModified%22%3A%222021-02-19T13%3A43%3A51Z%22%7D%7D%2C%7B%22key%22%3A%227PF66SW7%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Bengueddach%20et%20al.%22%2C%22parsedDate%22%3A%222017%22%2C%22numChildren%22%3A2%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EBengueddach%2C%20Hakim%2C%20Michel%20Lemullois%2C%20Anne%20Aubusson-Fleury%2C%20and%20France%20Koll.%202017.%20%26%23x201C%3BBasal%20Body%20Positioning%20and%20Anchoring%20in%20the%20Multiciliated%20Cell%20Paramecium%20Tetraurelia%3A%20Roles%20of%20OFD1%20and%20VFL3.%26%23x201D%3B%20%3Ci%3ECilia%3C%5C%2Fi%3E%206%20%281%29%3A%206.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1186%5C%2Fs13630-017-0050-z%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1186%5C%2Fs13630-017-0050-z%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Basal%20body%20positioning%20and%20anchoring%20in%20the%20multiciliated%20cell%20Paramecium%20tetraurelia%3A%20roles%20of%20OFD1%20and%20VFL3%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Hakim%22%2C%22lastName%22%3A%22Bengueddach%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne%22%2C%22lastName%22%3A%22Aubusson-Fleury%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22France%22%2C%22lastName%22%3A%22Koll%22%7D%5D%2C%22abstractNote%22%3A%22%22%2C%22date%22%3A%2212%5C%2F2017%22%2C%22language%22%3A%22en%22%2C%22DOI%22%3A%2210.1186%5C%2Fs13630-017-0050-z%22%2C%22ISSN%22%3A%222046-2530%22%2C%22url%22%3A%22http%3A%5C%2F%5C%2Fciliajournal.biomedcentral.com%5C%2Farticles%5C%2F10.1186%5C%2Fs13630-017-0050-z%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%5D%2C%22dateModified%22%3A%222021-02-19T13%3A42%3A18Z%22%7D%7D%2C%7B%22key%22%3A%22T4K8AFET%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Domingues%20et%20al.%22%2C%22parsedDate%22%3A%222017%22%2C%22numChildren%22%3A1%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EDomingues%2C%20M%26%23xE9%3Blanie%20J.%2C%20Juan%20Martinez-Sanz%2C%20Laura%20Papon%2C%20Lionel%20Larue%2C%20Liliane%20Mouawad%2C%20and%20Jacky%20Bonaventure.%202017.%20%26%23x201C%3BStructure-Based%20Mutational%20Analysis%20of%20ICAT%20Residues%20Mediating%20Negative%20Regulation%20of%20%26%23x3B2%3B-Catenin%20Co-Transcriptional%20Activity.%26%23x201D%3B%20%3Ci%3EPloS%20One%3C%5C%2Fi%3E%2012%20%283%29%3A%20e0172603.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1371%5C%2Fjournal.pone.0172603%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1371%5C%2Fjournal.pone.0172603%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Structure-based%20mutational%20analysis%20of%20ICAT%20residues%20mediating%20negative%20regulation%20of%20%5Cu03b2-catenin%20co-transcriptional%20activity%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22M%5Cu00e9lanie%20J.%22%2C%22lastName%22%3A%22Domingues%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Juan%22%2C%22lastName%22%3A%22Martinez-Sanz%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Laura%22%2C%22lastName%22%3A%22Papon%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Lionel%22%2C%22lastName%22%3A%22Larue%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Liliane%22%2C%22lastName%22%3A%22Mouawad%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jacky%22%2C%22lastName%22%3A%22Bonaventure%22%7D%5D%2C%22abstractNote%22%3A%22ICAT%20%28Inhibitor%20of%20%5Cu03b2-CAtenin%20and%20TCF%29%20is%20a%20small%20acidic%20protein%20that%20negatively%20regulates%20%5Cu03b2-catenin%20co-transcriptional%20activity%20by%20competing%20with%20TCF%5C%2FLEF%20factors%20in%20their%20binding%20to%20%5Cu03b2-catenin%20superhelical%20core.%20In%20melanoma%20cells%2C%20ICAT%20competes%20with%20LEF1%20to%20negatively%20regulate%20the%20M-MITF%20and%20NEDD9%20target%20genes.%20The%20structure%20of%20ICAT%20consists%20of%20two%20domains%3A%20the%203-helix%20bundle%20N-terminal%20domain%20binds%20to%20%5Cu03b2-catenin%20Armadillo%20%28Arm%29%20repeats%2010-12%20and%20the%20C-terminal%20tail%20binds%20to%20Arm%20repeats%205-9.%20To%20elucidate%20the%20structural%20mechanisms%20governing%20ICAT%5C%2F%5Cu03b2-catenin%20interactions%20in%20melanoma%20cells%2C%20three%20ICAT%20residues%20Y15%2C%20K19%20and%20V22%20in%20the%20N-terminal%20domain%2C%20contacting%20hydrophobic%20%5Cu03b2-catenin%20residue%20F660%2C%20were%20mutated%20and%20interaction%20was%20assessed%20by%20immunoprecipitation.%20Despite%20the%20moderate%20hydrophobicity%20of%20the%20contact%2C%20its%20removal%20completely%20abolished%20the%20interaction.%20In%20the%20ICAT%20C-terminal%20tail%20consensus%20sequence%2C%20neutralization%20of%20the%20electrostatic%20interactions%20between%20residues%20D66%2C%20E75%20and%20%5Cu03b2-catenin%20residues%20K435%2C%20K312%2C%20coupled%20to%20deletion%20of%20the%20hydrophobic%20contact%20between%20F71%20and%20%5Cu03b2-catenin%20R386%2C%20markedly%20reduced%2C%20but%20failed%20to%20abolish%20the%20ICAT-mediated%20negative%20regulation%20of%20M-MITF%20and%20NEDD9%20promoters.%20We%20conclude%20that%20in%20melanoma%20cells%2C%20anchoring%20of%20ICAT%20N-terminal%20domain%20to%20%5Cu03b2-catenin%20through%20the%20hook%20made%20by%20residue%20F660%2C%20trapped%20in%20the%20pincers%20formed%20by%20ICAT%20residues%20Y15%20and%20V22%2C%20is%20crucial%20for%20stabilizing%20the%20ICAT%5C%2F%5Cu03b2-catenin%20complex.%20This%20is%20a%20prerequisite%20for%20binding%20of%20the%20consensus%20peptide%20to%20Arm%20repeats%205-9%20and%20competition%20with%20LEF1.%20Differences%20between%20ICAT%20and%20LEF1%20in%20their%20affinity%20for%20%5Cu03b2-catenin%20may%20rely%20on%20the%20absence%20in%20ICAT%20of%20hydrophilic%20residues%20between%20D66%20and%20F71.%22%2C%22date%22%3A%222017%22%2C%22language%22%3A%22eng%22%2C%22DOI%22%3A%2210.1371%5C%2Fjournal.pone.0172603%22%2C%22ISSN%22%3A%221932-6203%22%2C%22url%22%3A%22%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%5D%2C%22dateModified%22%3A%222018-03-22T11%3A43%3A29Z%22%7D%7D%2C%7B%22key%22%3A%22XX6B2BKR%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Tassin%20et%20al.%22%2C%22parsedDate%22%3A%222016-02-08%22%2C%22numChildren%22%3A0%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3ETassin%2C%20Anne-Marie%2C%20Michel%20Lemullois%2C%20and%20Anne%20Aubusson-Fleury.%202016.%20%26%23x201C%3BParamecium%20Tetraurelia%20Basal%20Body%20Structure.%26%23x201D%3B%20%3Ci%3ECilia%3C%5C%2Fi%3E%205%20%281%29.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1186%5C%2Fs13630-016-0026-4%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1186%5C%2Fs13630-016-0026-4%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Paramecium%20tetraurelia%20basal%20body%20structure%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne%22%2C%22lastName%22%3A%22Aubusson-Fleury%22%7D%5D%2C%22abstractNote%22%3A%22%22%2C%22date%22%3A%222016-02-08%22%2C%22language%22%3A%22en%22%2C%22DOI%22%3A%2210.1186%5C%2Fs13630-016-0026-4%22%2C%22ISSN%22%3A%222046-2530%22%2C%22url%22%3A%22http%3A%5C%2F%5C%2Fwww.ciliajournal.com%5C%2Fcontent%5C%2F5%5C%2F1%5C%2F6%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%5D%2C%22dateModified%22%3A%222022-09-12T07%3A02%3A46Z%22%7D%7D%2C%7B%22key%22%3A%22BDSMJ6HC%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Gaume%20et%20al.%22%2C%22parsedDate%22%3A%222015-03-19%22%2C%22numChildren%22%3A0%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EGaume%2C%20Xavier%2C%20Anne-Marie%20Tassin%2C%20Iva%20Ugrinova%2C%20Fabien%20Mongelard%2C%20Karine%20Monier%2C%20and%20Philippe%20Bouvet.%202015.%20%26%23x201C%3BCentrosomal%20Nucleolin%20Is%20Required%20for%20Microtubule%20Network%20Organization.%26%23x201D%3B%20%3Ci%3ECell%20Cycle%3C%5C%2Fi%3E%2014%20%286%29%3A%20902%26%23x2013%3B19.%20%3Ca%20href%3D%27https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1080%5C%2F15384101.2014.1000197%27%3Ehttps%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1080%5C%2F15384101.2014.1000197%3C%5C%2Fa%3E.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22journalArticle%22%2C%22title%22%3A%22Centrosomal%20nucleolin%20is%20required%20for%20microtubule%20network%20organization%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Xavier%22%2C%22lastName%22%3A%22Gaume%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Iva%22%2C%22lastName%22%3A%22Ugrinova%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Fabien%22%2C%22lastName%22%3A%22Mongelard%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Karine%22%2C%22lastName%22%3A%22Monier%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Philippe%22%2C%22lastName%22%3A%22Bouvet%22%7D%5D%2C%22abstractNote%22%3A%22%22%2C%22date%22%3A%222015-03-19%22%2C%22language%22%3A%22en%22%2C%22DOI%22%3A%2210.1080%5C%2F15384101.2014.1000197%22%2C%22ISSN%22%3A%221538-4101%2C%201551-4005%22%2C%22url%22%3A%22http%3A%5C%2F%5C%2Fwww.tandfonline.com%5C%2Fdoi%5C%2Ffull%5C%2F10.1080%5C%2F15384101.2014.1000197%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%5D%2C%22dateModified%22%3A%222018-03-15T12%3A21%3A28Z%22%7D%7D%2C%7B%22key%22%3A%22XGP4H2KR%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Gogendeau%20et%20al.%22%2C%22parsedDate%22%3A%222015%22%2C%22numChildren%22%3A0%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EGogendeau%2C%20Delphine%2C%20Paul%20Guichard%2C%20and%20Anne-Marie%20Tassin.%202015.%20%26%23x201C%3BPurification%20of%20Centrosomes%20from%20Mammalian%20Cell%20Lines.%26%23x201D%3B%20In%20%3Ci%3EMethods%20in%20Cell%20Biology%3C%5C%2Fi%3E%2C%20129%3A171%26%23x2013%3B89.%20Elsevier.%20https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fbs.mcb.2015.03.004.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22bookSection%22%2C%22title%22%3A%22Purification%20of%20centrosomes%20from%20mammalian%20cell%20lines%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Delphine%22%2C%22lastName%22%3A%22Gogendeau%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Paul%22%2C%22lastName%22%3A%22Guichard%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne-Marie%22%2C%22lastName%22%3A%22Tassin%22%7D%5D%2C%22abstractNote%22%3A%22%22%2C%22bookTitle%22%3A%22Methods%20in%20Cell%20Biology%22%2C%22date%22%3A%222015%22%2C%22language%22%3A%22en%22%2C%22ISBN%22%3A%22978-0-12-802449-2%22%2C%22url%22%3A%22http%3A%5C%2F%5C%2Flinkinghub.elsevier.com%5C%2Fretrieve%5C%2Fpii%5C%2FS0091679X1500076X%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%5D%2C%22dateModified%22%3A%222018-03-22T11%3A37%3A02Z%22%7D%7D%2C%7B%22key%22%3A%2247RKUBPH%22%2C%22library%22%3A%7B%22id%22%3A3888256%7D%2C%22meta%22%3A%7B%22creatorSummary%22%3A%22Aubusson-Fleury%20et%20al.%22%2C%22parsedDate%22%3A%222015%22%2C%22numChildren%22%3A0%7D%2C%22bib%22%3A%22%3Cdiv%20class%3D%5C%22csl-bib-body%5C%22%20style%3D%5C%22line-height%3A%201.35%3B%20padding-left%3A%201em%3B%20text-indent%3A-1em%3B%5C%22%3E%5Cn%20%20%3Cdiv%20class%3D%5C%22csl-entry%5C%22%3EAubusson-Fleury%2C%20Anne%2C%20Jean%20Cohen%2C%20and%20Michel%20Lemullois.%202015.%20%26%23x201C%3BCiliary%20Heterogeneity%20within%20a%20Single%20Cell%3A%20The%20Paramecium%20Model.%26%23x201D%3B%20In%20%3Ci%3EMethods%20in%20Cell%20Biology%3C%5C%2Fi%3E%2C%20127%3A457%26%23x2013%3B85.%20Elsevier.%20https%3A%5C%2F%5C%2Fdoi.org%5C%2F10.1016%5C%2Fbs.mcb.2014.12.007.%3C%5C%2Fdiv%3E%5Cn%3C%5C%2Fdiv%3E%22%2C%22data%22%3A%7B%22itemType%22%3A%22bookSection%22%2C%22title%22%3A%22Ciliary%20heterogeneity%20within%20a%20single%20cell%3A%20The%20Paramecium%20model%22%2C%22creators%22%3A%5B%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Anne%22%2C%22lastName%22%3A%22Aubusson-Fleury%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Jean%22%2C%22lastName%22%3A%22Cohen%22%7D%2C%7B%22creatorType%22%3A%22author%22%2C%22firstName%22%3A%22Michel%22%2C%22lastName%22%3A%22Lemullois%22%7D%5D%2C%22abstractNote%22%3A%22%22%2C%22bookTitle%22%3A%22Methods%20in%20Cell%20Biology%22%2C%22date%22%3A%222015%22%2C%22language%22%3A%22en%22%2C%22ISBN%22%3A%22978-0-12-802451-5%22%2C%22url%22%3A%22http%3A%5C%2F%5C%2Flinkinghub.elsevier.com%5C%2Fretrieve%5C%2Fpii%5C%2FS0091679X14000521%22%2C%22collections%22%3A%5B%22R7I3GKDL%22%5D%2C%22dateModified%22%3A%222018-03-15T12%3A22%3A04Z%22%7D%7D%5D%7D
Carvalhal, Sara, Bruno Carmona, Anne-Marie Tassin, and João Gonçalves. 2024. “Editorial: Molecular Mechanisms of Cilia Related Diseases.” Frontiers in Molecular Biosciences 11:1421419. https://doi.org/10.3389/fmolb.2024.1421419.
Bodin, Alexia, Logan Greibill, Julien Gouju, Franck Letournel, Silvia Pozzi, Jean-Pierre Julien, Laurence Renaud, et al. 2023. “Transactive Response DNA-Binding Protein 43 Is Enriched at the Centrosome in Human Cells.” Brain: A Journal of Neurology, July, awad228. https://doi.org/10.1093/brain/awad228.
Borgne, Pierrick Le, Logan Greibill, Marine Hélène Laporte, Michel Lemullois, Khaled Bouhouche, Mebarek Temagoult, Olivier Rosnet, et al. 2022. “The Evolutionary Conserved Proteins CEP90, FOPNL, and OFD1 Recruit Centriolar Distal Appendage Proteins to Initiate Their Assembly.” PLOS Biology 20 (9): e3001782. https://doi.org/10.1371/journal.pbio.3001782.
Bouhouche, K., M. S. Valentine, P. Le Borgne, M. Lemullois, J. Yano, S. Lodh, A. Nabi, A. M. Tassin, and J. L. Van Houten. 2022. “Paramecium, a Model to Study Ciliary Beating and Ciliogenesis: Insights From Cutting-Edge Approaches.” Frontiers in Cell and Developmental Biology 10 (April). https://www.frontiersin.org/article/10.3389/fcell.2022.847908.
Klena, Nikolai, Maeva Le Guennec, Anne-Marie Tassin, Hugo van den Hoek, Philipp S Erdmann, Miroslava Schaffer, Stefan Geimer, et al. 2020. “Architecture of the Centriole Cartwheel-Containing Region Revealed by Cryo-Electron Tomography.” The EMBO Journal 39 (22): e106246. https://doi.org/10.15252/embj.2020106246.
Steib, Emmanuelle, Marine H. Laporte, Davide Gambarotto, Natacha Olieric, Celine Zheng, Susanne Borgers, Vincent Olieric, et al. 2020. “WDR90 Is a Centriolar Microtubule Wall Protein Important for Centriole Architecture Integrity.” Elife 9 (September):e57205. https://doi.org/10.7554/eLife.57205.
Gogendeau, Delphine, Michel Lemullois, Pierrick Le Borgne, Manon Castelli, Anne Aubusson-Fleury, Olivier Arnaiz, Jean Cohen, et al. 2020. “MKS-NPHP Module Proteins Control Ciliary Shedding at the Transition Zone.” PLoS Biology 18 (3): e3000640. https://doi.org/10.1371/journal.pbio.3000640.
Le Guennec, Maeva, Nikolai Klena, Davide Gambarotto, Marine H. Laporte, Anne-Marie Tassin, Hugo van den Hoek, Philipp S. Erdmann, et al. 2020. “A Helical Inner Scaffold Provides a Structural Basis for Centriole Cohesion.” Science Advances 6 (7): eaaz4137. https://doi.org/10.1126/sciadv.aaz4137.
Thomas, Lucie, Khaled Bouhouche, Marjorie Whitfield, Guillaume Thouvenin, Andre Coste, Bruno Louis, Claire Szymanski, et al. 2020. “TTC12 Loss-of-Function Mutations Cause Primary Ciliary Dyskinesia and Unveil Distinct Dynein Assembly Mechanisms in Motile Cilia Versus Flagella.” American Journal of Human Genetics 106 (2): 153–69. https://doi.org/10.1016/j.ajhg.2019.12.010.
Carmona, Bruno, Carolina Camelo, M. Mehraz, M. Lemullois, David C. Ferreira, Sofia Nolasco, M. Lince-Faria, et al. 2020. “Centrosome Positioning and Development of Ciliopathies: Role of the Human Centrosomal Protein TBCCD1.” Medicine 99 (9).
Fassad, Mahmoud R., Amelia Shoemark, Marie Legendre, Robert A. Hirst, France Koll, Pierrick le Borgne, Bruno Louis, et al. 2018. “Mutations in Outer Dynein Arm Heavy Chain DNAH9 Cause Motile Cilia Defects and Situs Inversus.” American Journal of Human Genetics 103 (6): 984–94. https://doi.org/10.1016/j.ajhg.2018.10.016.
Fassad, Mahmoud R., Amelia Shoemark, Pierrick le Borgne, France Koll, Mitali Patel, Mellisa Dixon, Jane Hayward, et al. 2018. “C11orf70 Mutations Disrupting the Intraflagellar Transport-Dependent Assembly of Multiple Axonemal Dyneins Cause Primary Ciliary Dyskinesia.” American Journal of Human Genetics 102 (5): 956–72. https://doi.org/10.1016/j.ajhg.2018.03.024.
Trépout, Sylvain, Anne-Marie Tassin, Sergio Marco, and Philippe Bastin. 2018. “STEM Tomography Analysis of the Trypanosome Transition Zone.” Journal of Structural Biology 202 (1): 51–60. https://doi.org/10.1016/j.jsb.2017.12.005.
Shi, Lei, France Koll, Olivier Arnaiz, and Jean Cohen. 2018. “The Ciliary Protein IFT57 in the Macronucleus of Paramecium.” The Journal of Eukaryotic Microbiology 65 (1): 12–27. https://doi.org/10.1111/jeu.12423.
Aubusson-Fleury, Anne, Guillaume Balavoine, Michel Lemullois, Khaled Bouhouche, Janine Beisson, and France Koll. 2017. “Centrin Diversity and Basal Body Patterning across Evolution: New Insights from Paramecium.” Biology Open 6 (6): 765–76. https://doi.org/10.1242/bio.024273.
Bengueddach, Hakim, Michel Lemullois, Anne Aubusson-Fleury, and France Koll. 2017. “Basal Body Positioning and Anchoring in the Multiciliated Cell Paramecium Tetraurelia: Roles of OFD1 and VFL3.” Cilia 6 (1): 6. https://doi.org/10.1186/s13630-017-0050-z.
Domingues, Mélanie J., Juan Martinez-Sanz, Laura Papon, Lionel Larue, Liliane Mouawad, and Jacky Bonaventure. 2017. “Structure-Based Mutational Analysis of ICAT Residues Mediating Negative Regulation of β-Catenin Co-Transcriptional Activity.” PloS One 12 (3): e0172603. https://doi.org/10.1371/journal.pone.0172603.
Tassin, Anne-Marie, Michel Lemullois, and Anne Aubusson-Fleury. 2016. “Paramecium Tetraurelia Basal Body Structure.” Cilia 5 (1). https://doi.org/10.1186/s13630-016-0026-4.
Gaume, Xavier, Anne-Marie Tassin, Iva Ugrinova, Fabien Mongelard, Karine Monier, and Philippe Bouvet. 2015. “Centrosomal Nucleolin Is Required for Microtubule Network Organization.” Cell Cycle 14 (6): 902–19. https://doi.org/10.1080/15384101.2014.1000197.
Gogendeau, Delphine, Paul Guichard, and Anne-Marie Tassin. 2015. “Purification of Centrosomes from Mammalian Cell Lines.” In Methods in Cell Biology, 129:171–89. Elsevier. https://doi.org/10.1016/bs.mcb.2015.03.004.
Aubusson-Fleury, Anne, Jean Cohen, and Michel Lemullois. 2015. “Ciliary Heterogeneity within a Single Cell: The Paramecium Model.” In Methods in Cell Biology, 127:457–85. Elsevier. https://doi.org/10.1016/bs.mcb.2014.12.007.