ID   DRC2_DANRE              Reviewed;         492 AA.
AC   Q6DHI2; Q7ZZA2;
DT   17-APR-2007, integrated into UniProtKB/Swiss-Prot.
DT   17-APR-2007, sequence version 2.
DT   03-AUG-2022, entry version 86.
DE   RecName: Full=Dynein regulatory complex subunit 2 {ECO:0000303|PubMed:24094744};
DE   AltName: Full=Coiled-coil domain-containing protein 65;
GN   Name=ccdc65; Synonyms=drc2 {ECO:0000303|PubMed:24094744};
GN   ORFNames=si:dz202l16.3;
OS   Danio rerio (Zebrafish) (Brachydanio rerio).
OC   Eukaryota; Metazoa; Chordata; Craniata; Vertebrata; Euteleostomi;
OC   Actinopterygii; Neopterygii; Teleostei; Ostariophysi; Cypriniformes;
OC   Danionidae; Danioninae; Danio.
OX   NCBI_TaxID=7955;
RN   [1]
RP   NUCLEOTIDE SEQUENCE [LARGE SCALE GENOMIC DNA].
RC   STRAIN=Tuebingen;
RX   PubMed=23594743; DOI=10.1038/nature12111;
RA   Howe K., Clark M.D., Torroja C.F., Torrance J., Berthelot C., Muffato M.,
RA   Collins J.E., Humphray S., McLaren K., Matthews L., McLaren S., Sealy I.,
RA   Caccamo M., Churcher C., Scott C., Barrett J.C., Koch R., Rauch G.J.,
RA   White S., Chow W., Kilian B., Quintais L.T., Guerra-Assuncao J.A., Zhou Y.,
RA   Gu Y., Yen J., Vogel J.H., Eyre T., Redmond S., Banerjee R., Chi J., Fu B.,
RA   Langley E., Maguire S.F., Laird G.K., Lloyd D., Kenyon E., Donaldson S.,
RA   Sehra H., Almeida-King J., Loveland J., Trevanion S., Jones M., Quail M.,
RA   Willey D., Hunt A., Burton J., Sims S., McLay K., Plumb B., Davis J.,
RA   Clee C., Oliver K., Clark R., Riddle C., Elliot D., Threadgold G.,
RA   Harden G., Ware D., Begum S., Mortimore B., Kerry G., Heath P.,
RA   Phillimore B., Tracey A., Corby N., Dunn M., Johnson C., Wood J., Clark S.,
RA   Pelan S., Griffiths G., Smith M., Glithero R., Howden P., Barker N.,
RA   Lloyd C., Stevens C., Harley J., Holt K., Panagiotidis G., Lovell J.,
RA   Beasley H., Henderson C., Gordon D., Auger K., Wright D., Collins J.,
RA   Raisen C., Dyer L., Leung K., Robertson L., Ambridge K., Leongamornlert D.,
RA   McGuire S., Gilderthorp R., Griffiths C., Manthravadi D., Nichol S.,
RA   Barker G., Whitehead S., Kay M., Brown J., Murnane C., Gray E.,
RA   Humphries M., Sycamore N., Barker D., Saunders D., Wallis J., Babbage A.,
RA   Hammond S., Mashreghi-Mohammadi M., Barr L., Martin S., Wray P.,
RA   Ellington A., Matthews N., Ellwood M., Woodmansey R., Clark G., Cooper J.,
RA   Tromans A., Grafham D., Skuce C., Pandian R., Andrews R., Harrison E.,
RA   Kimberley A., Garnett J., Fosker N., Hall R., Garner P., Kelly D., Bird C.,
RA   Palmer S., Gehring I., Berger A., Dooley C.M., Ersan-Urun Z., Eser C.,
RA   Geiger H., Geisler M., Karotki L., Kirn A., Konantz J., Konantz M.,
RA   Oberlander M., Rudolph-Geiger S., Teucke M., Lanz C., Raddatz G.,
RA   Osoegawa K., Zhu B., Rapp A., Widaa S., Langford C., Yang F.,
RA   Schuster S.C., Carter N.P., Harrow J., Ning Z., Herrero J., Searle S.M.,
RA   Enright A., Geisler R., Plasterk R.H., Lee C., Westerfield M.,
RA   de Jong P.J., Zon L.I., Postlethwait J.H., Nusslein-Volhard C.,
RA   Hubbard T.J., Roest Crollius H., Rogers J., Stemple D.L.;
RT   "The zebrafish reference genome sequence and its relationship to the human
RT   genome.";
RL   Nature 496:498-503(2013).
RN   [2]
RP   NUCLEOTIDE SEQUENCE [LARGE SCALE MRNA].
RG   NIH - Zebrafish Gene Collection (ZGC) project;
RL   Submitted (JUL-2004) to the EMBL/GenBank/DDBJ databases.
RN   [3]
RP   FUNCTION, AND DISRUPTION PHENOTYPE.
RX   PubMed=24094744; DOI=10.1016/j.ajhg.2013.08.015;
RA   Austin-Tse C., Halbritter J., Zariwala M.A., Gilberti R.M., Gee H.Y.,
RA   Hellman N., Pathak N., Liu Y., Panizzi J.R., Patel-King R.S.,
RA   Tritschler D., Bower R., O'Toole E., Porath J.D., Hurd T.W., Chaki M.,
RA   Diaz K.A., Kohl S., Lovric S., Hwang D.Y., Braun D.A., Schueler M.,
RA   Airik R., Otto E.A., Leigh M.W., Noone P.G., Carson J.L., Davis S.D.,
RA   Pittman J.E., Ferkol T.W., Atkinson J.J., Olivier K.N., Sagel S.D.,
RA   Dell S.D., Rosenfeld M., Milla C.E., Loges N.T., Omran H., Porter M.E.,
RA   King S.M., Knowles M.R., Drummond I.A., Hildebrandt F.;
RT   "Zebrafish ciliopathy screen plus human mutational analysis identifies
RT   C21orf59 and CCDC65 defects as causing primary ciliary dyskinesia.";
RL   Am. J. Hum. Genet. 93:672-686(2013).
CC   -!- FUNCTION: Component of the nexin-dynein regulatory complex (N-DRC), a
CC       key regulator of ciliary/flagellar motility which maintains the
CC       alignment and integrity of the distal axoneme and regulates microtubule
CC       sliding in motile axonemes (By similarity). Plays a critical role in
CC       the assembly of N-DRC and also stabilizes the assembly of multiple
CC       inner dynein arms and radial spokes. Coassembles with DRC1 to form a
CC       central scaffold needed for assembly of the N-DRC and its attachment to
CC       the outer doublet microtubules. {ECO:0000250|UniProtKB:A8JB22,
CC       ECO:0000269|PubMed:24094744}.
CC   -!- SUBUNIT: Component of the nexin-dynein regulatory complex (N-DRC).
CC       {ECO:0000250|UniProtKB:A8JB22}.
CC   -!- SUBCELLULAR LOCATION: Cytoplasm, cytoskeleton, flagellum basal body
CC       {ECO:0000250|UniProtKB:A8JB22}. Cell projection, cilium, flagellum
CC       {ECO:0000250|UniProtKB:A8JB22}. Cytoplasm, cytoskeleton, flagellum
CC       axoneme {ECO:0000250|UniProtKB:A8JB22}.
CC   -!- DISRUPTION PHENOTYPE: Morpholino knockdown of the protein causes strong
CC       ciliopathy phenotypes, including pronephric cysts, axis curvature,
CC       left-right asymmetry defects and hydrocephalus. Cilia length and number
CC       appear normal, but cilia are dyskinetic. {ECO:0000269|PubMed:24094744}.
CC   -!- SIMILARITY: Belongs to the DRC2 family.
CC   -!- SEQUENCE CAUTION:
CC       Sequence=CAD60659.1; Type=Erroneous gene model prediction; Evidence={ECO:0000305};
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DR   EMBL; AL591389; CAD60659.1; ALT_SEQ; Genomic_DNA.
DR   EMBL; BC075992; AAH75992.1; -; mRNA.
DR   RefSeq; NP_001002603.1; NM_001002603.2.
DR   RefSeq; XP_005165756.1; XM_005165699.2.
DR   AlphaFoldDB; Q6DHI2; -.
DR   SMR; Q6DHI2; -.
DR   STRING; 7955.ENSDARP00000043945; -.
DR   PaxDb; Q6DHI2; -.
DR   PRIDE; Q6DHI2; -.
DR   Ensembl; ENSDART00000043946; ENSDARP00000043945; ENSDARG00000032005.
DR   Ensembl; ENSDART00000177219; ENSDARP00000144012; ENSDARG00000115576.
DR   GeneID; 368824; -.
DR   KEGG; dre:368824; -.
DR   CTD; 85478; -.
DR   ZFIN; ZDB-GENE-030616-358; ccdc65.
DR   eggNOG; ENOG502QQDD; Eukaryota.
DR   GeneTree; ENSGT00940000153804; -.
DR   HOGENOM; CLU_026536_1_0_1; -.
DR   InParanoid; Q6DHI2; -.
DR   OMA; QNHERDV; -.
DR   OrthoDB; 815240at2759; -.
DR   PhylomeDB; Q6DHI2; -.
DR   TreeFam; TF326074; -.
DR   PRO; PR:Q6DHI2; -.
DR   Proteomes; UP000000437; Genome assembly.
DR   Proteomes; UP000814640; Chromosome 6.
DR   Bgee; ENSDARG00000032005; Expressed in testis and 11 other tissues.
DR   GO; GO:0005858; C:axonemal dynein complex; IEA:InterPro.
DR   GO; GO:0005930; C:axoneme; IBA:GO_Central.
DR   GO; GO:0031514; C:motile cilium; IEA:UniProtKB-SubCell.
DR   GO; GO:0070286; P:axonemal dynein complex assembly; IBA:GO_Central.
DR   GO; GO:0060285; P:cilium-dependent cell motility; IBA:GO_Central.
DR   GO; GO:0003352; P:regulation of cilium movement; IMP:ZFIN.
DR   InterPro; IPR039505; DRC1/2_N.
DR   InterPro; IPR039750; DRC1/DRC2.
DR   PANTHER; PTHR21625; PTHR21625; 1.
DR   Pfam; PF14772; NYD-SP28; 1.
PE   2: Evidence at transcript level;
KW   Cell projection; Cilium; Coiled coil; Cytoplasm; Cytoskeleton; Flagellum;
KW   Reference proteome.
FT   CHAIN           1..492
FT                   /note="Dynein regulatory complex subunit 2"
FT                   /id="PRO_0000284782"
FT   COILED          16..95
FT                   /evidence="ECO:0000255"
FT   COILED          256..318
FT                   /evidence="ECO:0000255"
FT   COILED          373..401
FT                   /evidence="ECO:0000255"
FT   CONFLICT        9
FT                   /note="A -> G (in Ref. 2; AAH75992)"
FT                   /evidence="ECO:0000305"
FT   CONFLICT        120
FT                   /note="K -> M (in Ref. 2; AAH75992)"
FT                   /evidence="ECO:0000305"
FT   CONFLICT        151
FT                   /note="V -> E (in Ref. 2; AAH75992)"
FT                   /evidence="ECO:0000305"
FT   CONFLICT        375
FT                   /note="E -> K (in Ref. 2; AAH75992)"
FT                   /evidence="ECO:0000305"
FT   CONFLICT        453
FT                   /note="R -> Q (in Ref. 2; AAH75992)"
FT                   /evidence="ECO:0000305"
SQ   SEQUENCE   492 AA;  57703 MW;  A88FEFFFFDA331DE CRC64;
     MPKKAGKKAG GKLAGLTEEE RLLYMQQKAQ AEEEIAKRKE DMLTHFLKDK LQKEEKNTVL
     NLHKLRQQWR AVLTQTKIAE LRNDMSVLSQ TFERVLDYKD NIIRSLQVDL SERELQSELK
     RSSHLHNVDC LLEIHKSRLA QLEFNFKSSL VELSSEYNTE REQILSQHQQ ECVDLDNVMF
     SMEKHYSDLD GEAKRDYQST RNQIKKRNFD DKQAVKEQMD GVVEKLWQDQ QQVLNHYNES
     TRDKIITTDD LMNKDVQSAK EIDSLKKHIQ KLQDSISTFR GQLSSGQTDK TAEQLRSERD
     ELAQEVQHFR VQLNAAQAIR KKHITKLTVQ SNDATKKLQE IVARGERLLR QCEMCCKLET
     EHEKVLPFYK SSLSEEEQKQ EKAKAMESSN EKLTQLMHDY SPLAKFWQRY NKVELDCLCV
     KREKLLLLQE NERLRLFLKQ YLDEVSVSDE SFRQQKLLVV SSPALQDTAA TDRHQQKRHV
     VQEAACIVQK QL