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1 ells, despite its sequence classification as axonemal.
2 ke polypeptides that have been thought to be axonemal.
3 eract and are hypothesized to function as an axonemal accessory complex, but only ODA5p was previousl
4 between the axonemal cytoskeleton, the peri-axonemal accessory structures, and multiple regulatory n
5 ARAM1 resulted in short cilia with decreased axonemal acetylation and polyglutamylation, but relative
6 s present in cytoplasm and flagella, remains axonemal after detergent treatment, and is extracted wit
7 rate that we have identified the first human axonemal AKAP, a protein that likely plays a role in the
8 ective for specific axonemal structures, two axonemal AKAPs have been identified: a 240-kD AKAP assoc
16 Nineteen specific cytosolic factors (Dynein Axonemal Assembly Factors; DNAAFs) are necessary for axo
17 nto respiratory cilia and cause a failure in axonemal assembly of the ODA component DNAH5 and the ODA
21 s of sup-pf-2-1 axonemes indicates that both axonemal ATPase activity and outer arm polypeptides are
23 the A-tubule, resulting in the deficiency of axonemal beating and hence gamete formation and egress.
25 ding, and may regulate the size and shape of axonemal bends through interactions with the radial spok
29 the BB, and microtubule geometry changes to axonemal by the end of the TZ, followed by the addition
30 is a key regulatory protein for Tetrahymena axonemal Ca(2+) responses, including ciliary reversal or
33 some flagellum and three constituents of the axonemal capping structure at the tips of both assemblin
34 ent cells identified that the absence of the axonemal CCDC39/CCDC40 heterodimer resulted in the loss
35 f GCP2 and GCP3, but not GCP4, disrupted the axonemal central pair microtubules, but not the subpelli
38 mydomonas motility mutants suggests that the axonemal CKI is located on the outer doublet microtubule
40 is an extension of the cell that contains an axonemal complex of microtubules and associated proteins
41 calized hMCA within the radial spokes of the axonemal complex of the sperm flagellum, and immunofluor
42 We propose that Oda5p is part of a novel axonemal complex that is required for outer arm assembly
43 ng physical interactions between these three axonemal complexes and a role for the MIA complex in the
44 emonstrate that the CSC connects three major axonemal complexes involved in dynein regulation: RS2, t
47 demonstrates that the PF6 polypeptide is an axonemal component that cosediments at 12.6S with severa
48 irectly in situ with an approximately 45 kDa axonemal component; this interaction is disrupted by the
49 ously shown to be necessary for transport of axonemal components, is also involved in the motility of
51 e precise localization of mRNAs encoding key axonemal constituents, allowing these proteins to incorp
53 doublet microtubules, acts to stabilize the axonemal core structure, and serves as a central hub for
56 reduced slightly with respect to that of the axonemal cytoplasmic reservoir, allowing cytoplasmic flo
57 e outcome of a dynamic interplay between the axonemal cytoskeleton, the peri-axonemal accessory struc
62 in mediating assembly of both ODAs and their axonemal docking machinery onto ciliary microtubules.
63 model of the repeating structure of a native axonemal doublet microtubule, which reveals the identiti
64 omics to resolve the 96-nm modular repeat of axonemal doublet microtubules (DMTs) from both sperm fla
65 present high-resolution structures of native axonemal doublet microtubules (DMTs) from sea urchin and
69 tigoneme complexes physically connect to the axonemal doublets 4 and 8, positioning them perpendicula
70 Studies of the in vitro binding of ODAs to axonemal doublets reveal a role for the ODA5/ODA10 assem
71 play an essential role in the regulation of axonemal dynein activity and thus of ciliary and flagell
72 The addition of kinase inhibitor restored axonemal dynein activity concomitant with the dephosphor
76 mport into cilia and flagella, multi-subunit axonemal dynein arms are thought to be stabilized and pr
77 unchanged or become elevated, the density of axonemal dynein arms is reduced in reptin(hi2394) mutant
78 cluding the intraflagellar transport system, axonemal dynein arms, radial spokes, the 96-nm ruler, an
81 Assembly Factors; DNAAFs) are necessary for axonemal dynein assembly, although the detailed mechanis
86 bules with MTBDs of cytoplasmic dynein-1 and axonemal dynein DNAH7 and determined their cryo-EM struc
89 Dense populations of microtubules driven by axonemal dynein form large vortices, providing insights
92 allele of the testis-specifically expressed axonemal dynein heavy chain (axDHC) gene, Dnahc8, has be
93 acterized an insertional mutation in a mouse axonemal dynein heavy chain gene (Mdnah5) that reproduce
95 culture demonstrated that the expression of axonemal dynein heavy chains correlated with the develop
96 fication and partial cloning of seven unique axonemal dynein heavy chains from rat tracheal epithelia
98 r specifically required for the stability of axonemal dynein heavy chains in cytoplasm and suggest th
99 gulate the cell-specific expression of these axonemal dynein heavy chains will further our understand
100 level of conservation does not extend to the axonemal dynein heavy chains, suggesting functional diff
101 Here we report the positional cloning of an axonemal dynein heavy-chain gene, left/right-dynein (lrd
102 ity, can influence the activity of outer arm axonemal dynein in motility assays using purified protei
103 Pontin is essential for the stabilization of axonemal dynein intermediate chain 1 (DNAI1) and DNAI2,
107 a novel RNP granule containing the mRNAs for axonemal dynein motor proteins becomes highly polarized
109 noregulatory complexes with their associated axonemal dynein motors provide a mechanism for the long-
111 cle cryo-EM reconstruction of a three-headed axonemal dynein natively bound to doublet microtubules i
116 to a higher rate of binding of Chlamydomonas axonemal dynein to Chlamydomonas microtubules than to po
122 amily proteins were originally identified in axonemal dyneins and subsequently found to function in m
128 ized oda mutants, but only a partial loss of axonemal dyneins as shown by both electron microscopy an
129 oward visualizing the ATPase activity of the axonemal dyneins during bending, we have investigated th
130 arious cellular transport systems, including axonemal dyneins generating the force for ciliary and fl
132 This allows for the incorporation of these axonemal dyneins into the axoneme directly from the cyto
133 tory beating patterns, the activities of the axonemal dyneins must be coordinated both spatially and
137 x specifically localizes the linear array of axonemal dyneins to the doublet microtubule by directly
139 tains an 18-residue insertion, found in many axonemal dyneins, that contacts the adjacent protofilame
140 sity directly influences the activity of the axonemal dyneins, the motors that drive the beating of t
142 ersity can directly regulate the activity of axonemal dyneins, we asked whether in vitro acetylation
150 hich is involved in IFT protein recruitment, axonemal engagement of IFT protein complexes, and so on.
152 ssary and sufficient for basal body docking, axonemal extension, and motility during the differentiat
156 CaM antibodies and Chlamydomonas reinhardtii axonemal extracts, we precipitated a complex that includ
157 identified a novel and distinct set of extra-axonemal flagellar filaments important for adhesion and
159 proteins are found almost exclusively in the axonemal fraction, and the methylated forms of these pro
160 homozygosity for the t allele of Dnahc8, an axonemal gamma-type dynein heavy chain (gammaDHC) gene,
161 eukaryotic flagellum/cilium is important for axonemal growth and signaling and has distinct biomechan
163 (rs2134256 and rs1354187) within the dynein axonemal heavy chain 5 (DNAH5) gene (Pmeta-int = 3.6 x 1
166 e ciliary dilation (CD), a highly structured axonemal inclusion of hitherto unknown composition and f
167 findings identified the essential role of an axonemal inner-arm dynein complex in cell division, and
168 rried out functional analyses of a flagellar axonemal inner-arm dynein complex in the bloodstream for
171 dition to the shared transduction mechanism, axonemal integrity and possibly ciliary motility are req
172 dynein family, consisting of cytoplasmic and axonemal isoforms, are motors that move towards the minu
174 a defect phenocopied by the silencing of the axonemal kinesin subunit KIF3A but not by chemical disru
175 res were quantified when possible, including axonemal length and diameter, microtubule conformations,
178 d/or short primary cilia, as well as reduced axonemal localization of ciliary proteins ARL13B and ade
179 Lechtreck and Witman determined the precise axonemal location of hydin, a protein that, when mutated
182 flagellar compartment and anchored into the axonemal microtubular scaffold via the ODA docking compl
183 ed multimolecular structures attached to the axonemal microtubule doublets and are essential for the
185 a length and shape in part via modulation of axonemal microtubule stability, suggesting that similar
187 ining 66 (CCDC66) and TOG array regulator of axonemal microtubules 1 (TOGARAM1) as ARMC9 interaction
188 ation regulates the assembly and dynamics of axonemal microtubules and acts either directly or indire
190 ermatozoa exhibited defective arrangement of axonemal microtubules and flagella outer dense fibers.
191 isms, and is required for the acetylation of axonemal microtubules and for the normal kinetics of pri
192 ment of multisubunit protein particles along axonemal microtubules and is required for assembly and m
194 ar machine-the axoneme-but it is unclear how axonemal microtubules are ornamented to support motility
196 r proteins and a novel protein organelle use axonemal microtubules as tracks to shuttle essential com
197 activated breakage of the nine outer-doublet axonemal microtubules at a specific site in the flagella
198 Fully assembled radial spokes, detached from axonemal microtubules during flagellar breakdown or turn
199 er centriole into the basal body, from which axonemal microtubules extend to form the ciliary compart
201 the centriole-derived basal body, from which axonemal microtubules grow and which assembles a gate to
206 lotype encoded) and wild-type dyneins to the axonemal microtubules of t-bearing or wild-type sperm, w
207 In addition, APC regulates the stability of axonemal microtubules through targeting Nek1, the ciliar
208 howed that, whereas the 9 + 2 arrangement of axonemal microtubules was intact, elongated cilia and ci
209 preferential depolymerization of a subset of axonemal microtubules, at either the distal or proximal
210 isotropic fluorescence signals when bound to axonemal microtubules, but the bifunctional probe is les
219 P (mantATP), which has been shown to support axonemal motility, binds all along isolated, immobilized
220 nsistent with this finding, FOXJ1-regulating axonemal motor protein expression is absent in respirato
221 few residual cilia that correctly expressed axonemal motor proteins were motile and did not exhibit
229 que cytoskeletal structure surrounds the 9+2 axonemal network in the principal piece of the flagellum
231 o the gamma heavy chain of the Chlamydomonas axonemal outer arm dynein, while a more poorly expressed
232 most highly conserved components of ciliary axonemal outer arm dyneins, and it associates with both
235 gene on the Y long arm result in loss of the axonemal outer dynein arms in the spermatid tail, while
237 at the ODA7 locus in Chlamydomonas prevents axonemal outer row dynein assembly by blocking associati
238 ive-cell imaging and morpholino depletion of axonemal Paralyzed Flagella 16 indicated that flagella-b
239 ed in biomolecular condensates termed dynein axonemal particles and synergized to control ciliogenesi
242 of microtubule sliding, indicating that the axonemal phosphatases, required for rescue, were retaine
243 he PKA binding domain results in unregulated axonemal PKA activity and inhibition of normal motility.
245 mediate the association between this 45 kDa axonemal polypeptide and the motor unit of the gamma HC.
249 ions in ZMYND10 result in the absence of the axonemal protein components DNAH5 and DNALI1 from respir
250 murine orthologue of Chlamydomonas PF16, an axonemal protein containing eight armadillo repeats pred
251 ole in sperm motility, most probably through axonemal protein phosphorylation or ion channel regulati
252 work demonstrates that TTC29 is a conserved axonemal protein required for flagellar structure and be
253 requires the transport of components such as axonemal proteins and signal transduction machinery to t
254 equires coordination between the assembly of axonemal proteins and the assembly of the flagellar memb
256 Integrated modeling identified 154 different axonemal proteins inside and outside the DMT and, togeth
257 liary membrane but also for the transport of axonemal proteins to the cilium by means of peripheral a
258 particles, motors, radial spokes, and other axonemal proteins were verified by coimmunoprecipitation
259 c-sheet protofilament to a reconstruction of axonemal protofilaments, we assigned polarity to the pro
262 n of multiple dynein motors within the 96 nm axonemal repeat and the highly coordinated interactions
265 of substructures in the internal part of the axonemal shaft requires the activity of kinesin homologu
268 rations of ADP of 114 microM, at pH 7.6, the axonemal Sp-AK could contribute approximately 31%, and S
269 vement regulator, advancing understanding of axonemal specialization in mammalian spermatozoa and its
274 egulate ciliary access of the IFT machinery, axonemal structural components, and signaling molecules,
275 n structural and phototransduction proteins, axonemal structure and disc membranes fail to form.
276 on extending from the basal body but lacking axonemal structure and remains undifferentiated until de
277 nexin-dynein regulatory complex (N-DRC), an axonemal structure critical for the regulation of dynein
278 lia, the Bbs1 M390R mutation does not affect axonemal structure, but it may play a role in the regula
279 preparation does not reveal the submembrane axonemal structure, which holds key implications for cil
282 s on motility mutants defective for specific axonemal structures, two axonemal AKAPs have been identi
284 efective motility due to mutation of various axonemal substructures were greatly enhanced compared wi
289 s, failure of dyneins to assemble within the axonemal superstructure leads to primary ciliary dyskine
290 ubulin, conserved protein Rib45, >95% of the axonemal tektins, and >95% of the calcium-binding protei
293 toreceptor cilium to form the outer segment, axonemal transport (IFT) in photoreceptors is extraordin
294 microtubules polymerized from Chlamydomonas axonemal tubulin with those from porcine brain tubulin,
295 s of cilia motility and induced a variety of axonemal ultrastructural defects similar to defects prev