BLD10/CEP135 is a microtubule-associated protein that controls the formation of the flagellum central microtubule pair.
Cilia and flagella are involved in a variety of processes and human diseases, including ciliopathies and sterility. Their motility is often controlled by a central microtubule (MT) pair localized within the ciliary MT-based skeleton, the axoneme. We characterized the formation of the motility apparatus in detail in Drosophila spermatogenesis. We show that assembly of the central MT pair starts prior to the meiotic divisions, with nucleation of a singlet MT within the basal body of a small cilium, and that the second MT of the pair only assembles much later, upon flagella formation. BLD10/CEP135, a conserved player in centriole and flagella biogenesis, can bind and stabilize MTs and is required for the early steps of central MT pair formation. This work describes a genetically tractable system to study motile cilia formation and provides an explanation for BLD10/CEP135's role in assembling highly stable MT-based structures, such as motile axonemes and centrioles.
Instituto Gulbenkian de Ciência, Rua da Quinta Grande 6, 2780-156 Oeiras, Portugal. zitasantos@igc.gulbenkian.pt
Zita
Z
Carvalho-Santos
Pedro
P
Machado
Inês
I
Alvarez-Martins
Susana M
SM
Gouveia
Swadhin C
SC
Jana
Paulo
P
Duarte
Tiago
T
Amado
Pedro
P
Branco
Micael C
MC
Freitas
Sara T N
ST
Silva
Claude
C
Antony
Tiago M
TM
Bandeiras
Mónica
M
Bettencourt-Dias
D000818:Animals; D029721:Drosophila Proteins / Q000235:genetics / Q000378:metabolism*; D004331:Drosophila melanogaster / Q000166:cytology / Q000235:genetics / Q000378:metabolism*; D005407:Flagella / Q000378:metabolism*; D008297:Male; D046529:Microscopy, Electron, Transmission; D008869:Microtubule-Associated Proteins / Q000378:metabolism*; D008870:Microtubules / Q000378:metabolism*; D011485:Protein Binding; D013091:Spermatogenesis
C542499:CEP135 protein, Drosophila; D029721:Drosophila Proteins; D008869:Microtubule-Associated Proteins