Human mutations in axonemal dyneins are indeed associated with Primary Ciliary Dyskinesia. The dnai2 ortholog has been linked to PCD, as have other dyneins, although mutations in dnah3 orthologs have not been identified or studied in humans. Our work suggests however that human dnah3 mutations likely would show a PCD phenotype. Here is the reference associating dnai2 to PCD: DNAI2 mutations cause primary ciliary dyskinesia with defects in the outer dynein arm. <up>Am J Hum Genet. 2008</up> Loges NT, Olbrich H, Fenske L, Mussaffi H, Horvath J, Fliegauf M, Kuhl H, Baktai G, Peterffy E, Chodhari R, Chung EM, Rutman A, O'Callaghan C, Blau H, Tiszlavicz L, Voelkel K, Witt M, Zietkiewicz E, Neesen J, Reinhardt R, Mitchison HM, Omran H Am J Hum Genet. 2008 Nov; 83(5):547-58. Daniel F. Eberl University of Iowa The human orthologs of one of the two fly genes we are reporting on is implicated in primary ciliary dyskinesia (DNAI2, see Loges NT et al. (2008), Am. J. Human Genet. 83, 547-58). We show that disrupting the respective fly gene (Dmdnai2) aslo causes cilium defects. Martin Göpfert Universität Göttingen