Axonemal lengths are disrupted in Kdm3a mutant mice. (A) Wild-type (WT) and Kdm3a^ΔJC/ΔJC mutant spermatozoa from mouse epididymis; the latter have rounded heads and short flagella. (B) Schematic of the gene-targeted deletion of Kdm3a^ΔJC mutant mouse model. Domains: yellow, C6-type zinc finger (ZF); blue, LXXLL nuclear receptor binding (NR); red, JmjC domain (JmjC). (C) Mean ± 5th–95th percentile of flagellar lengths of epididymal spermatozoa derived from N ≥ 3 per genotype; n is indicated below each box. (D) Mean ± 5th–95th percentile of MEF ciliary lengths of MEFs (N ≥ 3 per genotype) after 24-h serum depletion. (E) Mean ± 5th–95th percentile of MEF ciliary lengths transfected with GFP vector (control [CO]) or GFP-KDM3A_i2, showing partial rescue of the mutant short cilia phenotype. (F) Immunostaining illustrates ciliary accumulations of IFT88 in Kdm3a^ΔJC mutant MEFs. (G) Ratio of averaged fluorescence intensity of IFT88 relative to acetylated α-tubulin measured from the basal body to the ciliary tip along ciliary axonemes. Error bars represent ± SEM for >10 cilia per genotype. Asterisk denotes significant p-value from t test: *, P < 0.05; ***, P < 0.001; ****, P < 0.0001.