INPP5B can partly rescue the defect of OCRL in primary cilia development.

<p>(A) INPP5B protein levels in HEK293T cells. <i>GFP-Inpp5b</i> lentivirus was generated in HEK293T cells; both GFP-Inpp5b and endogenous INPP5B were immunoblotted in 40 microgram lysates; beta-actin levels are shown. (B) NHF 558, Lowe 1676, and Lowe 3265 fibroblasts were transduced with control, <i>Inpp5b</i> or <i>Inpp5b ΔCAAX</i> lentivirus, serum-starved for 48-hours, and immunostained with acetylated alpha -tubulin. Quantification of cilia length is shown (n >100 cilia, three independent experiments, unpaired t-test, * p = 0.004, ** p = 0.006). (C) <i>Inpp5b</i> mRNA partly rescued the loss of <i>Ocrl</i>. KV cilia of zebrafish embryos injected with <i>ocrl</i> MO (4 ng) with <i>Inpp5b</i> WT mRNA (500 ng) and <i>ocrl</i> MO (4 ng) with <i>Inpp5bΔCAAX</i> mRNA (500 ng) at 6-somite stage were immunostained with acetylated α-tubulin (red), representative images are shown (dash line indicates border of KV). Scale bar 10 micron. (D) Quantification of length of KV cilia in zebrafish embryos injected with <i>ocrl</i> MO (4 ng) with <i>Inpp5b</i> WT mRNA (500 ng) and <i>ocrl</i> MO (4 ng) with <i>Inpp5bΔCAAX</i> mRNA (500 ng). (N >20 embryos, three independent experiments, unpaired t-test, * p = 2.2E-04).