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. 2020 Nov 2;11:5520. doi: 10.1038/s41467-020-19113-0

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© The Author(s) 2020

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.

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Fig. 3 — a Cartoon of “9 + 2” axoneme in respiratory cilia. Axonemal defects that correlate with certain PCD-causing mutations are highlighted in red. Isolated ODA defects in DNAH5- (b), DNAI2- (c), and CCDC151-mutant (d) respiratory cilia do not affect axonemal CFAP45 (red) localization. Compound ODA and IDA defects in DNAAF1- (e) and DNAAF3-mutant (f) respiratory cilia do not affect axonemal CFAP45 (red) localization. N-DRC defects lacking IDAs and with tubular disorganization in CCDC40- (g) and CCDC39-mutant (h) respiratory cilia do not affect axonemal CFAP45 (red) localization. RS defects in RSPH4A- (i) and RSPH9-mutant (j) respiratory cilia do not affect axonemal CFAP45 (red) localization. k–o Certain DNAH11-mutant respiratory cilia show abnormal ciliary CFAP45 (red) localization (rectangles). p Healthy sibling of OP-406 II2 (o) shows wild type CFAP45 localization. Ciliary axonemes (green) are detected using anti-acetylated α tubulin (AcTub) antibody. Merge images include Hoechst stain (blue) to indicate nuclei. White scale bar equals 10 µm. b–p n = 18 images from three experiments. PCD individuals with respective mutations are indicated. DNAH11 mutations are a common cause of PCD with LRA abnormalities^33–36 but do not perturb the axonemal localization of ODA protein DNAH5 and IDA protein DNALI1^33,35. Per the study design, DNAH5 and DNALI1 are detectable by IFM in respiratory cilia from study cohort individuals (see “Methods”), suggesting that CFAP45 deficiency does not cause gross ODA and IDA abnormalities.