Phenotype Image Detail

Image

Caption

Impaired axonemal structure and ciliary function in Ulk4^{tm1a(KOMP)Wtsi}/Ulk4^{tm1a(KOMP)Wtsi} (-/-) ependymal cells. A-F, Transmission electron microscopic images of radial sections of ependymal cells. Clusters of multicilia with the same orientation appeared on P18 wild-type (WT) ependymal cells (A, D, arrows), but reduced (B, C, E) or absent (F) in mutant cells (B, C, E, F ). The black boxes in B and F show an immature cilium, namely a BB-vesicle fusion complex, beneath the mutant cell membrane. Cilium length was not shortened in the mutant mice (A, C-E), but the BB orientation was dispersed in the mutant cells (A, D). Arrows show normally embedded BBs in WT (D), but differently oriented in the mutant cells (E). The black box in E shows cross-sectional view of a BB foot in a vertical orientation. The black box in F shows a BB-vesicle fusion complex oriented parallel to the ependymal membrane, which varies by 90 from the normal orientation. G, WT littermates presented normal axonemes of 9 + 2. H-L, Some mutant motile cilia exhibited 9 + 2 (H), but others either lost the central doublets without (9 + 0; I ) or with disorganization of a peripheral doublet (also 9 + 0; J), or missed a peripheral doublet but had a supernumerary central doublet (8 + 4; K ), or lost peripheral and central doublets (8 + 0; L). Scale bars: A-C, 1000 nm; D-F, 500 nm; G-L, 100 nm. Insets in B, E, and F were magnified three times from the respective images. MV, Microvilli; N, nucleus; V, vesicle.

Copyright

This image is from Liu M, J Neurosci 2016 Jul 20;36(29):7589-600, an open-access article distributed under the terms of the Creative Commons Attribution-Noncommerical-NoDerivatives 4.0 International License. J:234631

Associated Alleles

Symbol	Name
Ulk4^tm1a(KOMP)Wtsi	unc-51-like kinase 4; targeted mutation 1a, Wellcome Trust Sanger Institute
Ulk4^tm1a(KOMP)Wtsi	unc-51-like kinase 4; targeted mutation 1a, Wellcome Trust Sanger Institute

Associated Genotypes

Allelic Composition	Genetic Background
Ulk4^tm1a(KOMP)Wtsi/Ulk4^tm1a(KOMP)Wtsi	involves: C57BL/6N