pgen.1010558.s002.pdf (172.51 kB)
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posted on 2023-01-10, 18:30 authored by Clorissa L. Washington-Hughes, Shubhrajit Roy, Herana Kamal Seneviratne, Senthilkumar S. Karuppagounder, Yulemni Morel, Jace W. Jones, Alex Zak, Tong Xiao, Tatiana N. Boronina, Robert N. Cole, Namandjé N. Bumpus, Christopher J. Chang, Ted M. Dawson, Svetlana LutsenkoCilia density is decreased in the 4 week-old Atp7b-/- ChPl compared to controls (a) Immunofluorescent staining for ciliary membrane marker Arl13b (green), gamma tubulin (red) on ChPl from 4 weeks Atp7b-/- and Wild type controls. The data here for Arl13b/γ-tub/DAPI merged is the same as in Fig 3G. Scale Bar 20 μm (b) Merged Z stack images for Arl13b (white) and gamma tubulin (red) from Wild type and Atp7b-/- ChPl were used to count the number of ciliated cells. The ciliated structures were marked using a white box. Scale Bar 20 μm (c) Percentage of ciliated epithelial cells between Atp7b-/- and wild type ChPl were calculated from these images. Data represented as Means ± SD. *P < 0.001 (Welch corrected t-test). n = 3–4
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xlink "> coppermenkes disease proteinchpl &# 8217despite normal expressionatp7b causes reorganizationsignificant cu deficitlimits cu transportchpl lacking atp7bpostnatal development atp7bidentify metabolic changesmetabolic changeschpl ).normal morphologynotable changesbrain developmentcu deficiencyactivates curesults identifyremains intracellularphosphatidylethanolamine conversionolder animalslocus coeruleusless cleardependent enzymeschoroid plexuscatecholamine imbalanceapical membrane
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