Phenotype Image Detail

Image

Caption

Accumulation of autofluorescent, electron-dense, membrane-enclosed material in Zfyve26^tm1.1Cahb/Zfyve26^tm1.1Cahb (KO) mice. (A, E) In KO mice autofluorescent material (excited at 488 nm) was already observed in neurons at 2 months of age (white arrows). (B, F) At 16 months of age these deposits were drastically enlarged in KO mice. Purkinje cell somata in (A-B ,E-F) are indicated by a dashed line. ML: molecular layer; PCL: Purkinje cell layer; GCL: granule cell layer. Scale bars: 25 um. (C-D, G-H) Although lipofuscin particles were found in both wild-type (WT) and KO mice at 16 months of age, abnormal clusters of membrane enclosed deposits were only observed in KO samples (H, highlighted region). For clarity the tissue surrounding the Purkinje cell soma has been dampened in (C) and (G). The magnification of the highlighted regions in (C) and (G) are shown in (D) and (H). A regular lipofuscin particle in (D) and (H) is marked with a black arrow. The borders of the abnormal cluster shown in (H) are indicated with arrowheads. (I) Large membrane enclosed deposits were also found in KO Purkinje cells. Regular lysosomal vesicles were noted both in WT (J) and KO samples (K). (L) In KO samples some lysosomal structures showed similarities with fingerprint bodies, which are typical for juvenile neuronal ceroid lipofuscinosis. Scale bars: 5 um (C, G), 2 um (D, H, I) and 0.5 um (J-L).

Copyright

This image is from Khundadze M, PLoS Genet 2013;9(12):e1003988, and is displayed under the terms of the Creative Commons Attribution 4.0 International License. J:223127

Associated Alleles

Symbol	Name
Zfyve26^tm1.1Cahb	zinc finger, FYVE domain containing 26; targeted mutation 1.1, Christian A Hubner

Associated Genotypes

Allelic Composition	Genetic Background
Zfyve26^tm1.1Cahb/Zfyve26^tm1.1Cahb	involves: 129S1/Sv * 129X1/SvJ * BALB/cJ * C57BL/6