PMC:539297 / 6145-11898 JSONTXT

{"target":"https://pubannotation.org/docs/sourcedb/PMC/sourceid/539297","sourcedb":"PMC","sourceid":"539297","source_url":"http://www.ncbi.nlm.nih.gov/pmc/539297","text":"Homozygous CbCln3Δex7/8 cells express mutant battenin and display JNCL-like pathology\nHomozygous Cb Cln3Δex7/8 cells were first examined for JNCL-like characteristics. Homozygous Cln3Δex7/8 knock-in mice express multiple Cln3 mRNA splice variants and mutant battenin protein that is detectable by batp1 antibody recognizing C-terminal epitopes [12]. To assess this molecular phenotype in CbCln3Δex7/8 cells, RT-PCR and anti-battenin (batp1) immunostaining were performed. As shown in Figure 2, Cln3 mRNA isoforms in wild-type and homozygous cells were similar to those observed in total RNA isolated from wild-type or homozygous Cln3Δex7/8 knock-in brain, respectively (Fig. 2). In addition, batp1 immunostaining detected mutant battenin product in homozygous CbCln3Δex7/8 cells, in a similar albeit reduced cytoplasmic, vesicular staining pattern as that seen in wild-type cells. Batp1 signal exhibited some overlap with the lysosomal marker, Lamp1, but had more significant overlap with early endosome antigen 1 (EEA1) and the late endosomal marker, Rab7 (Fig. 3). Only limited overlap was observed with recycling endosomes, as determined by transferrin receptor co-staining (data not shown). Intriguingly, Lamp1 and EEA1 immunocytochemical distribution were altered in homozygous CbCln3Δex7/8 cells, with less perinuclear clustering than in wild-type cells, and Rab7 staining was frequently less intense in homozygous CbCln3Δex7/8 cells (Fig. 3). Heterozygous CbCln3Δex7/8 cells contained a mixture of Cln3 mRNA products from both the wild-type allele and the mutant allele, and batp1 signal was similar to that seen in wild-type cells (data not shown).\nFigure 2 RT-PCR of Cln3 mRNA in wild-type and homozygous CbCln3Δex7/8 cells Cln3 Exon1-forward, Exon 15-reverse RT-PCR products are shown, from total wild-type (+/+) or homozygous mutant (Δex7/8/Δex7/8) brain and cell line RNA. Brain and cell line RT-PCR reaction products had identical band patterns on ethidium-bromide stained agarose gels. Wild-type RT-PCR product was a single ~1.6 kb band and mutant products were ~1.6, ~1.5, ~1.4, ~1.35, and ~1.3 kb, representing multiple mutant splice variants.\nFigure 3 Battenin and lysosomal and endosomal marker co-staining in wild-type and homozygous CbCln3Δex7/8 cerebellar precursor cells Batp1 immunostaining of wild-type (CbCln3+/+) and homozygous mutant (CbCln3Δex7/8/Δex7/8) cerebellar precursor cells is shown, with co-staining for lysosomes (Lamp 1), early endosomes (EEA1), and late endosomes (Rab7). Significant overlap of Batp1 signal (red) with EEA1 (green, middle panels) and Rab7 (green, bottom panels) can be seen as yellow when the two channels are merged (Merge). The degree of Batp1 overlap is greatest with Rab7. Only limited overlap between Batp1 (red) and Lamp 1 (green, top panels) can be seen. Batp1 signal in homozygous CbCln3Δex7/8 cells is significantly reduced, but significant overlap with EEA1 and Rab7, and very little Lamp 1 overlap, can be seen as yellow in the respective merged panels. Notably, Lamp 1 and EEA1 localization appear altered, and Rab7 staining was frequently less intense in homozygous CbCln3Δex7/8 cells. Wild-type and homozygous CbCln3Δex7/8 confocal images were captured with identical exposure settings. 60 × magnification. During sub-confluent growth conditions, neither wild-type nor homozygous CbCln3Δex7/8 cells displayed autofluorescence or subunit c inclusion formation (data not shown). However, when cells were aged at confluency (3+ days post-confluency), homozygous CbCln3Δex7/8 cellular subunit c levels were elevated beyond normal wild-type levels by immunostaining (Fig. 4a) and immunoblot analysis (Fig. 4b). Autofluorescent signal sometimes overlapped with subunit c signal, but also was elevated more diffusely (Fig. 4a). Moreover, although multilamellar \"fingerprint\" profiles were not detected, confluency-aged homozygous CbCln3Δex7/8 cells displayed numerous ultrastructural abnormalities including electron dense inclusions characteristic of lipofuscin and large autophagosomes that contained dense core structures, degenerating mitochondria, and many smaller vesicles (Fig. 4c). Inclusion bodies and autophagosomes were infrequently observed in confluency-aged wild-type cultures (data not shown).\nFigure 4 Subunit c accumulation in homozygous CbCln3Δex7/8 cerebellar precursor cells a. Subunit c immunostaining and autofluorescence of 7-day confluency-aged wild-type and homozygous CbCln3Δex7/8 cells is shown. Wild-type cultures (CbCln3+/+) exhibited limited subunit c immunostaining and autofluorescence. However, CbCln3Δex7/8/Δex7/8 cells contained numerous subunit c puncta. Autofluorescence (7 days AF) was also significantly elevated (right panels), although limited overlap with subunit c puncta was observed (arrows). 40 × magnification. b. Immunoblot analysis of subunit c protein at sub-confluency or 7-day confluency incubation is shown. Total protein extracts from sub-confluency wild-type (+/+) and homozygous mutant (Δex7/8/Δex7/8) cultures contained approximately equal levels of subunit c protein (α-sub c). 7-day confluency extract from homozygous CbCln3Δex7/8 cells (Δex7/8/Δex7/8) had elevated levels of subunit c protein (~1.5X), relative to wild-type extract (+/+). Protein levels were normalized to cytochrome c oxidase subunit IV (α-cox4). c. TEM analysis of inclusions in 7-day confluency-aged homozygous CbCln3Δex7/8 cells is shown. A large autophagosome contained by double membrane (arrows) is filled with degenerating mitochondria (Md), electron dense cores (left and right of *) and other smaller vesicular structures. A large electron-dense inclusion, with a lipofuscin (Ln) appearance, is also present. M, mitochondria. 10,000 × 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