FIGURE

Fig 6

ID

ZDB-FIG-210714-27

Publication

Pottie et al., 2021 - Loss of zebrafish atp6v1e1b, encoding a subunit of vacuolar ATPase, recapitulates human ARCL type 2C syndrome and identifies multiple pathobiological signatures

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Fig 6

Early respiratory chain deficits resulting from <italic toggle='yes'>atp6v1e1b</italic> depletion <italic toggle='yes'>in vivo</italic>.

(A) Representative images of ultrathin sections of the yolk from WT control and atp6v1e1b-deficient zebrafish and WT controls at 4 dpf. Asterisk, normal mitochondria. Diamond, dilated mitochondria. Scale bar = 500 nm. Results are representative of three independent experiments. (B) Measurement of the basal respiration and (C) extracellular acidification rate in atp6v1e1b^{hi577aTg/hi577aTg} and atp6v1e1b^cmg78/cmg78 zebrafish larvae at 2 dpf compared to WT controls. (D) Plot of OCR vs. ECAR values showed that both mutant zebrafish larvae are in a more quiescent aerobic state than their WTs. (E-H) OCR in WT control and atp6v1e1b-deficient zebrafish after administration of oligomycin and rotenone/antimycin a or FCCP and rotenone/antimycin a. ECAR: extracellular acidification rate; OCR: oxygen consumption rate. Data are expressed as mean ± standard error of the mean (SEM) from 5 biological replicates.

Expression Data

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Antibody Labeling

Phenotype Data

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Acknowledgments

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