Figure 1. Identification of Pex10CY/CY mutants with defects in peroxisome function.
A. Touch assay to examine movement during late stages of embryonic mouse development shows a progressive loss of locomotor movement in Pex10CY/CY mutants. B. Neonatal littermates within 2 hours of birth. Pex10CY/CY mice are cyanotic and show no milk in the stomach (black arrows). C. A cysteine in the conserved PEX10 zinc ring finger binding domain (red letters, C3HC4) is mutated in Pex10CY/CY mice (asterisk). D. Surviving Pex10CY/CY mouse (right) shown with wildtype littermate. E. Difference in weight between Pex10CY/CY mice (n=2) and wildtype littermates (n=3). F, G Antibodies to catalase label the peroxisomes in wildtype embryonic fibroblast (F) but shows only diffuse cytoplasmic labeling in Pex10CY/CY MEFs (G). H, I Peroxisomal import signal EGFP-SKL is localized to peroxisomes in wildtype MEFs (H) but no punctate labeling is seen in Pex10CY/CY MEFs (I). Scale bar=50 µm.