Faculty Research 1970 - 1979
Mitochondrial proline dehydrogenase deficiency in hyperprolinemic PRO/Re mice: genetic and enzymatic analyses.
Document Type
Article
Publication Date
1976
Keywords
Amino-Acid-Metabolism-Inborn-Errors: en, Amino-Acid-Oxidoreductases: me, Animal, Genes-Structural, Heat, Kinetics, Mice, Mice-Inbred-C57BL, Mitochondria-Liver: en, ul, Oxygenases: df, Proline: bl, me, Species-Specificity, SUPPORT-U-S-GOVT-P-H-S, Vitamin-K
First Page
739
Last Page
757
JAX Location
40,351
JAX Source
Biochem-Genet. 1976 Oct; 14(9-10):739-57.
Abstract
Genetic analyses, involving backcross and F2 matings, demonstrate that the type I hyperprolinemia of PRO/Re mice is caused by an abnormal allelet at a single locus designated pro-1. Mice homozygous for this allele (pro-1b/pro-1b) posses a deficiency in the activity of component 1 of mitochondrial proline dehydrogenase. In liver mitochondria of normal C57BL/6J mice, two proline dehydrogenase activity components are demonstrable by electrophoretic resolution of Triton X-100 solubilized extracts. In mitochondria of PRO/Re mice, the activity of component 1 is not readily detectable. Residual proline dehydrogenase activity in PRO/Re mitochondria appears, therefore, to be due in large measure to activity component 2 which is more stable to incubation at 40 C, exhibits slower electrophoretic mobility, and is less reactive to menadione. Kinetic analyses demonstrate a Km (proline) for the Triton X-100 solubilized enzyme activities of PRO/Re and C57BL/6J liver mitochondria of 0.4 M and 2.9 X 10(-3) M, respectively. C57BL/6J enzyme activity is inhibited by high substrate concentration. The actins of PRO/Re liver obtained by differential centrifugation. Abnormal control of respiratory chain function in PRO/Re mitochondria appears to involve primarily proline oxidation, as indicated by the level of activity of several inner membrane enzymes.
Recommended Citation
Blake RL,
Hall JG,
Russell ES.
Mitochondrial proline dehydrogenase deficiency in hyperprolinemic PRO/Re mice: genetic and enzymatic analyses. Biochem-Genet. 1976 Oct; 14(9-10):739-57.