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The in vitro metabolism of radioactive labeled testosterone and androstenedione by a minced preparation of pubic skin from a patient with the complete testicular feminization syndrome was studied. Double-labeled 5α-dihydrotestosterone and 5α-androstanedione were identified, and 5α-reductase activity was found to be quantitatively similar to that of the pubic skin from a normal subject. The in vivo effect of 5α-dihydrotestosterone-propionate was also studied in this patient with the use of two normal subjects as controls. A sharp decrease in urinary levels of phosphorus and nitrogen was noted in normal subjects, whereas in the patient absence of response to the esterified 5α-reduced testosterone was observed. These results give further support to the concept that the testicular feminization syndrome is not due to a 5α-reductase defect.