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The combination of testicular biopsy and intracytoplasmic sperm injection procedures has assisted potentially azoospermic men with Klinefelters’ syndrome to have children.  Approximately two-thirds of the time, a testicular sperm extraction procedure performed on a nonmosaic Klinefelter 47,XXY patient fails to isolate sperm. In the case report described herein, both wet preparation and histological examinations confirmed typical cellular morphologies associated with Klinefelter testicular tissue, including no spermatozoa.  It did however, observe the unusual presence of oocyte-like cells representing a unique, theoretical in vivo transformation of pluripotent stem cells, probably spermatogonial, into primordial germ cells and gametes of the opposite sex, as previously documented only in the experimental mouse model.