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Systematic detection of endocrine cells was performed in two genital tracts from patients with Peutz-Jeghers syndrome (PJS). These tissues proved to be particularly rich in endocrine cells. The specialized cells were distributed in the cervix and fallopian tubes. In the cervix, they were confined to remarkable mutinous tumors related to “adenoma malignum.” Serotonin, somatostatin, gastrin, and pancreatic polypeptide immunoreactive cells were characterized. In fallopian tubes, serotonin-storing cells and somatostatin cells were detected respectively among normal-appearing and mucinous areas of tubal epithelium; in addition, serotonin-storing cells were found in many mesonephric rests. This strongly contrasts with the usual paucity of endocrine cells in the female genital tract. However, none of the findings mentioned was really specific of PJS. In particular, endocrine cells seem to be an integral constituent of adenoma malignum, with or without PJS. These findings suggest a disturbance of tissular differentiation.