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Primary amenorrhea revealing an occult progesterone-secreting ovarian tumor.
- Source :
-
Fertility and sterility [Fertil Steril] 2008 Oct; Vol. 90 (4), pp. 1198.e1-5. Date of Electronic Publication: 2008 Mar 04. - Publication Year :
- 2008
-
Abstract
- Objective: To describe a patient with primary amenorrhea revealing an occult progesterone-secreting ovarian tumor.<br />Design: Case report.<br />Setting: University medical center.<br />Patient(s): A 20-year-old woman with primary amenorrhea.<br />Intervention(s): Investigations to identify the source of progesterone secretion.<br />Main Outcome Measure(s): Discovery of an occult progesterone-secreting ovarian tumor.<br />Result(s): Initial ovarian ultrasonography did not show any abnormal mass. Catheterization of ovarian veins suggested a right ovarian source of progesterone. After long-term follow-up, a right ovarian tumor became apparent and was surgically removed. After surgery, progesterone levels decreased and normal ovulatory cycles resumed. Pathologic and immunohistochemical analysis showed a Leydig cell tumor expressing cytochrome P450 side-chain cleavage and 3ss-hydroxysteroïd dehydrogenase enzymes, which are involved in progesterone biosynthesis, whereas P45017alpha-hydroxylase was not expressed, explaining the absence of hyperandrogenemia. Before surgery, two LH pulses were detected during a 6-hour study period and a lack of ovarian response to pulsatile GnRH administration.<br />Conclusion: This is the first case of isolated progesterone secretion by an occult ovarian Leydig cell tumor and a novel etiology of primary amenorrhea. The results also suggest that sustained progesterone can exert an inhibitory effect on gonadotropin secretion at both hypothalamic and pituitary levels.
- Subjects :
- Adult
Amenorrhea complications
Female
Humans
Incidental Findings
Neoplasms, Unknown Primary complications
Neoplasms, Unknown Primary therapy
Ovarian Neoplasms complications
Ovarian Neoplasms metabolism
Ovarian Neoplasms therapy
Amenorrhea diagnosis
Neoplasms, Unknown Primary diagnosis
Neoplasms, Unknown Primary metabolism
Ovarian Neoplasms diagnosis
Ovarian Neoplasms secondary
Progesterone metabolism
Subjects
Details
- Language :
- English
- ISSN :
- 1556-5653
- Volume :
- 90
- Issue :
- 4
- Database :
- MEDLINE
- Journal :
- Fertility and sterility
- Publication Type :
- Academic Journal
- Accession number :
- 18304537
- Full Text :
- https://doi.org/10.1016/j.fertnstert.2007.11.064