Skip to main content
SpringerLink
Log in

A sertoli-leydig cell tumor and pregnancy

Clinical, endocrine, radiologic, and electron microscopic findings

  • Case Report
  • Published:
Archives of gynecology Aims and scope Submit manuscript

Summary

An extremely rare case of a conception occurring in a 26-year-old patient with a small virilizing Sertoli-Leydig cell tumor (diameter: 0.6 cm), bilateral polycystic ovaries and non-tumorous adrenal hyperandrogenism is presented. Prepregnancy findings included hirsutism, clitoromegaly, secondary amenorrhea, and elevated peripheral plasma testosterone (T; 5.7 ng/ml). Extensive basal steroid screening, dynamic function tests, conventional radiologic procedures, selective glandular vein catheterization, and laparoscopy failed to localize unequivocally the source of androgen excess, but suggested bilateral adrenal involvement. The patient conceived during the diagnostic work-up; peripheral T levels increased to 12.1 ng/ml within the first trimester. An exploratory laparotomy with left adrenalectomy, right adrenal biopsy and left ovarian wedge resection revealed an incompletely removed Sertoli-Leydig cell tumor, but normal adrenal histology. The pregnancy was terminated, a left oophorectomy and right ovarian wedge resection were performed at 14 weeks' gestation. Subsequently, peripheral androgens returned to normal, regular menses resumed, and hirsutism disappeared. Three years later the patient delivered a healthy female infant.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  • Abraham GE, Hopper K, Tulchinsky D, Swerdloff RS, Odell WD (1971) Simultaneous measurement of plasma progesterone, 17-hydroxyprogesterone and estradiol-17β by radioimmunoassay. Anal Lett 4: 325–335

    Google Scholar 

  • Arakaki DT, Sparkes RS (1963) Microtechnique for culturing leucocytes from whole blood. Cytogenetics 2: 57–60

    Google Scholar 

  • Beierwaltes WH, Liebermann LM, Ansari AN, Nishiyana H (1971) Visualization of human adrenal glands in vivo by scintillation scanning. J Am Med Ass 216: 275–277

    Google Scholar 

  • Bernardo LA, Petrali JP, Weiss LP, Sternberger LA (1978) Adrenal luteinizing hormone releasing hormone receptors. J Histochem Cytochem 26: 613–617

    PubMed  Google Scholar 

  • Brown JB, McLeod SC, McNaughtan C, Smith MA, Smyth B (1968) Rapid method for estimating oestrogens in urine using a semiautomatic extractor. J Endocrinol 42: 5–15

    Google Scholar 

  • Buster JE, Abraham GE (1972a) Radioimmunoassay of plasma dehydroepiandrosterone sulfate. Anal Lett 5: 543–551

    Google Scholar 

  • Buster JE, Abraham GE (1972b) Radioimmunoassay of plasma dehydroepiandrosterone. Anal Lett 5: 203–215

    Google Scholar 

  • Coyotupa J, Parlow AF, Abraham GE (1972) Simultaneous radioimmunoassay of plasma testosterone and dihydrotestosterone. Anal Lett 5: 329–340

    Google Scholar 

  • Cruikshank DP, Yannone ME, Chapler FK (1974) Arrhenoblastoma associated with adrenal androgenic hyperfunction. Obstet Gynecol 43: 535–538

    PubMed  Google Scholar 

  • De Lange WE, Pratt JJ, Doorenbos H (1980) A gonadotrophin responsive testosterone producing adrenocortical adenoma and high gonadotrophin levels in an elderly woman. Clin Endocrinol 12: 21–28

    Google Scholar 

  • Fonatsch C (1979) Ionisierende Strahlen: Genetische und teratologische Aspekte. Dtsch Ä rztebl 17: 1153–1158

    Google Scholar 

  • Galle PC, McCool JA, Eisner CW (1978) Arrhenoblastoma during pregnancy. Obstet Gynecol 51: 359–364

    PubMed  Google Scholar 

  • Govan ADT (1976) Ovarian tumors: Clinical and pathological features. Clin Obstet Gynecol 3: 89–157

    Google Scholar 

  • Greenblatt RB, Mahesh VB, Gambrell RD (1972) Arrhenoblastoma — Three case reports. Obstet Gynecol 39: 567–576

    PubMed  Google Scholar 

  • Hall PE (1978) Method manual (2nd ed) — Programme for the provision of matched assay reagents for the radioimmunoassay of hormones in reproductive physiology. WHO Special Programme of Research in Human Reproduction, Genf

    Google Scholar 

  • Little GW, Estep HL, Kendell JW, William WC, Thornes AW (1959) Clinical application of a new test of pituitary reserve. J Clin Endocrinol 19: 875–894

    Google Scholar 

  • Maroulis GB, Manlimos FS, Abraham GE (1977) Comparison between urinary 17-ketosteroids and serum androgens in hirsute patients. Obstet Gynecol 49: 454–458

    PubMed  Google Scholar 

  • Meldrum DR, Abraham GE (1979) Peripheral and ovarian venous concentrations of various steroid hormones in virilizing ovarian tumors. Obstet Gynecol 53: 36–43

    PubMed  Google Scholar 

  • Moltz L (1979) Differential-diagnostic clarification of androgen-producing tumours. In: Hammer-stein J, Lachnit-Fixson U, Neumann F, Plewig G (eds) Androgenization in women. Excerpta Medica, Int Congr Series 493, Amsterdam 1980, pp 114–124

    Google Scholar 

  • Moltz L (1980) Localization of androgen-secreting tumors by ovarian-adrenal vein catheterization. Acta Endocrinol [Suppl] (Copenh) 234: 166–168

    Google Scholar 

  • Moltz L (1981) Differentialdiagnose von Androgenisierungserscheinungen durch Bestimmung der Hormonkonzentration im Organvenenblut von Ovarien und Nebennieren (selektive Katheterisierung). Habilitationsschrift, Freie Universität Berlin

    Google Scholar 

  • Moltz L (1982) Rationeller Einsatz endokrinologischer und radiologischer Verfahren bei der Differentialdiagnose von Androgenisierungserscheinungen der Frau. Geburtshilfe Frauenheilkd 42: 321–326

    PubMed  Google Scholar 

  • Moltz L, Römmler A, Schwartz U, Bidlingmaier F, Hammerstein J (1979) Peripheral steroid-gonadotropin interactions and diagnostic significance of double-stimulation tests with luteinizing hormone-releasing hormone in polycystic ovarian disease. Am J Obstet Gynecol 134: 813–818

    PubMed  Google Scholar 

  • Moltz L, Schwartz U, Pickartz H, Hammerstein J, Wolf U (1981) XY gonadal dysgenesis: aberrant testicular differentiation in the presence of H-Y antigen. Obstet Gynecol 58: 17–25

    PubMed  Google Scholar 

  • Netter A, Jayle MF, Musset R, Lambert A, Mauvais-Jarvis A (1960) Les épreuves dynamiques dans le syndrome de Stein-Leventhal. Ann Endocrinol 21: 590–600

    Google Scholar 

  • Nieuweboer B, Gerhards E, Hoffmann U (1969) Methode zur gaschromatografischen Testosteronbestimmung im Harn. Arzneimittelforsch 19: 761–764

    PubMed  Google Scholar 

  • Novak ER, Lambrou CD, Woodruff JD (1975) Ovarian tumors in pregnancy. Obstet Gynecol 46: 401–406

    PubMed  Google Scholar 

  • Pedowitz P, O'Brien FB (1960) Arrhenoblastoma of the ovary. Obstet Gynecol 16: 62–77

    PubMed  Google Scholar 

  • Römmler A (1978) Short term regulation of LH and FSH secretion in cyclic women. I. Altered pituitary response to a second of two LH-RH injections at short intervals. Acta Endocrinol (Copenh) 87: 248–258

    Google Scholar 

  • Roth LM, Cleary RE, Rosenfield RL (1974) Sertoli-Leydig cell tumor of the ovary, with an associated mucinous cystadenoma. Lab Invest 31: 648–657

    PubMed  Google Scholar 

  • Schwartz U, Moltz L, Hammerstein J (1981) Die hyperandrogenämische Ovarialinsuffizienz. Gynäkologe 14: 119–130

    Google Scholar 

  • Scully RE (1979) Ovarian tumors with endocrine manifestations. In: DeGroot LJ, Cahill GF, Odell WD, Martini L, Potts JT, Nelson DH, Steinberger E, Winegrad AI (eds) Endocrinology, vol 3. Grune and Stratton, New York, pp 1473–1488

    Google Scholar 

  • Simmer H, Hillemanns HG (1962) Arrhenoblastom und Schwangerschaft. Arch Gynecol 196: 541–574

    Google Scholar 

  • Sörensen R, Moltz L (1981) Diagnostik bei progredientem Hirsutismus — Kathetertechnik und Ergebnisse. Fortschr Röntgenstr 135: 257–266

    Google Scholar 

  • Thorn GW, Goetz FC, Streeten DHP, Dingman JF, Arnos WL (1953) Use of intravenous ACTH-test in clinical practice. J Clin Endocrinol 13: 604–613

    Google Scholar 

  • Tucci JR, Zäh W, Kalderon AE (1973) Endocrine studies in an arrhenoblastoma responsive to dexamethasone, ACTH and human chorionic gonadotropin. Am J Med 55: 687–694

    PubMed  Google Scholar 

  • Zimmermann W, Anton HU, Pontius D (1952) Die Eliminierung störender Chromogene bei der Bestimmung der 17-Ketosteroide mit m-Dinitrobenzol. Hoppe Seylers Z Physiol Chem 289: 91–102

    PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Division of Gynecological Endocrinology, Sterility and Family Planning, Dept. of Obstetrics and Gynecology, Klinikum Steglitz, Freie Universität Berlin, Hindenburgdamm 30, D-1000, Berlin 45

    L. Moltz, U. Schwartz & J. Hammerstein

  2. Dept. of Pathology, Klinikum Steglitz, Freie Universität Berlin, Hindenburgdamm 30, D-1000, Berlin 45

    H. Pickartz

  3. Dept. of Radiology, Klinikum Steglitz, Freie Universität Berlin, Hindenburgdamm 30, D-1000, Berlin 45

    R. Sörensen

Authors
  1. L. Moltz
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. H. Pickartz
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. R. Sörensen
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. U. Schwartz
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. J. Hammerstein
    View author publications

    You can also search for this author in PubMed Google Scholar

Additional information

Supported by a research grant from the Deutsche Forschungsgemeinschaft Mo 294/1-4

Rights and permissions

Reprints and permissions

About this article

Cite this article

Moltz, L., Pickartz, H., Sörensen, R. et al. A sertoli-leydig cell tumor and pregnancy. Arch. Gynecol. 233, 295–308 (1983). https://doi.org/10.1007/BF02133804

Download citation

  • Received:

  • Accepted:

  • Issue Date:

  • DOI: https://doi.org/10.1007/BF02133804

Key words

Search

Navigation