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Bilateral gonadoblastoma in a 17-year-old patient with 46XY pure gonadal dysgenesis (Swyer syndrome)

  • R. Słopień1
  • G. Jarzabek-Bielecka2,*,
  • P. Jasinski4
  • M. Englert-Golon3
  • B. Burchardt3
  • M. Pisarska-Krawczyk5
  • S. Sajdak3

1Department of Gynecological Endocrinology, Poznan University of Medical Sciences, Poznan, Poland

2Unit of Developmental Age Gynecology and Sexology, Division of Gynecology, Department of Perinatology and Gynecology, Poznan University of Medical Sciences, Poznan, Poland

3Department of Operative Gynecology, Poznan University of Medical Sciences, Poznan, Poland

4Department of Pathology Gynecological and Obstetric Clinical Hospital in Poznan, Poznan, Poland

5President Stanisław Wojciechowski Vocational State School, Kalisz, Poland

DOI: 10.12892/ejgo4384.2019 Vol.40,Issue 2,April 2019 pp.325-327

Accepted: 06 September 2017

Published: 10 April 2019

*Corresponding Author(s): G. Jarzabek-Bielecka E-mail: jtckcac@gmail.com

Abstract

Gonadoblastomas are benign tumors composed of germ cells and sex cords derivatives resembling immature granulosa and Sertoli cells. They occur almost entirely in patients with gonadal dysgenesis and in those carrying the Y chromosome. The authors present a case of a 17-year-old patient admitted because of primary amenorrhea and delayed puberty diagnosed, and operated because of bilateral gonadoblastoma.

Keywords

Gonadoblastoma; Gonadal dysgenesis; Swyer syndrome

Cite and Share

R. Słopień,G. Jarzabek-Bielecka,P. Jasinski,M. Englert-Golon,B. Burchardt,M. Pisarska-Krawczyk,S. Sajdak. Bilateral gonadoblastoma in a 17-year-old patient with 46XY pure gonadal dysgenesis (Swyer syndrome). European Journal of Gynaecological Oncology. 2019. 40(2);325-327.

References

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[2] Coutin A.S., Hamy A., Fondevilla M., Savigny B., Paineau J., Visset J.: “Pure 46 XY gonadal dysgenesis”. J. Gynecol. Obstet. Biol. Reprod. (Paris), 1996, 25, 792.

[3] Behtash N., Karimi Zarchi M.: “Dysgerminoma in three patients with Swyer syndrome”. World J. Surg. Oncol., 2007, 5, 71.

[4] Gibbons B., Tan S.Y., Yu C.C., Cheah E., Tan H.L.: “Risk of gonadoblastomas in female patients with Y chromosome abnormalities and dysgenetic gonads”. J. Paediatr. Child Health, 1999, 35, 210.

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[6] Ben Romdhane K., Bessrour A., Ben Amor M.S., Ben Ayed M.:“Pure gonadal dysgenesis with 46 XY karyotyping (Swyer’s syndrome) with gonadoblastoma, dysgerminoma and embryonal carcinoma”. Bull. Cancer, 1988, 75, 263.

[7] Vollrath D., Foote S., Hilton A., Brown L.G., Beer-Romero P., Bogan J.S., Page D.C.: “The human Y chromosome: a 43-interval map based on naturally occurring deletions”. Science, 1992, 258, 52.

[8] Coutin A.S., Hamy A., Fondevilla M., Savigny B., Paineau J., Visset J.: “Pure 46XYgonadal dysgenesis”. J. Gynecol. Obstet. Biol. Reprod. (Paris), 1996, 25, 792.

[9] Cools M., Stoop H., Kersemaekers A.M., Drop S.L., Wolffenbuttel K.P., Bourguignon J.P., et al.: “Gonadoblastoma arising in undiferentiated gonadal tissue within dysgenetic gonads”. J. Clin. Endocrinol. Metab., 2006, 91, 2404.

[10] Dumic M., Jukic S., Batinica S., Ille J., Filipovic-Grcic B.: “Bilateral gonadoblastoma in a 9-month-old infant with 46,XY gonadal dysgenesis”. J. Endocrinol. Invest., 1993, 16, 291.

[11] Bremer G.L., Land J.A., Tiebosch A., van der Putten H.W.: ‘Five different histological subtypes of germ cell malignancies in an XY female”. Gynecol. Oncol., 1993, 50, 247.

[12] Seraj I.M., Chase D.R., Chase R.L., King A., Jacokes A.: “Malignant teratoma arising in a dysgenetic gonad”. Gynecol. Oncol., 1993, 50, 254.

[13] O’Neill J.A., Grosfeld J.L.: “Principles of Pediatric Surgery”. New York: Mosby (Elsevier), 2004.

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