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Complete hydatidiform mole and a coexistent fetus following ovulation induction in a patient with Sheehan’s syndrome: a first case report and review of literature

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Abstract

Pregnancy in Sheehan’s syndrome (SS) is extremely rare. We present the first reported case of twin pregnancy with complete hydatiform mole (CHM) and a coexistent fetus (CHCF) in a patient with SS. A 29-year-old Chinese patient with SS became pregnant following one cycle of ovulation induction with human menopausal gonadotropin after secondary infertility. A normal live fetus and a low echogenic mass suspected hydatidiform mole (HM) were detected by ultrasound examinations at gestational week 8. The couple highly desired to continue the pregnancy because it is very hard to get pregnant for the patients with SS. However, the pregnancy was terminated for the size of the HM component increased rapidly at gestational week 15. Histological examinations confirmed CHCF. Genetic studies showed that the CHM genome was derived from paternal diploidy, and the normal fetus was from biparental genomes. Furthermore, a literature review on these topics is included. This case highlighted that even in a patient with SS, twin pregnancy with CHCF can still occur after ovulation induction.

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References

  1. Kelestimur F (2003) Sheehan’s syndrome. Pituitary 6(4):181–188

    Article  PubMed  Google Scholar 

  2. Kristjansdottir HL, Bodvarsdottir SP, Sigurjonsdottir HA (2011) Sheehan’s syndrome in modern times: a nationwide retrospective study in Iceland. Eur J Endocrinol 164(3):349–354

    Article  CAS  PubMed  Google Scholar 

  3. Grimes HG, Brooks MH (1980) Pregnancy in Sheehan’s syndrome. Report of a case and review. Obstet Gynecol Surv 35(8):481–488

    Article  CAS  PubMed  Google Scholar 

  4. Kriplani A, Goswami D, Agarwal N, Bhatla N, Ammini AC (2000) Twin pregnancy following gonadotrophin therapy in a patient with Sheehan’s syndrome. Int J Gynaecol Obstet 71(1):59–63

    Article  CAS  PubMed  Google Scholar 

  5. Malhotra N, Deka D, Takkar D, Kochar S, Goel S, Sharma MC (2001) Hydatiform mole with coexisting live fetus in dichorionic twin gestation. Eur J Obstet Gynecol Reprod Biol 94(2):301–303

    Article  CAS  PubMed  Google Scholar 

  6. Lee SW, Kim MY, Chung JH, Yang JH, Lee YH, Chun YK (2010) Clinical findings of multiple pregnancy with a complete hydatidiform mole and coexisting fetus. J Ultrasound Med 29(2):271–280

    PubMed  Google Scholar 

  7. Massardier J, Golfier F, Journet D, Frappart L, Zalaquett M, Schott AM, Lenoir VT, Dupuis O, Hajri T, Raudrant D (2009) Twin pregnancy with complete hydatidiform mole and coexistent fetus: obstetrical and oncological outcomes in a series of 14 cases. Eur J Obstet Gynecol Reprod Biol 143(2):84–87

    Article  PubMed  Google Scholar 

  8. Gualandi F, Sensi A, Trabanelli C, Falciano F, Bonfatti A, Calzolari E (2000) Prenatal UPD testing survey in Robertsonian translocations. Prenat Diagn 20(6):465–468

    Article  CAS  PubMed  Google Scholar 

  9. Barkan AL (1989) Pituitary atrophy in patients with Sheehan’s syndrome. Am J Med Sci 298(1):38–40

    Article  CAS  PubMed  Google Scholar 

  10. Ozbey N, Inanc S, Aral F, Azezli A, Orhan Y, Sencer E, Molvalilar S (1994) Clinical and laboratory evaluation of 40 patients with Sheehan’s syndrome. Isr J Med Sci 30(11):826–829

    CAS  PubMed  Google Scholar 

  11. Shahmanesh M, Ali Z, Pourmand M, Nourmand I (1980) Pituitary function tests in Sheehan’s syndome. Clin Endocrinol (Oxf) 12(3):303–311

    Article  CAS  Google Scholar 

  12. Shivaprasad C (2011) Sheehan’s syndrome: Newer advances. Indian J Endocrinol Metab 15(Suppl 3(S)):203–207

    Article  Google Scholar 

  13. Algun E, Ayakta H, Harman M, Topal C, Aksoy H (2003) Spontaneous pregnancy in a patient with Sheehan’s syndrome. Eur J Obstet Gynecol Reprod Biol 110(2):242–244

    Article  CAS  PubMed  Google Scholar 

  14. See TT, Lee SP, Chen HF (2005) Spontaneous pregnancy and partial recovery of pituitary function in a patient with Sheehan’s syndrome. J Chin Med Assoc 68(4):187–190

    Article  PubMed  Google Scholar 

  15. Hao J, Liu M, Mo Z (2012) The symptoms get worse after pregnancy in Sheehan’s syndrome: a case report. Case Rep Med 2012:271345

    PubMed Central  PubMed  Google Scholar 

  16. Jain D (2013) A ray of hope for a woman with Sheehan’s syndrome. BMJ Case Rep

  17. Vieira HB, Knoepfelmacher M, Salgado LR, Wajchenberg BL, Liberman B (1995) Preservation of gonadotrophic function and pregnancy in Sheehan’s syndrome: a case report and review of the literature. Rev Assoc Med Bras 41(2):135–138

    CAS  PubMed  Google Scholar 

  18. Cohen BL, Baillie P (1980) Sheehan’s syndrome followed by successful pregnancy: a case report. S Afr Med J 57(20):838–840

    CAS  PubMed  Google Scholar 

  19. Briet JW (1998) Diabetes insipidus, Sheehan’s syndrome and pregnancy. Eur J Obstet Gynecol Reprod Biol 77(2):201–203

    Article  CAS  PubMed  Google Scholar 

  20. Barbieri RL, Randall RW, Saltzman DH (1985) Diabetes insipidus occurring in a patient with Sheehan’s syndrome during a gonadotropin-induced pregnancy. Fertil Steril 44(4):529–531

    CAS  PubMed  Google Scholar 

  21. Kilicli F, Dokmetas HS, Acibucu F (2013) Sheehan’s syndrome. Gynecol Endocrinol 29(4):292–295

    Article  CAS  PubMed  Google Scholar 

  22. Yildirim M, Noyan V, Tiras MB (2000) Estrogen-progestogen pre-treatment before HMG induction in hypogonadotropic patients. Int J Gynaecol Obstet 71(3):249–250

    Article  CAS  PubMed  Google Scholar 

  23. Sheehan HL (1948) Post-partum necrosis of the anterior pituitary. Ir J Med Sci 270:241–255

    PubMed  Google Scholar 

  24. Jorgensen PI, Sele V, Buus O, Damkjoer M (1973) Detailed hormonal studies during and after pregnancy in a previously hypophysectomized patient. Acta Endocrinol (Copenh) 73(1):117–132

    CAS  Google Scholar 

  25. Ferraz TJ, Bartosch CM, Ramalho CM, Carvalho FA, Carvalho BC, Brandao OG, Montenegro NA (2013) Complete mole in a dichorionic twin pregnancy after intracytoplasmic sperm injection. Rev Bras Ginecol Obstet 35(1):39–43

    Article  PubMed  Google Scholar 

  26. Genest DR (2001) Partial hydatidiform mole: clinicopathological features, differential diagnosis, ploidy and molecular studies, and gold standards for diagnosis. Int J Gynecol Pathol 20(4):315–322

    Article  CAS  PubMed  Google Scholar 

  27. Sebire NJ, Foskett M, Paradinas FJ, Fisher RA, Francis RJ, Short D, Newlands ES, Seckl MJ (2002) Outcome of twin pregnancies with complete hydatidiform mole and healthy co-twin. Lancet 359(9324):2165–2166

    Article  PubMed  Google Scholar 

  28. Fishman DA, Padilla LA, Keh P, Cohen L, Frederiksen M, Lurain JR (1998) Management of twin pregnancies consisting of a complete hydatidiform mole and normal fetus. Obstet Gynecol 91(4):546–550

    Article  CAS  PubMed  Google Scholar 

  29. Vaisbuch E, Ben-Arie A, Dgani R, Perlman S, Sokolovsky N, Hagay Z (2005) Twin pregnancy consisting of a complete hydatidiform mole and co-existent fetus: report of two cases and review of literature. Gynecol Oncol 98(1):19–23

    Article  PubMed  Google Scholar 

  30. Aguin E, Aguin V, Cisneros L, Aguin T, Van de Ven C, Bahado-Singh R (2012) Does cerclage improve neonatal outcomes in a molar pregnancy and a coexistent fetus? A case report. BMC Res Notes 5:621

    Article  PubMed Central  PubMed  Google Scholar 

  31. Malinowski W, Biskup I, Dec W (1995) Sad fetus syndrome-gestational trophoblastic disease concurrent with a living fetus or fetuses. Acta Genet Med Gemellol (Roma) 44(3–4):193–202

    CAS  Google Scholar 

  32. Dolapcioglu K, Gungoren A, Hakverdi S, Hakverdi AU, Egilmez E (2009) Twin pregnancy with a complete hydatidiform mole and co-existent live fetus: two case reports and review of the literature. Arch Gynecol Obstet 279(3):431–436

    Article  PubMed  Google Scholar 

  33. Piura B, Rabinovich A, Hershkovitz R, Maor E, Mazor M (2008) Twin pregnancy with a complete hydatidiform mole and surviving co-existent fetus. Arch Gynecol Obstet 278(4):377–382

    Article  PubMed  Google Scholar 

  34. Bruchim I, Kidron D, Amiel A, Altaras M, Fejgin MD (2000) Complete hydatidiform mole and a coexistent viable fetus: report of two cases and review of the literature. Gynecol Oncol 77(1):197–202

    Article  CAS  PubMed  Google Scholar 

  35. Sun CJ, Zhao YP, Yu S, Fan L, Wu QQ, Li GH, Zhang WY (2012) Twin pregnancy and partial hydatidiform mole following in vitro fertilization and embryos transfer: a novel case of placental mosaicism. Chin Med J (Engl) 125(24):4517–4519

    Google Scholar 

  36. Huang X, Wang H, Zhao X, Xu X, Chen Q (2007) Gestational trophoblastic disease following in vitro fertilization. Arch Gynecol Obstet 275(4):291–293

    Article  PubMed  Google Scholar 

  37. Montes-de-Oca-Valero F, Macara L, Shaker A (1999) Twin pregnancy with a complete hydatidiform mole and co-existing fetus following in vitro fertilization: case report. Hum Reprod 14(11):2905–2907

    Article  CAS  PubMed  Google Scholar 

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Conflict of interest

The authors declare no conflict of interest.

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Authors and Affiliations

  1. Reproductive Medicine Center, Department of Obstetrics and Gynaecology, The First People’s Hospital of Foshan, Foshan, 528000, Guangdong, People’s Republic of China

    Xuekun Huang, Yonghan Huang & Juanhua Huang

  2. Department of Neurology, Chinese PLA General Hospital, Beijing, 100853, People’s Republic of China

    Jingyao Liang

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  1. Xuekun Huang
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  2. Jingyao Liang
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  3. Yonghan Huang
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  4. Juanhua Huang
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Corresponding author

Correspondence to Xuekun Huang.

Additional information

X. Huang and J. Liang contributed equally to this work.

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Huang, X., Liang, J., Huang, Y. et al. Complete hydatidiform mole and a coexistent fetus following ovulation induction in a patient with Sheehan’s syndrome: a first case report and review of literature. Arch Gynecol Obstet 289, 1145–1150 (2014). https://doi.org/10.1007/s00404-013-3118-z

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  • DOI: https://doi.org/10.1007/s00404-013-3118-z

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