Spontaneous Ovarian Hyperstimulation Syndrome Diagnosed Within First Trimester And Persisted Up To Birth: Case Report

Abstract

Ovarian hyperstimulation syndrome OHSS most commonly occurs iatrogenically in association with assisted reproductice technologies. Its incidence is 0,2- 1%. Although the etiopathogenesis of spontaneous ovarian hyperstimulation is unclear, there are only presented limited cases of pregnancies associated with spontaneous ovarian hyperstimulation in literature. It was suggested that polycystic ovary syndrome, twin and molar pregnancies in which the endogenous hCG levels were higher than normal and hypothyroidism could be risk factors for spontaneous ovarian hyperstimulation. In addition, It is suggested that the FSH receptor mutations or polymorphisms in a single gene has been responsible for spontaneous OHSS. This report describes a case in which a naturally conceived pregnancy was associated with spontaneous ovarian hyperstimulation with no regression during pregnancy. Such cases should be investigated in terms of gene mutations, and patients should be followed closely due to clinical manifestation.

Keywords

• Spontaneous Ovarian hyperstimulation syndrome,FSH receptor mutation,Single gene polymorphism

Birinci Trimesterde Tanısı Konulan ve Doğuma Kadar Sebat Eden Spontan Ovarian Hiperstimülasyon Sendromu: Olgu Sunumu

Abstract

Ovarian hiperstimülasyon sendromu OHSS yardımcı üreme teknikleri ile birlikte gelişen yatrojenik bir durumdur. Sıklığı %0,2-1 arasında değişmektedir. Etyopatogenezi tam olarak aydınlatılmamış olup, literatürde nadir olarak spontan hiperstimülasyon sendromu bildirilmiştir. Polikistik over sendromu, endojen hCG seviyelerinin yüksek seyrettiği çoğul ve molar gebelikler ve hipotiroidi, risk faktörleri olarak saptanmıştır. Ayrıca FSH reseptör mutasyonu veya tek gen polimorfizmin de sorumlu olabileceği ileri sürülmüştür. Biz de herhangi bir risk faktörü tespit edilmeyen ve gebelik boyunca gerilemeyen dirençli spontan OHSS vakası sunmayı amaçladık. Bu tür vakalarda gen mutasyonları araştırılmalı, klinik seyir açısından hastalar yakın takibe alınmalıdır.

Keywords

• Spontan Ovarian hiperstimülasyon sendromu,FSH reseptör mutasyonu,Tek gen polimorfizmi

References

1. Delvigne A, Rozenberg S. Epidemiology and prevention of ovarian hy- perstimulation syndrome (OHSS): a review. Hum Reprod Update 2002; 8:559.
2. Binder H, Dittrich R, Einhaus F, et al. Update on ovarian hyperstimulation syndrome: Part 1--Incidence and pathogenesis. Int J Fertil Womens Med 2007; 52:11.
3. Abu-Louz SK, Ahmed AA, Swan RW. Spontaneous ovarian hyperstimu- lation syndrome with pregnancy. Am J Obstet Gynecol 1997; 177: 476- 477.
4. Smits G, Olatunbosun O, Delbaere A, Pierson R, Vassart G, Costagliola S. Ovarian hyperstimulation syndrome due to a mutation in the folliclestimu- lating hormone receptor. N Engl J Med 2003; 349: 760-766.
5. Hanevik HI, Hilmarsen HT, Skjelbred CF, et al. A single nucleotide poly- morphism in BMP15 is associated with high response to ovarian stimu- lation. Reprod Biomed Online 2011; 23:97.
6. Cappa F, Pasqua C, Tobia M, Ventura T. [Ascites and hydrothorax due to endogenous hyperstimulation of H.C.G. in a case of hydatidiform mole destruens with secondary irreversible kidney insufficiency due to disse- minated intravascular coagulation]. Riv Ital Ginecol 1976; 56:363.
7. Zalel Y, Katz Z, Caspi B, Ben-Hur H, Dgani R, Insler V. Spontaneous ovari- an hyperstimulation syndrome concomitant with spontaneous pregnancy in a woman with polycystic ovarian disease. Am J Obstet Gynecol 1992; 167: 122-124.
8. Akbay E., Uzunçakmak C., İdil NS, Akçiğ Z., Özel G., Yaşar L A. Recur- rent Spontaneous Ovarian Hyperstimulation Syndrome with Hypothyroi- dism: A Case Report, Bakırköy Tıp Dergisi 2010;6:42-45

9. Wiser A, Levron J, Kreizer D, et al. Outcome of pregnancies complicated by severe ovarian hyperstimulation syndrome (OHSS): a follow-up be- yond the second trimester. Human Reproduction. 2004;20:910-4.
10. Tokmak A, Guzel AI, Kangal K, Yalinkaya A. Spontaneous Ovarian Hy- perstimulation Syndrome in a Term Pregnancy. Cukurova Med J. 2015; 40(3): 623-626.