Eftekhar, Z.

Rahimi-Moghaddam, P.

Tahmasbi, M.

Yarandi, F.

eprints Iran University of Medical Sciences

Full Terms & Conditions of access and use can be found athttp://www.tandfonline.com/action/journalInformation?journalCode=ijog20Download by: [Universite Laval] Date: 08 November 2015, At: 00:08Journal of Obstetrics and GynaecologyISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20An ovarian torsion in severe spontaneous ovarianhyperstimulation syndrome associated with asingleton pregnancyZahra Eftekhar, Parvaneh Rahimi-Moghaddam, Fariba Yarandi & MamakTahmasbiTo cite this article: Zahra Eftekhar, Parvaneh Rahimi-Moghaddam, Fariba Yarandi & MamakTahmasbi (2005) An ovarian torsion in severe spontaneous ovarian hyperstimulation syndromeassociated with a singleton pregnancy, Journal of Obstetrics and Gynaecology, 25:4, 393-394To link to this article:  http://dx.doi.org/10.1080/01443610500135636Published online: 02 Jul 2009.Submit your article to this journal Article views: 20View related articles An ovarian torsion in severe spontaneous ovarian hyperstimulationsyndrome associated with a singleton pregnancyZAHRA EFTEKHAR1, PARVANEH RAHIMI-MOGHADDAM2, FARIBA YARANDI1, &MAMAK TAHMASBI11Department of Gynecologic Oncology, Mirza-Koochak-Khan Hospital, Tehran University of Medical Sciences and 2RaziInstitute for Drug Research, Iran University of Medical Sciences, Tehran, IranIntroductionOvarian hyperstimulation syndrome (OHSS) is a life-threateningcondition, usually associated with pharmacological ovarian stimu-lation. Spontaneous OHSS is a rare condition, usually appearing inmultiple gestation, hypothyroidism or polycystic ovarian syndrome(Baksu et al. 2004). Pharmacologically-induced or spontaneous,OHSS is characterized by ovarian enlargement along with a ﬂuidshift into extravascular spaces causing ascites, pleural and/orpericardial effusion, hypovolemia and oliguria. A clear pathogen-esis of this condition is still not understood. We report a case ofspontaneous OHSS in a singleton pregnancy complicated withovarian torsion. Another similar case has been reported recently(Baksu et al. 2004).Case reportA 30-year-old woman, gravida 5, para 3, +1, was admitted at 11weeks gestation because of abdominal pain for 1 week. Hermedical history was unremarkable. Her last menstrual period(LMP) occurred 11 weeks before admission. Her previouspregnancies were spontaneous. The abdomen was severelydistended and diffusely tender. Vaginal examination revealed anormal cervix, an enlarged uterus in size of 10 – 11 weeks gestationand enlarged ovaries (15 cm each), which were tender.Laboratory results included: human chorionic gonadotropin(hCG) level; 19 500 mIU/ml, total protein; 4.2 g/dl (normal: 6 –8 g/dl), albumin; 2.3 g/dl (normal: 3.5 – 5.5 g/dl); alpha fetopro-tein (AFP) 54.0 ng/ml; carcino-embryonic antigen (CEA)5 1 ng/ml; carcinogenic antigen 125 (Ca 125): 295 U/ml (normalrange: 0 – 35 U/ml).Ultrasonography revealed a single live intrauterine 10-weekgestation with bilateral multilobular ovarian cysts that had smoothsurfaces and clear contents. The right ovary was 1406 148 mm insize and the left ovary was 137 6 97 mm. Fluid in pelvis andabdomen (ascites) was detected (Figure 1).She was maintained at bed rest with ﬂuid restriction. Due todifﬁculty in breathing, a diagnostic/therapeutic paracentesis wasdone. The extracted ﬂuid proved to be exudative, the microbialcultures were negative and no malignant cells were found.Because of hypoalbuminemia, she received 100 g albumin. Shewas discharged after 35 days with a live 15-week fetus. The rightovary decreased in size to 766 74 mm and the left to 796 96 mm.There were no ascites in the time of discharge. Total protein andalbumin increased to 9.1 g/dl and 4.8 g/dl, respectively.After 2 weeks, she was re-admitted with complaints of rightlower abdomen pain and tenderness along with nausea andvomiting. In physical examination, the right lower quadrant ofthe abdomen was severely tender with rebound tenderness.Surgical exploration was carried out with a primary diagnosis ofright ovarian torsion. In operation, it was observed that the rightovary was twisted through 1808. The ovary was unwound andseveral large ovarian cysts were aspirated. The patient wasdischarged 1 week after surgery. Her condition was then checkedevery week for 1 month and followed at normal prenatal check-ups. She had a normal delivery of a healthy female infant weighing3100 g at term.DiscussionThe complex clinical picture of severe ovarian hyperstimulation isan infrequent but well-described condition associated withFigure 1. Ultrasonographic appearance of (a) an intrauterinesingleton pregnancy and (b) an ovarian enlargement associatedwith multilubolar ovarian cystic structure.Obstetric case reports 393Downloaded by [Universite Laval] at 00:08 08 November 2015 ovulation induction especially in patients with polycystic ovariansyndrome, probably because of a higher sensitivity of follicles togonadotropin stimulation (Baksu et al. 2004). Mild forms areinfrequently associated with spontaneous ovulation and concep-tion, primarily in the cases of multiple-pregnancies. Severe formsof this syndrome have been reported in severely hypothyroidpregnant women. This might be due to the fact that thyroidstimulating hormone (TSH) and follicle-stimulating hormone(FSH) can bind to each other’s receptor and in hypothyroidismTSH is increasing signiﬁcantly. Also, hCG plays a crucial role inthe development of OHSS. Several cases of OHSS have beenassociated with multiple pregnancies or hydatidiform moles,conditions with very high levels of hCG. It has been shown thathCG can stimulate TSH and FSH receptors in vitro. A recentstudy identiﬁes a mutant follitropin receptor responsible for OHSS(Smits et al. 2003).OHSS rarely happens in a singleton pregnancy in womenwith no underlying disease. Only a few such cases havebeen reported (Baksu et al. 2004; Abu-Louz et al. 1997). Thisrare condition should be kept in mind in any pregnantwoman showing clinical features of OHSS. The possibility ofovarian rupture and torsion should always be considered inthese patients. If surgery is necessary, only homeostatic measuresshould be undertaken and if torsion of the adnexa isencountered, unwinding the adnexa is possible, even when theadnexae are already ischemic.ReferencesAbu-Louz SK, Ahmed AA, Swan RW. 1997. Spontaneous ovarianhyperstimulation syndrome with pregnancy. American Journalof Obstetrics and Gynecology 177:476 – 477.Baksu A, Baksu B, Goker N. 2004. Laparoscopic unwinding andcyst aspiration of an ovarian torsion in spontaneous ovarianhyperstimulation syndrome associated with a singleton preg-nancy. Australian New Zealand Journal of Obstetrics andGynaecoogy 44:270 – 272.Smits G, Olatunbosun O, Delbaere A, Pierson R, Vassart G,Costagliola S. 2003. Ovarian hyperstimulation syndrome due toa mutation in the follicle-stimulating hormone receptor. NewEngland Journal of Medicine 349:760 – 766.Correspondence to: Z. Eftekhar, Division of Oncology, Department of Obstetrics and Gynecology, Mirza Koochak Khan Hospital,Nejatollahi St., Karim Khan Zand Ave., Tehran, Iran. Tel: + 98 21 8904172; Fax: + 98 21 8904172; E-mail: par127@mail.usask.caDOI: 10.1080/01443610500135636Ovarian hyperstimulation associated with a spontaneous pregnancyS. O¨ZDEN, B. GU¨RBU¨Z, S. YALTI, B ERGU¨L, & M. O¨ZTURKMENZeynep Kamil Women and Children Education and Research Hospital, Istanbul, TurkeyCase reportA 19-year primigravidida was admitted to the hospital because ofabdominal pain, bloating and mild brown vaginal discharge. Shehad an 12-weekperiod of amenorrhea. She denied the recent use ofan ovulation inducing drug. There were no signs of chronicanovulation, hyperandrogenism or hypothyroidism. She had ahistory of regular menses.Laboratory testing showed normal haemoglobin (12.1 g/dl),hematocrit (35.6%), BUN (10 mg%), creatinine (0.6 mg%), uricacid (3.4 mg%), thyroxine 8.0 ng/ml, TSH 1.0 mU/ml levels andslight hyponatremia (133 mEq/ml). Triiodotyronine resin uptakewas 24%. Liver function tests were normal.Transvaginal ultrasonography showed a 7 weeks’ gestationwithout cardiac activity (Figure 1). There was massive ascites inthe abdomen (Figure 2). Enlarged ovaries measuring1026 108 mm on the right and 996 80 mm on the left sideswere seen to contain a great number of cysts between 15 mm and40 mm (Figure 3).Dilatation and curettage was performed because of a missedabortion.Histopathological examination of uterine contents showed noevidence of gestational trophoblastic disease. The patient wasdischarged from hospital after 3 days of hospitalization advisingweekly examination. Unfortunately, she was admitted to a privatehospital and the ascites and ovarian cysts were aspirated atlaparotomy. She was invited to our hospital 7 days after dischargefrom this hospital. Ascitie ﬂuid and ovarian cysts had disappeared.Ovarian diameters were normal.DiscussionOvariant hyperstimulation syndrome (OHSS) is characterized by acontinuum of disorders ranging from a mild set of ﬁndings to asevere and potentially life-threatening entity and is most oftenassociated with the administration of exogenous gonadotropins.OHSS is very rarely seen in pregnancy where ovulatory drugs orFigure 1. Transvaginal ultrasonography showing 7 weeks’ gestationwithout cardiac activity.394 Obstetric case reportsDownloaded by [Universite Laval] at 00:08 08 November 2015 

An ovarian torsion in severe spontaneous ovarian hyperstimulation syndrome associated with a singleton pregnancy

http://eprints.iums.ac.ir/12164/1/Journal%20of%20Obstetrics%20%26%20Gynaecology%20Volume%2025%20issue%204%202005%20%5Bdoi%2010.1080_01443610500135636%5D%20Eftekhar%2C%20Zahra%3B%20Rahimi-Moghaddam%2C%20Parvaneh%3B%20Yarandi%2C%20Fariba%3B%20Ta%20--%20An%20ovarian%20torsion%20in%20severe%20spontaneou.pdf

An ovarian torsion in severe spontaneous ovarian hyperstimulation syndrome associated with a singleton pregnancy

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