OVARIAN HYPERSTIMULATION SYNDROME (OHSS) IN A SPONTANEOUS SINGLETON PREGNANCY

A .Amini MD, F. Kheshti MD, M.H. Badakhsh MD

Department of Obstetrics and Gynecology, Iran University of Medical Science and Health Services, Tehran, Iran

Case Report

A 24-year old primiparous woman was hospitalized with severe abdominal pain and dyspnea. The pain started 10 days prior to admission and increased 3-4 days prior to referral. She did not have any previous medicosurgical disease and her gynecological history was unremarkable. No medication was administered prior to admission.

Menarche commenced at the age of 13 and the subsequent menstrual cycles were regular, with mild dysmenorrhea. Her last normal menstural period was 7 weeks prior to admission. She was a well-developed, well-nourished 77kg woman in acute distress and moderate dyspnea. Temperature, pulse rate, and blood pressure were within normal limits. Her physical examination revealed a moderately distended, tender and tense abdomen with positive shifting dullness on percussion. The bowel sounds were normal. Upon pelvic examination the patient had a mild clear cervical discharge with no signs of pregnancy. Uterus and ovaries could not be palpated because of severe distention and tenderness. Pelvic sonography revealed an intrauterine single viable fetus of 7- weeks gestation according to crown-rump length measurement. Both ovaries were enlarged, polycystic, and measured 15x13 cm and 18x14 cm respectively, each containing 12-13 enlarged follicles. A significant amount of free fluid was observed in the abdomen and pelvis. The Liver and kidneys were normal.

Paraclinical study revealed: Hb; 15.1 gr/dl, Hct; 50.1%, WBC; 12700, Na;137 mEq/l, K; 5 mEq/l, BUN; 15 mg/dl, Creatinine 0.6 mg/dl, FBS; 95 mg/dl, AST; 46 U/l, ALT; 21 U/l alkaline phosphatase; 104 U/lit. Total protein; 5.6 g/dl, albumin 3.2 g/dl, total bilirubin 0.5 mg/dl and direct bilirubin 0.1 mg/dl.

PT, PTT, and thyroid function tests were normal. Beta HCG was 4900 U/l (RIA). The 24-hr urine out-put was 1120 cc. Chest X-ray and ECG were normal.

The patient was given indomethacin orally at a daily dose of 75 mg for 20 days. Intravenous albumin and Furosemide were also prescribed as deemed necessary during her 26 days of hospitalization. Repeated paracenteses were done to alleviate severe dyspnea and abdominal distension. During hospitalisation she also received low dose heparin prophylaxis. Resolution of the fluid and decrease in follicular size occurred gradually as demonstrated by clinical and ultrasonographic studies. The patient was discharged from hospital after 26 days weighting 66kg while presenting the following chemical profile: Hb; 11.5 g/dl, Hct; 35.9%, Na; 139 mEq/l and K; 3.8 mEq/l. Three months later, at 20 weeks of gestation, ultrasonography revealed normal sized ovaries and there was no free fluid in the abdomen and pelvis. The fetus was compatible with 20 weeks of gestation and the placenta was grade I and posteriorly located. Amniotic fluid was also within normal limits.

The pregnancy proceeded normally and a healthy infant girl was born at 38 weeks of gestation through normal vaginal delivery.

Discussion

We report a case of OHSS occuring spontaneously in association with a pregnancy of 7-week gestation. The patient did not receive any medication, and no drugs were given for ovulation induction.

Diagnosis was further confirmed by complete resolution of the ovarian hyperstimulation, as demonstrated by clinical and ultrasonography and other laboratory data.

Glatunbosun et al ¹, in 1996 reported three cases of spontaneous OHSS associated with pregnancy as the first cases to result in live birth. Other cases associated with spontaneous pregnancy have been reported by Rosen and Lew ².

Zaley and colleagues ³ described a case of OHSS associated with spontaneous pregnancy in a woman with polycystic ovarian disease. Rotmensch and Scommegna ⁴ reported another case of spontaneous OHSS in an ovulatory non- pregnant patient with trisomy 21 and hypothyroidism.

The etiology and pathophysiologic characteristics of OHSS are poorly understood. Various factors including estrogen, histamine, prostaglandins, aldosterone, renin, angiotensin II have been implicated in the development of this condition. Recent studies show high renin-like activity and elevated angiotension II immunoreactivity in both plasma and ascitic fluid (angiotension II being 6-9 fold higher than plasma). These findings are in favor of ovarian origin of the elevated renin-like activity and angiotensin II immunoreactivity in ascitic fluid of severe OHSS and suggest a stimulatory role of hCG on the ovarian renin-angiotensin system during severe OHSS.

Angiotensin II probably does not cause ascites in severe OHSS, though it may contribute to its maintenance. The efficacy of paracentesis during severe OHSS could be explained at least partially by the removal of great amounts of angiotensin II from the peritoneal cavity. Both antihistamines and Indomethacin have been demonstrated to ameliorate the hyperstimulation in animal studies, but their efficacy in human pregnancy are unknown. ^5-9

One third of the patients developing OHSS after IVF-ET had no previous risk criteria. Exogenous and/or endogenous hCG is suggested as an etiologic factor. ¹⁰

In pregnant patients with severe OHSS, treatment with low dose heparin through the first trimester to prevent the serious complications such as thrombosis and thrombo-embolism, should be considered. ¹¹

Isik and colleagues ¹² believe that human albumin is effective in prevention of moderate to severe OHSS.

References

1. Glatunbosun O A, Gilliland B, Brydon L A, et al. Spontaneous OHSS in four consecutive pregnancy. Clin Exp Obstet Gynecol 1996; 23(3); 127-32.
2. Rosen G F, Lew MW. Severe ovarian hyperstimulation in a spontaneous singleton pregnancy. Am J Obstet Gynecol 1991; 165(5pt1): 1312-3.
3. Zale Y, Katz Z, Caspi B. Spontaneous ovarian hyperstimulation syndrome concomitant with spontaneous pregnancy in a woman with polycystic ovarian disease. Am J Obstet Gynecol 1992; 167: 122-4.
4. Rotmensch S, Scommegna A. Spontaneous OHSS associated with hypothyroidism. Am J Obstet Gynecol 1989; 160: 1220-2.
5. Delbaere A, Bergmann P J, Gervy-Decoster C, et al. Increase angiotensin II in ascites during severe ovarian hyperstimulation syndrome: Role of early pregnancy and ovarian gonadotropin stimulation. Fertil Steril. 1997; 7(6): 1038-45.
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7. Golan A, Ron-el R, Herman A, et al. Ovarian hyper-stimulation syndrome: An update review. Obster Gynecol Surv 1989; 44: 430-40.
8. Itskovitz Eldor J, Kol S, Lewit N, Sealey JE. Ovarian origin of plasma and peritoneal fluid Prorenin in Early Pregnancy and in Patients with OHSS. J Clin Endocrinol Metab 1997; 82(2): 461-4.
9. Speroff L, Glass RH, Kase NGI. Induction of ovulation. In: Speroff L, Clinical Gynecologic Endocrinology and Infertility 5^th ed. Lippincott Williams & Wilkins 1994.
10. Delvigne A, Randromme J, Leroy F. Unpredictable case of complicated ovarian hyperstimulation in IVF. Int J Fertil Women Med 1997; 42(4): 264-7.
11. Hignett MI, Spence JE, Claman P. Internal jugular vein thrombosis. A late complication of OHSS despite mini-dose heparin Prophylaxis. Hum Report 1995; 10(12): 312-13.
12. Isik Az, Gokmen O, Zeynelogin HB, et al. Intravenous albumin prevents moderate-severe OHSS in IVF patient: a prospective randomized and controlled study. Eu J Obstet Gynecol Report Biol 1996; 70(2): 179-83.

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