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Hemorrhagic Corpus Luteum Cyst Torsion in Term Pregnancy: a Case Report

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Hemorrhagic Corpus Luteum Cyst Torsion in Term Pregnancy: a Case Report Hemorrhagic corpus luteum cyst torsion HEMORRHAGIC CORPUS LUTEUM CYST TORSION IN TERM PREGNANCY: A CASE REPORT Pao-Hui Huang, Kun-Bow Tsai,1 Eing-Mei Tsai, and Jinu-Huang Su Departments of Obstetrics and Gynecology and 1Pathology, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan. Hemoperitoneum during pregnancy resulting from spontaneous rupture of adnexal torsion is a rare cause of fetal and maternal death. Presenting symptoms include severe abdominal pain, followed rapidly by maternal shock and fetal distress. It is hard to localize the adnexae in advanced pregnancy. Here, we present a case of spontaneous rupture of hemorrhagic corpus luteum cyst torsion that had not been previously diagnosed by ultrasound during term pregnancy. The patient was sent to our emergency room for sudden onset of severe low abdominal pain. Treatment consists of maintenance of adequate circulating intravascular volume and rapid surgical intervention. Preoperative diagnosis of adnexal torsion during term pregnancy is very difficult, although it is always identified during surgery. Key Words: hemorrhagic corpus luteum, cyst torsion, term pregnancy (Kaohsiung J Med Sci 2003;19:75–8) Adnexal torsion is a rare occurrence during pregnancy. emergency service with the chief complaint of acute It has been described as a severe complication of abdominal pain and loss of fetal movement for 4 hours. ovarian hyperstimulation syndrome and after ovarian There was no history of vaginal bleeding, abdominal stimulation for in vitro fertilization (IVF), with the trauma, fever, vomiting, headache, blurring of vision, highest incidence during the first trimester [1]. It is or oliguria. Physical examination showed that her rare during the second trimester [2] and exceptional consciousness was clear and she was coherent and during the third trimester [3]. Adnexal torsion is afebrile but irritable, with a pulse rate of 140/minute, generally diagnosed during surgery. A case of blood pressure of 90/60 mmHg, and marked pallor. hemoperitoneum during term pregnancy resulting The uterus was of term size, tense and tender, with a from rupture of hemorrhagic corpus luteum cyst well-defined contour and no fetal heart beat. Abdomi- torsion is presented in this report. nal ultrasound confirmed intrauterine fetal death (IUFD) with the placenta in the upper uterine segment and no retroplacental blood clot. The adnexae and cul- CASE PRESENTATION de-sac were poorly identified. Laparotomy was per- formed immediately for acute abdominal pain with A 35-year-old female patient, gravida 2, para 0, who term pregnancy. At laparotomy, right adnexal torsion had had uneventful antenatal care, presented at the and internal bleeding were observed (Figure 1). Blood was found in the peritoneum (1,200 mL). Right salpingo-oophorectomy was performed and the left Received: September 17, 2002 Accepted: November 26, 2002 ovary was unwound and preserved. A lower segment Address correspondence and reprint requests to: Dr. Pao-Hui cesarean section was then performed and a fresh Huang, Department of Obstetrics and Gynecology, Kaohsiung st stillborn female weighing 3,800 g was delivered. The Medical University Hospital, 100 Shih-Chuan 1 Road, Kaohsiung City 807, Taiwan. placenta was fundal in location with no retroperitoneal E-mail: [email protected] blood clot and the uterus was intact. Four units of Kaohsiung J Med Sci February 2003 • Vol 19 • No 2 75 P.H. Huang, K.B. Tsai, E.M. Tsai, and J.H. Su blood were transfused intraoperatively. The postop- vary depending on whether the rotation is sudden and erative period was uneventful. The removed ovary complete or progressive. With progressive torsion, weighed 65.1 g and measured 13 x 11 x 5.5 cm. Grossly, lymphatic drainage is compromised, which leads to it was brown and coated with blood clots. It contained massive enlargement of the ovary due to lymphatic multiple cysts, a nodule, and diffuse hemorrhage. The edema [13]. This is followed by venous obstruction fallopian tube measured 6.1 cm in length and 0.8 cm in and hemorrhagic infarction [14]. The final step is diameter. Grossly, it was brown-black. interruption of the arterial blood supply, which may Microscopically, there was intense hemorrhagic inf- result in gangrene, infection, peritonitis, and possible arction of the ovary (Figure 2): there were multiple death [15]. Rupture of adnexal torsion during degenerated or destructed cysts, including multiple pregnancy may also occur secondary to softening of hemorrhagic corpus luteums and several follicular the lesion following stroma decidualization [16]. cysts. There were aggregates of inflammatory cells. Therefore, the clinical presentation is variable, rang- The fallopian tube revealed congested vessels and ing from a benign asymptomatic state to circulatory focal hemorrhage (Figure 3). collapse. Mild cases require observation, while, in severe cases, laparotomy is mandatory. The sonographic detection rate of adnexal masses during DISCUSSION pregnancy is approximately 1% [17]. Although the Adnexal torsion is an uncommon cause of surgical emergency [3]. It usually occurs during the reproductive years and represents 2.7% of all gynecologic emergencies [3]. Its incidence is 1 in 5,000 during pregnancy, occurring more frequently in the first trimester after IVF and ovarian stimulation as treatment for infertility [3]. The proportion of adnexal torsion at the outset of pregnancy varies from 18% to 28.7%, according to previously reported series [4–12]. The condition is rare during the second trimester [2] and exceptional during the third trimester [3]. Adnexal torsion consists of total or partial rotation of the adnexa around its vascular axis. Clinical findings Figure 2. Cystic changes in the corpus luteum with intense hemorrhagic infarction. (Hematoxylin & Eosin, original magnification, x 8) Figure 1. Both right ovary and right fallopian tube are twisted. The right ovary appears as an enlarged cystic mass with a ruptured hole (arrow). An area of hemorrhagic infarction is evident (arrowhead), as are hemoperitoneum and blood clot (white Figure 3. Marked congestion and hemorrhage in the fallopian arrow). tube. (Hematoxylin & Eosin, original magnification, x 4) 76 Kaohsiung J Med Sci February 2003 • Vol 19 • No 2 Hemorrhagic corpus luteum cyst torsion mass had been followed sonographically, the growing Steril 1994;61:383–5. uterus made detection of the persisting mass 3. Mancuso A, Broccio G, Angio L. Adnexal torsion in pregnancy. Acta Obstet Gynecol Scand 1997;76:83–4. impossible. The management of such sonographically 4. Hibbard LT. Adnexal torsion. Am J Obstet Gynecol 1985;152: identified adnexal masses in the gravid patient re- 456–61. mains controversial [18]. Torsion is seen two or three 5. Lee CH, Raman S, Sivanesaratnam V. Torsion of ovarian times more frequently in the right fallopian tube than tumors: a clinicopathologic study. Int J Gynecol Obstet 1989;28: in the left [19], which is consistent with our case. This 21–5. is thought to be due to the prevention of torsion by the 6. Shalev E, Peleg D. Laparoscopic treatment of adnexal torsion. Surg Gynecol Obstet 1993;176:448–50. sigmoid colon on the left side or to slow venous flow 7. Zweizig S, Perron J, Grubb D, et al. Conservative management on the right side, which may result in congestion [19]. of adnexal torsion. Am J Obstet Gynecol 1993;168:1791–5. This case illustrates the importance of considering 8. Oelsner G, Bider D, Goldenberg M, et al. Long-term follow-up adnexal torsion in the differential diagnosis of of the twisted ischemic adnexa managed by detorsion. Fertil abdominal pain in term pregnancy, especially when Steril 1993;60:976–9. ultrasound examination reveals no specific finding. 9. Chapron C, Capella-Allouc S, Dubuisson JB. Treatment of adnexal torsion using operative laparotomy. Hum Reprod 1996; Thus, ultrasonography is not always useful in the 11:998–1003. detection of adnexal masses, especially in term 10. Baker TE, Copas PR. Adnexal torsion: a clinical dilemma. J pregnant women. Since the rupture of the twisted Reprod Med 1995;40:447–9. ovarian corpus luteum cyst leads to massive internal 11. Morice P, Louis-Sylvestre C, Chapron C, Dubuission JB. bleeding, IUFD may be caused by acute hemodynamic Laparoscopy for adnexal torsion in pregnant women. J Reprod changes in the mother in shock. Ultrasound Med 1997;42:435–9. 12. Bromley B, Benaceraf B. Adnexal mass during pregnancy: examination did not disclose the right tortuous adnexal accuracy of sonographic diagnosis and outcome. J Ultrasound mass and hemoperitoneum because both a poorly Med 1997;16:447–52. distended bladder caused by anuria in shock and an 13. Minor MR, Livolsi VA. Massive edema of the ovary. Am J Dis enlarged uterus in term pregnancy attenuated the Child 1997;131:1295–8. resolution of the ultrasound. Rapid diagnosis of this 14. Hinshaw DB, Kugel AI. Torsion and infarction of the normal ovary: a cause of the acute abdomen. Am J Dis Child 1956;92: rare complication is essential because these patients 57–9. are often first seen in the emergency service. Aggressive 15. Nissen ED, Kent DR, Nissen SE, Feldman BM. Unilateral fluid and blood replacement together with prompt tuboovarian autoamputation. J Reprod Med 1997;19:151–3. surgical intervention provide the only chance for a 16. Bider D, Mashiach S, Dulitzky M, et al. Clinical, surgical and favorable outcome for both mother and child. Even pathologic finding of adnexal torsion in pregnant and non- with these interventions, fetal mortality remains high pregnant women. Surg Gynecol Obstet 1991;173:363–6. 17. Nelson MJ, Cavalieri R, Graham D. Cysts in pregnancy at 31% [20]. Our patient underwent definitive surgical discovered by sonography. J Clin Ultrasound 1986;14:509–12. treatment within hours of the initial presentation. 18. Hogston P, Lilford RJ. Ultrasound study of ovarian cysts in pregnancy: prevalence and significance. Br J Obstet Gynaecol 1986;93:625–8. REFERENCES 19. Vierhout ME, Wallenburg HC. Torsion of the fallopian tube: a case report of a bilateral non-simultaneous torsion and a 1.
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