An unusual case of heterotopic twin pregnancy ... - Wiley Online Library

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Apr 20, 2004 - Wedge-shaped defects of the uterine incision follow- ing Cesarean .... †Department of Perinatology, S üleymaniye Maternity. Hospital for ...
Ultrasound Obstet Gynecol 2004; 23: 626–628 Published online in Wiley InterScience (www.interscience.wiley.com).

Letters to the Editor An unusual case of heterotopic twin pregnancy managed successfully with selective feticide Wedge-shaped defects of the uterine incision following Cesarean section are a well-known phenomenon1 – 8 . Though the relevance of these defects is still uncertain, they have been associated with severe complications including spontaneous or instrumental rupture, abnormal placentation, menorrhagia and dysmenorrhea1 . Implantation of the conceptus within a Cesarean section scar is considered to be the rarest and one of the most dangerous types of ectopic pregnancy. We report the case of a heterotopic twin pregnancy with one fetus located at the fundus uteri and the other within the Cesarean section scar in the cervico-isthmic region. A 23-year-old woman, gravida 2 para 1, with a previous Cesarean delivery was referred because of the abnormal location of one of the two gestational sacs of a twin pregnancy. She had conceived without any kind of infertility therapy. There was scant bleeding, but no pelvic pain since the last menstrual period. Standard blood counts and a routine urine analysis were in the normal range. Transvaginal examination (GE Logiq 400 MD, 5-MHz transvaginal probe, GE Medical Systems, Milwaukee, WI, USA) revealed a diamniotic, dichorionic, twin pregnancy. Both fetuses had normal fetal cardiac activity and crown–rump length measurements (4.8 and 4.9 mm) in accordance with 6 + 2 weeks’ gestation. One gestational sac was in the upper fundus, whereas the second one was located in a large tent-shaped incisional defect just above the gaping (7 mm) internal cervical os (Figure 1). The heterotopic placenta at the Cesarean section scar was in close proximity to the maternal bladder. The cervical canal was funnel-shaped and 37 mm in length. The patient opted for a selective termination of the abnormally located fetus, which was accomplished at 7 + 2 weeks’ gestation by intrathoracic injection of 0.5 mL 7.5% potassium chloride (KCl) via a 22G 20-cm needle inserted transvaginally. On follow-up, persistence of placental vascularization and even some growth of the placental mass was noted despite the disappearance of the tiny fetal mass. At 26 + 6 weeks’ gestation a detachment of the abnormally located placenta from its bed without any bleeding was noted. At 30 + 3 weeks’ gestation a sudden rupture of membranes was followed by minimal vaginal bleeding and preterm labor. The baby was delivered by Cesarean section. The placenta at the uterine incision site was completely detached without any significant bleeding from the placental bed. There was no sign of abruption of the normally located placenta. The male baby weighing 1530 g with Apgar scores of 7 and 9 at 1 and 5 min, respectively, was taken to the neonatal intensive care unit for the first 8 days, was discharged

Copyright  2004 ISUOG. Published by John Wiley & Sons, Ltd.

from hospital in good condition and at the time of writing is doing well at home. The mother was discharged 2 days after the operation without any complications. Ectopic pregnancy in a previous Cesarean section scar is a rare phenomenon. A recent review has identified only 19 cases reported in the English language scientific literature since 19662 . Cesarean section scar pregnancy may result in heavy blood loss controllable by drastic measures such as uterine artery ligation, embolization or emergency hysterectomy3 – 5 . Consequently, early detection of Cesarean section pregnancy usually necessitates some kind of intervention to prevent a dramatic course. Though interventions such as laparoscopic evacuation5 or bilateral uterine artery embolization3,4 have been undertaken in some cases of Cesarean scar pregnancy, the current standard is a medical approach usually comprising a full course of maternal methotrexate therapy6 – 8 , which unfortunately is not an option in heterotopic pregnancy. There are few cases of cervical heterotopic pregnancy treated by conservative measures. Elective feticide with KCl in one case was shown to be successful in terminating the heterotopic fetus but placental vascularization remained intact, necessitating a full course of methotrexate in the immediate postpartum period9 . Jozwiak et al. attempted a novel approach using hysteroscopic removal followed by roller-ball coagulation of the bleeding sites. The pregnancy then continued successfully to be terminated by a near-term Cesarean section without any abnormal bleeding10 . Obviously this approach needs an intact internal cervical os to keep the intrauterine part unaffected. In this first case of heterotopic Cesarean section scar pregnancy, selective feticide with KCl proved to be a successful conservative mode of management.

Figure 1 Ultrasound image showing a heterotopic twin pregnancy with one gestational sac located in the fundus uteri and the other within the Cesarean section scar.

LETTERS TO THE EDITOR

Letters to the Editor

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In this era of increasing Cesarean section rates throughout the world, many Cesarean section scar pregnancies will inevitably follow. Consequently we think that the standard informed consent form should list Cesarean section scar pregnancy as a remote but serious complication of Cesarean section. H. F. Yazicioglu*†, S. Turgut‡, R. Madazli§, ¨ ¨ M. Aygun†, Z. Cebi† and S. Sonmez† †Department of Perinatology, Suleymaniye Maternity ¨ Hospital for Research and Training, Suleymaniye Egitim ¨ ve Arastirma Hastanesi, Prof. Siddik Sami Onar Cad. 45/1, Eminon ¨ u, ¨ ‡Meltem Private Maternity Hospital and §Department of Perinatology, Cerrahpasa Faculty of Medicine, Istanbul, Turkey *Correspondence. (e-mail: [email protected]) DOI: 10.1002/uog.1050 Published online 20 April 2004

References 1. Morris H. Surgical pathology of the lower uterine segment Caesarean section scar: is the scar a source of clinical symptoms? Int J Gynecol Pathol 1995; 14: 16–20. 2. Fylstra DL. Ectopic pregnancy within a Cesarean scar: a review. Obstet Gynecol Surv 2002; 57: 537–543. 3. Yang MJ, Jeng MH. Combination of transarterial embolization of uterine arteries and conservative surgical treatment for pregnancy in a Cesarean section scar. A report of 3 cases. J Reprod Med 2003; 48: 213–216. 4. Ghezzi F, Lagana D, Franchi M, Fugazzola C, Bolis P. Conservative treatment by chemotherapy and uterine arteries embolization of a Cesarean scar pregnancy. Eur J Obstet Gynecol Reprod Biol 2002; 103: 88–89. 5. Lee CL, Wang CJ, Chao A, Yen CF, Soong YK. Laparoscopic management of an ectopic pregnancy in a previous Caesarean section scar. Hum Reprod 1999; 14: 1234–1236. 6. Haimov-Kochman R, Sciaky-Tamir Y, Yanai N, Yagel S. Conservative management of two ectopic pregnancies implanted in previous uterine scars. Ultrasound Obstet Gynecol 2002; 19: 616–619. 7. Lam PM, Lo KW. Multiple-dose methotrexate for pregnancy in a Cesarean section scar. A case report. J Reprod Med 2002; 47: 332–334. 8. Nawroth F, Foth D, Wilhelm L, Schmidt T, Warm M, Romer T. Conservative treatment of ectopic pregnancy in a Cesarean section scar with methotrexate: a case report. Eur J Obstet Gynecol Reprod Biol 2001; 99: 135–137. 9. Monteagudo A, Tarricone NJ, Timor-Tritsch IE, Lerner JP. Successful transvaginal ultrasound-guided puncture and injection of a cervical pregnancy in a patient with simultaneous intrauterine pregnancy and a history of a previous cervical pregnancy. Ultrasound Obstet Gynecol 1996; 8: 381–386. 10. Jozwiak EA, Ulug U, Akman MA, Bahceci M. Successful resection of a heterotopic cervical pregnancy resulting from intracytoplasmic sperm injection. Fertil Steril 2003; 79: 428–430.

fluid (Figure 1). This finding was discovered when a 31year-old Caucasian woman presented during her first pregnancy for a second-trimester ultrasound examination that showed a singleton intrauterine pregnancy with a normal and active fetus with measurements consistent with 26 weeks’ gestation. A cyst of about 4 cm in diameter was noted within the amniotic fluid close to the umbilical cord insertion into the placenta. The umbilical cord was morphologically normal with normal blood velocity waveforms. Color flow Doppler ruled out any vascular involvement with the cyst. During our counseling session we mentioned to the patient that (1) the clinical significance of placental surface cysts depends on the association of fetal growth restriction and (2) that most placental surface simple cysts are associated with normal pregnancy outcomes1 . The patient voiced her understanding of this information and agreed to a follow-up ultrasound evaluation and a postpartum examination of the placenta and infant. Ultrasound evaluation at 33 weeks’ gestation demonstrated a fetus with appropriate interval growth. The remaining ultrasonographic findings were

Figure 1 Ultrasonic image showing an anechogenic cystic structure within the amniotic fluid at 26 weeks’ gestation.

Placental surface cyst with contents less echogenic than amniotic fluid on a second-trimester ultrasonographic evaluation We present a case of a placental surface cyst whose contents were less echogenic than the surrounding amniotic

Copyright  2004 ISUOG. Published by John Wiley & Sons, Ltd.

Figure 2 Gross examination of the placenta revealed a surface cyst with a subchorionic fibrin clot. The umbilical cord was normal.

Ultrasound Obstet Gynecol 2004; 23: 626–628.