CASE REPORT Successful management of heterotopic cesarean scar pregnancy combined with intrauterine pregnancy after in vitro fertilization–embryo transfer Chao-Nin Wang, M.D.,a,b Chun-Kai Chen, M.D.,a,b Hsin-Shih Wang, M.D., Ph.D.,a,b Ho-Yen Chiueh, M.D.,a,b and Yung-Kuei Soong, M.D., M.P.H.a,b a
Department of Obstetrics and Gynecology, Chang-Gung Memorial Hospital, and Medicine, Taoyuan, Taiwan
b
Chung Gung University College of
Objective: To present a case of cesarean scar pregnancy combined with intrauterine pregnancy after IVF–embryo transfer. Successful embryo reduction was performed and preserved the normal intrauterine gestation. Design: Case report. Setting: Tertiary referral case center. Patient(s): A woman with cesarean scar pregnancy combined with intrauterine pregnancy after IVF–embryo transfer. Intervention(s): Early diagnosis of heterotopic cesarean scar pregnancy and selective embryo reduction was performed by ultrasound-guided potassium chloride (KCl) directed injection. Main Outcome Measure(s): Successful pregnancy outcome. Result(s): A 38-year-old woman achieve pregnancy by IVF–embryo transfer. Heterotopic cesarean scar pregnancy was diagnosed at 7 weeks gestational age. A transvaginal ultrasound-guided KCl injection was given to terminate the cesarean scar embryo and a healthy infant was delivered 6 months later. Conclusion(s): Heterotopic cesarean scar pregnancy after IVF is extremely rare. Transvaginal intracardiac injection of KCl is a safe and reliable method to terminate the cesarean scar pregnancy. Satisfactory pregnancy outcome should be achieved. (Fertil Steril 2007;88:706.e13–6. 2007 by American Society for Reproductive Medicine.) Key Words: Cesarean scar pregnancy, heterotopic pregnancy, potassium chloride, selective embryo reduction
Cesarean scar pregnancy had been considered as a rare condition but the reported cases in the medical literature have increased. Without proper treatment of cesarean scar pregnancy, it would result in massive hemorrhage and severe maternal morbidity or mortality. At the same time, heterotopic pregnancy also increased in incidence due to the prevalence of assisted reproduction techniques (ART) (1). Cesarean scar pregnancy combined with normal intrauterine pregnancy is extremely rare. The heterotopic gestational sac should be removed carefully and preservation of the intrauterine pregnancy at the same time is a great challenge. We report a successfully treated case by direct injection of potassium chloride (KCl) to terminate a 10-week gestation and preserve the intrauterine ongoing pregnancy. Six months later, the woman delivered a healthy baby, weighing 1,820 g, by cesarean section due to preterm labor and antepartum Received July 3, 2006; revised November 27, 2006; accepted November 30, 2006. Reprint requests: Chun-Kai Chen, M.D., Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, 5, Fu-Hsing Street, Kwei-Shan, Tao-Yuan, 333 Taiwan (FAX: 886-3-3288252; E-mail:
[email protected]).
hemorrhage. At present, there are only two case reports in the medical literature. We provide our experience of this case and the case is compared with those of the literature. A written informed consent document was obtained from the patient before the surgery. The treatment procedure was not against any ethic principles; all data were collected from chart reviews and did not require approval of the Institutional Review Board.
CASE REPORT A 38-year-old woman, gravida 4, para 3, sought IVF treatment as a result of secondary infertility and a previous tubal sterilization during the last cesarean section 13 years ago. She had undergone three lower segment transverse cesarean deliveries before. Poor embryo development was noted during the first treatment cycle. On the second cycle, a total of 13 oocytes were retrieved 36 hours after hCG was administered. Three embryos with good morphology were transferred into the uterus under abdominal ultrasound guidance 52 hours after oocyte retrieval. A pregnancy test was positive 14 days after embryo transfer.
706.e13 Fertility and Sterility Vol. 88, No. 3, September 2007 Copyright ª2007 American Society for Reproductive Medicine, Published by Elsevier Inc.
0015-0282/07/$32.00 doi:10.1016/j.fertnstert.2006.11.192
FIGURE 1 One intrauterine gestational sac implanted at the fundus (T1) and the other sac implanted at anterior cervicoisthmic region with echogenic ring invading into the previous cesarean section scar (T2).
Wang. Cesarean scar and intrauterine pregnancy. Fertil Steril 2007.
Three weeks later, vaginal sonographic examination showed one intrauterine gestational sac at the fundus and the other sac implanted at the anterior cervicoisthmic region with an echogenic ring invading into the previous cesarean section scar. Both gestations had cardiac activity (Fig. 1). After thorough counseling, the couple decided to receive fetal reduction of the cesarean scar pregnancy with an injection of 0.2 mL of KCl at 10 weeks gestational age. Injection guided by vaginal ultrasound was performed through the
FIGURE 2
vaginal route with a 20-gauge needle (Echotip disposable Chiba needle, Cook Ob/Gyn, Spencer, Indiana). She tolerated the procedure well except for a small amount of vaginal bleeding for 1 week. The procedure was successful and the heterotopic pregnancy was terminated as proved by transvaginal ultrasound (Fig. 2). The prenatal course was uneventful although an amorphous mass 3 by 3 by 3 cm could be still detected at the previous cesarean scar site at gestational age of 32 weeks. Three weeks later, a male baby, weighing 1,820 g, was delivered by cesarean section due to preterm labor and tocolysis failure. During the operation, an amorphous mass, measuring 3 by 3 by 2 cm, bulged out immediately after incision of the lower segment of the uterus, which was suggestive of remaining gestational tissue of the scar pregnancy (Fig. 3). The male baby was delivered smoothly and the incised uterus was closed layer by layer. Unfortunately, the active bleeding points from the low segments of the uterus were noted and could not be stopped by suture materials. Massive blood loss, more than 4,000 mL, was noted during cesarean section. Immediate blood transfusion and bilateral internal iliac arteries ligation were performed. Fortunately, the bleeding was stopped and the woman was transferred intubated to the intensive care unit. Twenty-four hours later she was extubated and transferred to the ward and 10 days later, she was discharged from our hospital. DISCUSSION Heterotopic pregnancy is unusual and the incidence is estimated at 1 in 30,000 deliveries (2). Recently, with the improvement of ART, the incidence may have increased to 1% of pregnancies achieved by artificial conceptions (3). Most of the heterotopic pregnancies are tubal pregnancies and conservative or surgical interventions terminate the ectopic gestational sac and preserve the intrauterine gestations.
The cesarean scar gestational sac was without fetal heart beat after KCl injection (T2). The intrauterine pregnancy was ongoing (T1).
FIGURE 3
Wang. Cesarean scar and intrauterine pregnancy. Fertil Steril 2007.
Wang. Cesarean scar and intrauterine pregnancy. Fertil Steril 2007.
Fertility and Sterility
The remaining gestational mass (arrow) bulged from the low segment incision wound during cesarean section.
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TABLE 1 Review of successful management of heterotropic cesarean scar pregnancy. Reference no.
Author
Year
Case no.
ART
Treatment
Pregnancy outcome
8
Salomon et al.
2003
1
KCl injection
9
Hsieh et al.
2004
1
IVF-embryo transfer IVF-embryo transfer
Successful cesarean section delivery Ongoing pregnancy
Fine needle aspiration
Wang. Cesarean scar and intrauterine pregnancy. Fertil Steril 2007.
However, if the ectopic gestational sac has implanted at the previous cesarean scar defect, the cesarean scar gestational sac may result in massive vaginal bleeding and risk of uterine rupture. Conservative treatment of cesarean scar pregnancy had been reported in the medical literature. An injection of methotrexate (MTX) could treat the scar pregnancy successfully (4, 5). The heterotopic cesarean scar pregnancy was different because we had to preserve the normal intrauterine pregnancy. Pegel et al. (6) described the first case of MTX injection to terminate intrauterine and cervical pregnancies. The normal intrauterine pregnancy cannot be preserved. Furthermore, the embryo toxicity and potential teratogenicity of MTX to the intrauterine fetus was still under discussion (7). Two successful management of heterotopic cesarean scar pregnancies have been reported in medical literature (Table 1). Salomon et al. (8) reported the first heterotopic cesarean scar pregnancy combined with an intrauterine pregnancy, which successfully terminated the ectopic gestational sac by KCl administration by transvaginal ultrasound guidance. The remaining normal implanted gestational development was ongoing and a healthy girl, weighing 2,800 g, was delivered by cesarean section. An amorphous bulging mass, measuring 3 by 3 cm was found in the lower segment. The mass was composed of placental and deciduous tissue but no remaining embryo. Hsieh et al. (9) reported another heterotopic cesarean scar pregnancy combined with intrauterine pregnancy and successfully preserved the normal gestational sac by needle aspiration of the embryo, which located at the cesarean scar defect. This gestation was also achieved by IVF–embryo transfer and the remaining gestational pregnancy was preserved. In our case, the cesarean scar pregnancy was terminated carefully by a KCl injection and the intrauterine pregnancy was successfully preserved until the third trimester. The serial follow-up ultrasound still revealed the reluctant placenta tissue shadows and suggested possible blood circulation. Massive vagina bleeding occurred during the operation. The remaining trophoblastic tissue over the lower segments of the uterus was blamed for the massive blood loss. Gyamfi et al. (10) also described massive bleeding from the retained trophoblastic tissue, which occurred during 706.e15
Wang et al.
cesarean section, after successful fetal reduction of a cervical heterotopic pregnancy by KCl injection. Therefore, the retained gestational tissue at early trimester should be carefully followed up by ultrasound. The possibility of persisting blood flow and uncontrolled bleeding during delivery may occur. A question is raised whether the cesarean scar pregnancy could be avoided by improving methods of embryo transfer back to the uterine cavity. Ultrasound-guided embryo transfer has been helpful to lessen the chance of cesarean scar pregnancy. In this case, the ectopic pregnancy occurred anyway. Nevertheless, the incidence of cesarean scar pregnancy is too low to make any comments at present. In conclusion, the cesarean section rate has increased and ART is more popular and mature in this decade. The incidence of heterotopic pregnancy has increased and heterotopic cesarean scar pregnancy will still occur in the future. Improvement in ultrasonography makes the early diagnosis of this rare condition possible. After an early diagnosis, a KCl injection is a reliable method to terminate the heterotopic gestational sac and preserve the intrauterine pregnancy at the same time. There were no significant side effects with the normal gestational sac and the ectopic gestational embryo can be reduced smoothly. The retained gestational tissue with its abundant supplied vessels probably contributed to the massive uterine bleeding during delivery. Therefore, intensive management and preparation before the operation should be kept in mind and the possible complication of a massive blood loss could be avoided and a satisfactory pregnancy outcome acheived.
REFERENCES 1. Abusheikba N, Salha O, Brinssden P. Extrauterine pregnancy following assisted conception treatement. Hum Reprod Update 2000;6:80–2. 2. Leveno KJ, Cunningham FG, Gant NF, Gilstrap LC, Hauth JC, Wenstrom KD, eds. Williams obstetrics. 21st edition. New York: McGraw-Hill, 2001. 3. Molloy D, Deambrosis W, Keeping D, Hynes J, Harrison K, Hennessey J. Multiple-sited (heterotopic) pregnancy after in vitro fertilization and gamete intrafallopian transfer. Fertil Steril 1990;53:1068–71. 4. Lam PM, Lo KW. Multiple-dose methotrexate for an ectopic pregnancy in a cesarean section scar. A case report. J Reprod Med 2002; 47:332–4. 5. Seow KM, Cheng WC, Chuang J, Lee C, Tsai YL, Hwang JL. Methotrexate for cesarean scar pregnancy after in vitro fertilization and embryo transfer. A case report. J Reprod Med 2000;45:754–7.
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6. Peleg D, Bar-Hava I, Neuman-Levin M, Ashkenazi J, Ben-Rafael Z. Early diagnosis and successful nonsurgical treatment of viable combined intrauterine and cervical pregnancy. Fertil Steril 1994;62: 405–8. 7. Timor-Trisch IE. Is it safe to use methotrexate for selective injection in heterotopic pregnancy? Am J Obstet Gynecol 1998;178: 193–4. 8. Salomon LJ, Fernadez H, Chauveaud A, Doumerc S, Frydman R. Successful management of a heterotopic Caesarean scar pregnancy: potas-
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sium chloride injection with preservation of the intrauterine gestation. Case report. Hum Reprod 2003;18:189–91. 9. Hsieh BC, Hwang JL, Pan HS, Huang SC, Chen CY, Chen PH. Heterotopic Caesarean scar pregnancy combined with intrauterine pregnancy successfully treated with embryo aspiration for selective embryo reduction. Case report. Hum Reprod 2004;19:285–7. 10. Gyamfi C, Cohen S, Stone J. Maternal complication of cervical heterotopic pregnancy after successful potassium chloride fetal reduction. Fertil Steril 2004;82:940–3.
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