Hysteroscopic management of heterotopic cesarean scar pregnancy

CASE REPORT Hysteroscopic management of heterotopic cesarean scar pregnancy Chin-Jung Wang, M.D.,a Fengpo Tsai, M.D.,b Chaowen Chen, M.D.,b and Angel Chao, M.D., Ph.D.a a

Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou and Chang Gung University College of Medicine, Kwei-Shan, Tao-Yuan; and b Poyuan Women Clinic, Changhua, Taiwan

Objective: To report a cesarean scar pregnancy (CSP) with a coexistent viable intrauterine pregnancy. Design: Case report. Setting: Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou, Taiwan. Patient(s): A 31-year-old woman, with previous cesarean delivery, presented with vaginal bleeding, was transferred to our hospital at 7 weeks’ gestation for heterotopic pregnancy after an IVF–embryo transfer. A diagnosis of intrauterine pregnancy combined with CSP was made by ultrasonography. Intervention(s): Hysteroscopic-directed evacuation of CSP. Main Outcome Measure(s): Good hemostasis at cesarean site and ongoing intrauterine pregnancy. Result(s): A healthy baby was delivered by cesarean delivery at term. Conclusion(s): With the increasing number of IVF–embryo transfers, the amount of heterotopic pregnancies is also increasing. Hysteroscopic management of CSP is a minimally invasive procedure that leads to successful obstetric outcomes in the corresponding intrauterine pregnancies. (Fertil Steril 2010;94:1529.e15–e18. 2010 by American Society for Reproductive Medicine.) Key Words: Cesarean scar, heterotopic, hysteroscopy, pregnancy, assisted reproductive technology

With the increasing use of assisted reproductive technologies (ART), more cases of heterotopic pregnancies were being diagnosed. The incidence of heterotopic pregnancies in ART is 0.2%– 1% (1). Management of cesarean scar pregnancy (CSP) itself carries risk of uncontrollable bleeding, not to mention the salvage of the concomitant intrauterine pregnancy. In our previous report, successful removal of CSP using operative hysteroscopy was demonstrated (2–4). We report here a case of heterotopic pregnancy of intrauterine and cesarean after IVF–embryo transfer and close monitoring of early pregnancy and prompt removing of the CSP to preserve the intrauterine pregnancy.

CASE REPORT A 31-year-old Vietnamese woman, gravida 2, para 1, had a history of bilateral tubal occlusion and one lower segment transverse cesarean delivery. She underwent IVF–embryo transfer at a local IVF clinic. Four embryos were transferred into the uterus under transabdominal ultrasound guidance. A positive pregnancy test was noted 14 days after embryo transfer. Three weeks later, she presented with vaginal spotting and transabdominal ultrasound revealed two intrauterine gestational sacs, one in the middle of the uterine cavity and the Received November 2, 2009; revised February 13, 2010; accepted February 17, 2010; published online March 29, 2010. C.-J.W. has nothing to disclose. F.T. has nothing to disclose. C.C. has nothing to disclose. A.C. has nothing to disclose. Supported by Chang Gung Memorial Hospital, Linkou, Taiwan, Grant CMRPG340753. Reprint requests: Angel Chao, M.D., Ph.D., Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou, Taiwan (FAX: 886-3-3288252; E-mail: [email protected]).

0015-0282/$36.00 doi:10.1016/j.fertnstert.2010.02.039

second within the isthmic area of the lower anterior wall of the uterus. A diagnosis of heterotopic CSP was highly suspected. Because of the patient’s strong desire to preserve her normal pregnancy, she was transferred to our endoscopic division. Transvaginal ultrasonography (TVUS) confirmed the diagnosis (Fig. 1). After counseling, the couple opted for conservative treatment with operative hysteroscopy, which is approved by the Institutional Review Board (IRB no.98-3535B) of the Chang Gung Memorial Hospital. The operative hysteroscopy was performed at 7 weeks’ gestation. Under spinal anesthesia, the patient was placed in the dorsolithotomy position. After a speculum was placed inside the vagina, a tenaculum was applied to the cervix and gentle traction was exerted to align the uterus. The cervix was carefully dilated by Hegar dilators to 11 mm, not beyond the endocervical canal, and a continuous flow 26F hysteroscopic resectoscope (Karl Stortz, Tuttlingen, Germany) with a 900 wire loop electrode was introduced under ultrasound control. Uterine distension was achieved using distilled water propelled by simple gravity. The height of the intravenous bag was positioned approximately 100 cm above the patient’s uterus (5, 6). An Aspen Excalibur (Aspen Labs, Englewood, CO) electrosurgical generator was used on a setting of 80 W of cutting waveform current and 100 W of coagulation current. The intervention began by identifying the implantation of the ectopic sac. The electric loop of the resectoscope was used to push the gestational sac, exposing the vessel bed of the implantation site. A coagulation current of 100 W was used for hemostasis. A placenta forceps was then used to pull out the gestational sac under sonographic guidance, and suction curettage was used to clear the residual gestational tissue. Finally, a hysteroscopic rolling ball was used to stop the bleeding point. The operating time was 15 minutes.

Fertility and Sterility Vol. 94, No. 4, September 2010 Copyright ª2010 American Society for Reproductive Medicine, Published by Elsevier Inc.

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FIGURE 1 Transvaginal ultrasonography shows the intrauterine pregnancy and the cesarean scar pregnancy at 7 weeks’ gestation. GS ¼ gestational sac; CX ¼ cervix; CSP ¼ cesarean scar pregnancy; CDS ¼ cul-de-sac.

Vaginal bleeding was minimal at the end of the procedure. Postoperative ultrasound showed a normal intrauterine pregnancy with complete disappearance of the ectopic gestational sac (Fig. 2). The patient had an unremarkable postoperative course and was discharged the next day. She recovered with no vaginal spotting and the remaining intrauterine pregnancy proceeded until 39th week of gestation. A healthy male baby, weighed 3,250 g, was delivered by cesarean section.

DISCUSSION

Wang. Heterotopic cesarean scar pregnancy. Fertil Steril 2010.

Hysteroscopic removal of the CSP provides an alternative treatment to preserve the viable intrauterine gestation. To our knowledge, this is the first case report of a successful conservative treatment of heterotopic CSP after IVF–embryo transfer using hysteroscopy. Suspicion on heterotopic pregnancies should be particularly considered in women with abdominal pain or vaginal bleeding after IVF–embryo transfer. Transvaginal ultrasonographic examination is a tool that makes early diagnosis possible, even in asymptomatic cases. Different types of heterotopic pregnancies were noted that include cervical, tubal, and cornual, where removal of the ectopic gestation usually allow the intrauterine pregnancy to proceed to term (7, 8). At present there is no standard treatment protocol for heterotopic pregnancy involving CSP. Conservative management that has been reported sporadically included laparoscopic excision, fetal

FIGURE 2 Hysteroscopic management of heterotopic cesarean scar pregnancy under ultrasound guidance. (A) Before hysteroscopic treatment, cesarean scar pregnancy with fetal heart beat (FHB) is seen. Arrow shows cesarean scar site. (B) Gestational villi are seen through the distension medium. (C) After hysteroscopic treatment, only one viable fetus is left. (D) Clear cervical area after the evacuation.

Wang. Heterotopic cesarean scar pregnancy. Fertil Steril 2010.

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Heterotopic cesarean scar pregnancy

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TABLE 1 Review of English literature describing successful management of heterotopic cesarean scar pregnancy.

Reference

Pre-op Case Age Gravity Previous Mode of Gestation Fetal b-hCG level no. (y) and parity LSCS (n) conception (wk) heart beat (mIU/mL)

Management

Operating Post-op time (min) stay (d)

Salomon et al. (9)

1

36

G4, P1

1

IVF

8

þ

N/K

TVS guidance, potassium chloride-directed injection

N/K

N/K

Hsieh et al. (10)

2

38

G4, P2

2

IVF

6

þ

N/K

N/K

1

Yazicioglu et al. (11)

3

23

G2, P1

1

Spontaneous

6

þ

N/K

N/K

N/K

Wang et al. (12)

4

38

G4, P3

3

IVF

10

þ

N/K

TVS guidance, embryo aspiration, preserving twin pregnancy TVS guidance, potassium chloride-directed injection TVS guidance, potassium chloride-directed injection

N/K

N/K

Demirel et al. (13)

5

34

G2, P1

1

Spontaneous

6

þ

N/K

45

N/K

Present case

6

31

G3, P1

1

IVF

7

þ

99,544

Laparoscopic removal of cesarean scar pregnancy Hysteroscopic-directed evacuation þ D&C

15

1

Outcome CS at 36 wk due to premature rupture of the membranes Live female 2,800 g CS at 32 wk due to preterm labor Live twin CS at 30 wk due to abruptio placenta Live male 1,530 g CS delivery at 35 wk due to preterm labor Live male 1,820 g Immediate PPH managed by hypogastric artery ligation Unremarkable pregnancy CS at 38 wk Live singleton Unremarkable pregnancy CS at 39 wk Live male 3,250 g

Note: Pre-op ¼ preoperative; Post-op ¼ postoperative; N/K ¼ not known; TVS ¼ transvaginal sonography; PPH ¼ postpartum hemorrhage; D&C ¼ dilatation and curettage; CS ¼ cesarean section. Wang. Heterotopic cesarean scar pregnancy. Fertil Steril 2010.

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reduction by potassium chloride, or embryo aspiration (9–13). Successful management of heterotopic CSP relied on either fetal reduction with gestational tissue in situ or removal of ectopic gestational tissue (Table 1). For fetal reduction with gestational tissue in situ, although four patients with heterotopic CSP received this procedure under TVUS guidance ended with live births by cesarean deliveries, these pregnant courses were not uneventful (Table 1). All babies were born prematurely due to various causes (premature rupture of the membranes, abruptio placentae, or preterm labor). Immediate postpartum hemorrhage was even encountered in one pregnancy and bilateral hypogastric arteries ligation was necessary to stop the bleeding and preserve the uterus (Table 1). Furthermore, the retained gestational tissues might affect the muscle strength of the uterine lower segment in the ongoing pregnancy. Consequently, the residual placenta and deciduous tissues in a weak myometrium might predispose to early uterine premature contraction. Instead, surgical removal of heterotopic CSP carries uneventful prenatal courses and the pregnant women delivered live births at term. Laparoscopic removal of ectopic mass in heterotopic CSP with good pregnancy outcome has been reported (13). However, to follow the principles of laparotomy (12), proper anatomic dissection, trim of unhealthy tissues, and repair of the uterine defect should be performed at the same time by laparoscopy. Massive operative blood loss is another main concern. Thus, laparoscopic management is not suitable for an inexperienced laparoscopic surgeon.

We previously reported the management of the first trimester CSP using hysteroscopy (4). Accumulating experiences from this approach we chose this minimally invasive method to remove the heterotopic CSP. Although the uterine pregnancy was uneventful, the possibility of complications involving uterine defects after removal of gestational tissue by hysteroscopic surgery should be considered. Complications with advanced pregnancy of the intrauterine embryo, such as increased the possibility of intermittent antepartum spotting or uterine rupture before cesarean section, should be watched for. Different from the management of single CSP, hysteroscopic manipulation should be reduced to as much a minimum as possible because the intrauterine embryo might be disturbed by the upward force of the distended medium during hysteroscopy. After identifying the location of the CSP and coagulating the vessel beds of the implantation site, the hysteroscope should be withdrawn immediately and replaced by placenta forceps and suction curettage to remove the gestational tissues under sonographic guidance. Caution with avoidance of damaging the embryo should be strictly followed. Based on the reported cases and our own experience we suggest surgical removal of the ectopic mass for the management of first trimester heterotopic CSP. For experienced hysteroscopists, this case report offers an important alternative treatment for heterotopic pregnancy involving CSP with a short operative time and minimum blood loss. Early diagnosis is warranted to preserve the viability of the intrauterine fetus and avoid maternal morbidity.

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10. 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. 11. Yazicioglu HF, Turgut S, Madazli R, Aygun M, Cebi Z, Sonmez S. An unusual case of heterotopic twin pregnancy managed successfully with selective feticide. Ultrasound Obstet Gynecol 2004;23:626–7. 12. Wang CN, Chen CK, Wang HS, Chiueh HY, Soong YK. Successful management of heterotopic cesarean scar pregnancy combined with intrauterine pregnancy after in vitro fertilization–embryo transfer. Fertil Steril 2007;88:706:e13–6. 13. Demirel LC, Bodur H, Selam B, Lembet A, Ergin T. Laparoscopic management of heterotopic cesarean scar pregnancy with preservation of intrauterine gestation and delivery at term: case report. Fertil Steril 2009;91:1293:e5–7.

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