Case Report: Cervical pregnancy — a conservative stepwise approach

M. Yitzhak1, R. Orvieto1, S. Nitke1, M. Neuman-Levin2, Z. Ben-Rafael1 and A. Schoenfeld1,3

1 Departments of Obstetrics and Gynecology and 2 Radiology, Rabin Medical Center, Beilinson Campus, Petah Tiqva and Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel


    Abstract
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
A case of cervical pregnancy resistant to intramuscular methotrexate therapy is presented, which was successfully treated by intra-arterial methotrexate followed by selective prophylactic hypogastric artery embolization to avoid aggravating the vaginal bleeding. It is suggested that, in cervical pregnancies in which fertility preservation is desired, a stepwise conservative approach should be applied before resorting to surgical intervention.

Key words: : cervical pregnancy/conservative treatment/intra-arterial embolization/MTX


    Introduction
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
Cervical pregnancy is a rare form of ectopic pregnancy which is often associated with significant morbidity and devastating effects on future fertility. It accounts for <1% of all ectopic gestations. Its incidence varies between 1 in 1000 to 16 000 pregnancies, with the highest figures reported from Japan, which also has a high incidence of antecedent curettage (Rock and Thompson, 1997Go). The diagnosis of cervical pregnancy is commonly delayed and is often made intraoperatively in the presence of massive blood loss, necessitating an emergency hysterectomy in ~50% of cases. Early diagnosis has been improved by ultrasonography, with a consequent decrease in morbidity and mortality. During the last decade, in an attempt to avoid hysterectomy and preserve fertility, a more conservative therapeutic approach was developed, including chemotherapy, cerclage, hypogastric iliac artery ligation, and arterial embolization under angiographic control as reviewed by Ushakov et al. (Ushakov et al., 1996Go). In this report, we present a stepwise conservative approach, using invasive angiographic techniques, for cervical pregnancy resistant to treatment with methotrexate (MTX).


    Case report
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
A 31-year-old woman, gravida 2, para 1, was admitted to our department at 5 weeks gestation with painless vaginal bleeding of 2 days duration. Her medical history was unremarkable, with no previous intrauterine procedures, pelvic inflammatory disease, or intrauterine devices. Vital signs were stable, and the abdomen was soft and not tender. Pelvic examination revealed a barrel-shape uterine cervix with minimal bright bleeding protruding through a closed external os. The uterus was slightly enlarged and had no adnexal masses. Transabdominal and transvaginal ultrasound examinations (Aloka SSD 650, Tokyo, Japan, 5 MHz) confirmed the presence of a cervical pregnancy with fetal pole and fetal cardiac activity (Figure 1Go). Quantitative beta-human chorionic gonadotrophin (HCG) concentration (Immulite HCG kit, Euro/Dpc Ltd, UK) was 8620 mIU/ml on admission, and almost twice that (15 800 mIU/ml) the next day. ßHCG was measured at frequent intervals thereafter, but only the values relevant to this report are given.



View larger version (81K):
[in this window]
[in a new window]
 
Figure 1. Cervical pregnancy (Cx), as seen by transvaginal ultrasonography.

 
In an attempt to preserve fertility, we offered the patient conservative management with i.m. MTX. The potential risks and alternative methods of treatment were explained to her, and written informed consent was obtained. The most commonly used treatment regimen in our department was applied. This consisted of i.m. MTX 1 mg/kg and folinic acid 0.1 mg/kg given alternately every other day for 4 days. After completion of treatment, however, a rise in ßHCG level (19 690 mIU/ml) was observed (Figure 2Go), and the ultrasound examination revealed an unchanged gestational sac size with persistent fetal cardiac activity. On the basis of these findings and the patient's haemodynamically stable state, we decided to follow our previously successful experience with direct intra-arterial MTX administration (Peleg et al., 1994Go). The following day, the patient underwent percutaneous catheterization of the femoral artery with a flexible angiographic catheter (4.1 C1 Cobra catheter; Cordis, Boston, MA, USA), which was advanced under fluoroscopy to the left hypogastric artery. MTX 50 mg was administered over 20 min and the catheter was removed.



View larger version (14K):
[in this window]
[in a new window]
 
Figure 2. Beta-human chorionic gonadotrophin (BHCG) concentration during the course of disease. MTX = methotrexate.

 
During the next few days the ßHCG level decreased slightly (Figure 2Go), measuring 10 230 mIU/ml at 1 week, with the concomitant disappearance of fetal cardiac activity. HCG continued to decrease for the next 2 weeks and then stabilized around 2700 mIU/ml (Figure 2Go). Though serial ultrasound examinations showed no change in the intracervical sac dimensions, bright red vaginal bleeding requiring the use of two perineal pads during one night reappeared. As a result, and in an attempt to avoid subsequent life-threatening cervical bleeding, the following day we decided to perform prophylactic selective hypogastric artery embolization using the aforementioned technique. An effort was made to advance the catheter tip so that the anterior branches of both hypogastric arteries were embolized at their distal ends. Gelfoam pledgets, 1x1 mm, were injected until the uterine arterial flow ceased (Figure 3Go). Two days later vaginal bleeding ceased and the patient was discharged. The ßHCG concentration continued to drop on out-patient follow-up, as shown in Figure 2Go. One week after discharge, a collapsed gestational sac was demonstrated; coincidental with the commencement of menstruation, the gestational sac disappeared.




View larger version (338K):
[in this window]
[in a new window]
 
Figure 3. Angiographic presentation of the pelvic vessels: (A) before, and (B) after hypogastric artery embolization (emb.) and gelfoam pledget injection.

 

    Discussion
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
There are two main treatment options for cervical pregnancy when fertility is desired: surgical and pharmacological. The different methods described (Ushakov et al., 1996Go) include cervical cerclage, intracervical balloon tamponade of the cervix, vaginal packing, local haemostatic sutures, curettage followed by local prostaglandin instillation (Spitzer et al., 1997Go), ligation of the descending branches of the uterine arteries, and bilateral hypogastric artery ligation. Since the early 1980s there have been many reports of the successful and unsuccessful use of chemotherapy; MTX has been variously administered by the i.m., i.v., intracervical and intra-amniotic routes (Mantalenakis et al., 1995Go; Nomiyama et al., 1997Go). The presence of fetal cardiac activity or advanced gestational age was associated with higher rates of treatment failure (Barham and Paine, 1989Go).

Peleg et al. (1994) described the successful use of intra-arterial MTX for combined intrauterine and cervical pregnancy, which allowed for rapid local accumulation of the drug without increasing the severity of toxicity. With today's more widespread application of arterial angiography in gynaecology and obstetrics, selective arterial embolization has become accepted as a highly effective technique for controlling acute and chronic genital bleeding (Chin et al., 1989Go; Vedantham et al., 1997Go). Selective arterial embolization is optimally employed prior to hypogastric artery ligation or hysterectomy and has various therapeutic advantages, such as avoidance of surgical risk and preservation of fertility. The procedure was recently used by Cosin et al. (1997) to avoid surgery in a patient with a cervical pregnancy.

Recently, Nomiyama et al. (1997) reported on a conservative treatment using preventive selective uterine artery embolization, followed by intra-amniotic MTX instillation. Although selective hypogastric artery embolization is considered a safe procedure, short-term complications and serious tissue ischaemia have been documented (Stancato-Pasik et al., 1997Go). That is the reason we performed this procedure as a last resort and before proceeding to surgical intervention.

In our case, in an attempt to preserve fertility, we chose a stepwise conservative approach (Figure 4Go). We suggest that MTX, which seems by far the best choice for treatment of cervical pregnancies, should be offered first by the i.m. route, by the routine protocol most commonly used by the department, which is considered simple and safe. If on follow-up evaluation, ßHCG concentrations do not decrease (>15% from baseline) or persistent fetal cardiac activity is observed, direct intra-arterial MTX should be instituted (Peleg et al., 1994Go). We prefer this approach to proceeding to direct puncture and feticide because of the possibility of starting an incomplete abortion with consequent life-threatening haemorrhage. During MTX administration, an increase in bleeding pattern or the reappearance of vaginal bleeding may require further intervention with intra-arterial embolization. Any profuse bleeding during these therapeutic measures, with consequent haemodynamic compromise of the patient, may necessitate surgical intervention (Ushakov et al., 1996Go), such as curettage with Foley catheter tamponade, Shirodkar-type cervical cerclage, cervical hysterectomy, bilateral uterine or iliac artery ligation and hysterectomy.



View larger version (19K):
[in this window]
[in a new window]
 
Figure 4. The suggested stepwise conservative approach to cervical pregnancy. Decreased or increased beta-human chorionic gonadotrophin (ßHCG) concentrations — change of < or >15% from baseline, respectively. Cx = cervical; MTX = methotrexate.

 


    Acknowledgments
 
We wish to thank Prof. I.Meizner and Dr. R.Mashiach for their invaluable assistance.


    Notes
 
3 To whom correspondence should be addressed at: Department of Obstetrics and Gynecology, Rabin Medical Center, Beilinson Campus, Petah Tiqva 49 100, Israel Back


    References
 Top
 Abstract
 Introduction
 Case report
 Discussion
 References
 
Barham, J.M. and Paine, M. (1989) Reproductive performance after cervical pregnancy: a review. Obstet. Gynecol. Surv., 44, 650–655.[Medline]

Chin, H.G., Scott, D.R., Resnik, R. et al. (1989) Angiographic embolization of intractable puerperal hematomas. Am. J. Obstet. Gynecol., 160, 434–438.[ISI][Medline]

Cosin, J.A., Bean, M., Grow, D. et al. (1997) The use of methotrexate and arterial embolization to avoid surgery in a case of cervical pregnancy. Fertil. Steril., 67, 1169–1171.[ISI][Medline]

Mantalenakis, S., Tsalikis, T., Grimbizis, G. et al. (1995) Successful pregnancy after treatment of cervical pregnancy with methotrexate and curettage. J. Reprod. Med., 40, 409–414.[ISI][Medline]

Nomiyama, M., Arima, K., Iwasaka, T. et al. (1997) Conservative treatment using a methotrexate-lipidol emulsion containing non-ionic contrast medium for a cervical ectopic pregnancy. Hum. Reprod., 12, 2826–2829.[Abstract]

Peleg, D., Bar Hava, I., Neuman-Levin, M. et al. (1994) Early diagnosis and successful nonsurgical treatment of viable combined intrauterine and cervical pregnancy. Fertil. Steril., 62, 405–408.[ISI][Medline]

Rock, J.A. and Thompson, J.D. (eds) (1997) Te Linde's Operative Gynecology. 18th edition. Lippincott-Raven Publishers, Philadelphia, PA, pp. 523–524.

Spitzer, D., Steiner, H., Graf, A. et al. (1997) Conservative treatment of cervical pregnancy by curettage and local prostaglandin injection. Hum. Reprod., 12, 860–866.[Abstract]

Stancato-Pasik, A., Mitty, H.A., Richard, H.M. and Eshkar, N. (1997) Obstetric embolotherapy: effect on menses and pregnancy. Radiology, 204, 791–793.[Abstract]

Ushakov, F.B., Elchalal, U, Aceman, P.J. et al. (1996). Cervical pregnancy: past and future. Obstet. Gynecol. Surv., 52, 45–57.

Vedantham, S., Goodwin, C.S., McLucas, B. et al. (1997) Uterine artery embolization: an underused method of controlling pelvic hemorrhage. Am. J. Obstet. Gynecol., 176, 938–948.[ISI][Medline]

Submitted on June 8, 1998; accepted on December 9, 1998.