Human Reproduction, Vol. 16, No. 3, 584-586,
March 2001
© 2001 European Society of Human Reproduction and Embryology
Cervical ectopic twin pregnancy: diagnosis and conservative treatment: Case report
1 Department of Obstetrics and Gynaecology and 2 Department of Pathology, Institut Universitari Dexeus, Paseo de la Bonanova 89–91, 08017 Barcelona, Spain
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Abstract |
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A case of cervical ectopic twin pregnancy with cardiac activity in both embryos is presented. It was diagnosed in the eighth week of gestation by ultrasonography, and treated conservatively with intra-amniotic administration of methotrexate under ultrasonographic guidance followed by curettage. This procedure allows subsequent gestations.
Key words: cervical twin pregnancy/methotrexate/reproduction/ultrasound
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Introduction |
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Cervical pregnancy represents a rare and dangerous type of ectopic pregnancy, ranging in incidence between 1:1000 and 1:18 000 pregnancies (Yankowitz et al., 1990
). Ectopic twin pregnancy is quite uncommon in the cervix; only one case of twin pregnancy has been reported in the cervico-isthmic region (Iloabachie et al., 1993). The diagnosis is usually done at the end of the first trimester of gestation, and in the past, treatment consisted of hysterectomy, allowing no possibility of future gestations. Nowadays, ultrasonographic diagnosis of ectopic pregnancy allows conservative treatment such as curettage followed by cervical tamponade, cervical cerclage after curettage or cervical amputation with hipogastric ligation (Parente et al., 1983). Local or systemic treatment with methotrexate (Farabow et al, 1983) seems to be an appropriate choice to preserve fertility (Oyer et al., 1988; Stovall et al., 1988; Kaplan et al., 1989; Kung and Chang, 1999); in some cases additional procedures are needed such as curettage (Timor-Tritsch et al., 1994), hipogastric embolization (Frates et al., 1994; Cosin et al., 1997) or bilateral uterine artery ligation. This paper describes a case of cervical ectopic twin pregnancy diagnosed ultrasonograpically at 8 weeks gestation, treated conservatively with local administration of methotrexate under ultrasonographic guidance, followed by curettage.
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Case report |
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A 39 year old female who had had two abortions became pregnant after one cycle of IVF. The patient weighed 53 kg. Previously she had been unsuccessfully submitted to six cycles of artificial conjugal insemination. The hysteroscopy performed for infertility studies revealed a hypotrophy of the external canal. According to our institution's protocol, the IVF cycle was performed following a long protocol, retrieving 12 oocytes. Four embryos were obtained by intracytoplasmatic sperm injection (ICSI). Three embryos were replaced without ultrasonographic guidance with a Wazel catheter. The remaining embryo was cryopreserved. Four weeks later, the patient was seen with vaginal bleeding. Ultrasonography showed an empty uterine cavity and a bizygotic twin gestation in the cervix below the concentration of the internal os (Figure 1
) and with cardiac activity in both embryos (Figure 2). Colour Doppler study showed peritrophoblastic flow. The ß-human chorionic gonadotrophin (ßHCG) concentration was 83.465 IU/l.
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Both gestational sacs were punctured under ultrasonographic control and their contents aspirated for cytological study. In each gestational sac 50 mg of methotrexate was introduced. The study revealed the presence of cytotrophoblast in both gestational sacs. No haemorrhage, complications or other adverse effects were seen after the procedure. The patient was discharged in good condition the following day and monitoring by daily transvaginal ultrasound and measurement of ßHCG concentrations every 3 days for 10 days was performed. The ßHCG concentrations showed a moderate increase (97.700 IU/l) in the second sample after treatment. The weekly monitoring performed afterwards showed a decrease in the ßHCG concentration (Figure 3
). Ultrasonographic follow-up only revealed heterogeneity of the sacs' contents and peripheral vascularization (Figure 4).
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At 36 days after a fibrinohaematic material with necrotic decidua was vaginally discharged. After 40 days, vaginal bleeding required cervical and endometrial curettage as well as blood transfusion. Histological studies showed deciduo-ovular rests. Hysteroscopy 8 months later displayed a right lateral side synechia.
The patient underwent a new IVF cycle with long protocol obtaining six oocytes, all of them fertilized by conventional IVF. Three embryos were transferred with ultrasound guidance according to conventional techniques (Coroleu et al., 2000) and the three remaining were cryopreserved. Five weeks later one pregnancy was confirmed by transvaginal ultrasound with an 11 mm gestational sac containing one embryo with cardiac activity and a normal cervix uterus. Amniocentesis was performed and showed a fetus with karyotype 46 XY. At 42 weeks a healthy live male was vaginally delivered.
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Discussion |
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Transvaginal ultrasound diagnosis of cervical ectopic pregnancy requires the presence of a gestational sac in a widened cervical canal and the exclusion of a gestational sac in the uterine cavity. It should not be confused with the product of conception in transit within the cervical canal. Hence the diagnosis of ectopic pregnancy should be accompanied by colour Doppler study to identify the peritrophoblastic flow (Jurkovic et al., 1996
). The case presented not only showed embryonic cardiac activity but also the characteristic peritrophoblastic flow with high peak systolic velocity and low impedance to flow. Ectopic twin pregnancies have rarely been reported in the ovary (Ohba et al., 1992), broad ligament (Deshpande et al., 1999), Fallopian tube (Lema, 1990; Ansari et al., 2000) and in the cervico–isthmic region (Iloabachie et al., 1993), most of them associated with endometriosis or following IVF and treated surgically.
The therapeutical approach can be radical or conservative. When radical, a hysterectomy is usually performed when haemorrhage occurs as an emergency situation. The conservative treatment can be surgical or medical. Surgical procedures imply hypogastric or uterine artery ligation or embolization, a curettage with or without cerclage, or the insertion of a Foley probe in the cervical canal. According to the reported cases, curettage seems to increase the haemorragic risk requiring hysterectomy in many occasions. Another therapeutical choice is local administration of methotrexate or potassium chloride (Jurkovic et al., 1996; Carreno et al., 2000). Methotrexate is an agonist of folic acid that participates in DNA synthesis. It has been used for treatment of choriocarcinoma and ectopic pregnancy due to its capacity to stop proliferative cell activity (Fernandez et al., 1998). Its first application in the treatment of cervical pregnancies was described by Farabow et al. (Farabow et al., 1983). Adverse effects of its administration include nausea (50%), vomiting (17%), diarrhoea (14%) and less frequently oral ulcers and stomatitis (5%) (Wiebe, 1999).
The intra-cervical or intra-amniotic dose of methotrexate is usually 12.5 to 50.5 mg, varying according to different authors (Timor-Tritsch et al., 1994; Mantalenakis et al., 1995). In the present case methotrexate was administrated into each gestational sac with transvaginal ultrasonographic guidance in a unique dose of 50 mg for each. The local treatment was chosen to avoid the adverse effects of systemic methotrexate administration. Methotrexate alone or combined with procedures such as curettage or cervical tamponade is effective in those ectopic pregnancies of 12 weeks of gestation or less and does not produce adverse effects (Kung et al., 1997). According to this review 66% (12/18) of patients with complete remission of a cervical ectopic pregnancy treated with methotrexate required additional approaches, cervical curettage being the therapy most commonly used. As in other reports (Balasch et al., 1994), hysteroscopic follow-up revealed no anatomical or functional changes in the cervix in the case reported here.
As proven in the present case, conservative treatment of cervical ectopic pregnancy is a therapeutic choice, even in the case of a twin pregnancy.
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Acknowledgments |
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The authors are grateful to the Càtedra d'Investigació en Obstetrícia i Ginecologia (Universitat Autónoma de Barcelona) for its support.
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3 To whom correspondence should be addressed. E-mail: marpas{at}iudexeus.uab.es
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References |
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Ansari, S.M., Nessa, L. and Saha, S.K. (2000) Twin ectopic pregnancy 10 years after permanent sterilization. Australas Radiol., 44, 107–108.[Medline]
Balasch, J., Peñarrubia, J., Ballesca, J.L. et al. (1994) Intrauterine insemination, cervical pregnancy and successful treatment with methotrexate. Hum. Reprod., 9, 1580–1583.
Carreno, C.A., King, M., Johnson, M.P. et al. (2000) Treatment of heterotopic cervical and intrauterine pregnancy. Fetal. Diagn. Ther., 15, 1–3.[ISI][Medline]
Coroleu, B., Carreras, O., Veiga, A. et al. (2000) Embryo transfer under ultrasound guidance improves pregnancy rates after in-vitro fertilization. Hum. Reprod., 15, 616–620.
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]
Deshpande, N., Mathers, A. and Achrya, U. (1999) Broad ligament twin pregnancy following in-vitro fertilization. Hum. Reprod., 14, 852–854.
Farabow, W.S., Fulton, J.W., Flecther, V. et al. (1983) Cervical pregnancy treated with methotrexate. N. C. Med. J., 44, 91–93.[ISI][Medline]
Fernandez, H., Yves Vincent, S.C., Panthier, S. et al. (1998) Randomized trial of conservative laparoscopic treatment and methotrexate administration in ectopic pregnancy and subsequent fertility. Hum. Reprod., 13, 3239–3243.
Frates, M.C., Berenson, C.B., Doubilet, P.M. et al. (1994) Cervical ectopic pregnancy: results of conservative treatment. Radiology, 191, 773–775.
Iloabachie, G.C., Igwebe, A.O., and Izuroa, K.L. (1993) Cervico-isthmic twin pregnancy carried to 37 weeks. Int. J. Gynaecol. Obstet., 40, 59–61.[Medline]
Jurkovic, D., Hacket, E. and Campbell, S. (1996) Diagnosis and treatment of early cervical pregnancy: a review and report of two cases treated conservatively. Ultrasound Obstet. Gynecol., 8, 373–380.[ISI][Medline]
Kaplan, B.R., Brandt, T., Javaheri, G. et al. (1989) Successful treatment of a live cervical pregnancy with methotrexate and folinic acid. A case report. J. Reprod. Med., 34, 853–856.[ISI][Medline]
Kung, F.T. and Chang, S.Y. (1999) Efficacy of methotrexate treatment in viable and nonviable cervical pregnancies. Am. J. Obstet. Gynecol., 181, 1438–1444.[ISI][Medline]
Kung, F.T., Chang, S.Y., Tsai, Y.C. et al. (1997) Subsequent reproduction and obstetric outcome after methotrexate treatment of cervical pregnancy: a review of original literature and international collaborative follow-up. Hum. Reprod., 12, 591–595.
Lema, V.M. (1990) Unilateral tubal twin pregnancy. East Afr. Med. J., 67, 918–921.[ISI][Medline]
Mantalenakis, S., Tsalikis, T., Grimbizis, G. et al. (1995) Successful pregnancy after treatment of cervical pregnancy with MTX and curettage, a case report. J. Reprod. Med., 40, 409–414.[ISI][Medline]
Ohba, T., Miyazaki, K., Kouno, T. et al. (1992) Ovarian twin pregnancy. Acta Obstet. Gynecol. Scand., 71, 305–307.[ISI][Medline]
Oyer, R., Tarakjian, D., Lev-Toaff, A. et al. (1988) Treatment of cervical pregnancy with methotrexate. Obstet. Gynecol., 71, 469–471.[ISI][Medline]
Parente, J.T., Ou, C.S., Levy, J. et al. (1983) Cervical pregnancy analysis: a review and report of five cases. Obstet. Gynecol., 62, 79–82.
Stovall, T.G., Ling, F.W., Smith, W. et al. (1988) Successful nonsurgigal treatment of cervical pregnancy with methotrexate. Fertil. Steril., 50, 672–674.[ISI][Medline]
Timor-Tritsch, I., Monteagudo, A., Mandeville, E.O. et al. (1994) Successful management of viable pregnancy by local injection of methotrexate guided by transvaginal ultrasonography. Am. J. Obstet. Gynecol., 170, 737–739.[ISI][Medline]
Wiebe, ER. (1999) Comparing abortion induced with methotrexate and misoprostol to methotrexate alone. Contraception, 59, 7–10.[ISI][Medline]
Yankowitz, J., Leake, J., Huggins, G. et al. (1990) Cervical ectopic pregnancy. Review of the literature and report of a case treated by single-dose methotrexate therapy. Obstet. Gynecol. Survey, 45, 405–414.[Medline]
Submitted on July 27, 2000; accepted on November 24, 2000.
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