CICINELLI, Ettore

COLAFIGLIO G

LANZILLOTTI G

SALIANI N

TROJANO G

English

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CASE REPORTSuccessful management of a cervical twin pregnancy:neoadjuvant systemic methotrexate and prophylactichigh cervical cerclage before curettageGiuseppe Trojano, M.D.,a Giuseppe Colafiglio, M.D.,a Nicola Saliani, M.D.,aGiuseppe Lanzillotti, M.D.,b and Ettore Cicinelli, M.D.aa Department of Gynaecology, Obstetrics and Neonatology, University Hospital, Bari; and b Division of Gynaecology andObstetrics, ‘‘Perrino’’ Hospital, Brindisi, ItalyObjective: To report our experience in managing a case of viable cervical twin pregnancy with systemic metho-trexate and 2 weeks later with endocervical curettage after placing prophylactic high cervical cerclage.Design: Case report.Setting: University medical center.Patient(s): A 36-year-old woman, gravida 2, para 0, complaining of vaginal bleeding and pelvic pain, in whomcervical twin pregnancy was diagnosed at the sixth week of gestation.Intervention(s): Systemic methotrexate was given as first-line treatment. Two weeks later because of persistingbleeding, endocervical curettage was performed after closing cervical arteries and placing but not tighteninghigh cervical cerclage.Main Outcome Measure(s): Pregnancy termination, bleeding control, and preservation of fertility.Result(s): Notwithstanding the ligature of cervical arteries at curettage, heavy bleeding occurred, which was con-trolled rapidly by tightening the cerclage. Curettage then was completed successfully. Postoperative period wasuneventful. Two years later the woman delivered vaginally.Conclusion(s): In case of cervical twin pregnancy methotrexate pretreatment and prophylactic placement of highcervical cerclage, but not ligature of cervical arteries, before curettage showed to be effective in terminating preg-nancy ensuring effective bleeding control and preservation of fertility. (Fertil Steril 2009;91:935.e17–e19. 2009by American Society for Reproductive Medicine.)Key Words: Cervical pregnancy, cervical twin pregnancy, cervical cerclage, cervical arteries ligature, methotrex-ate, conservative treatment0015-0282/09/$36.00doi:10.1016/j.fertnstert.2008.11.009 Copyright ª2009 American Socgation of hypogastric and uterine arteries (10, 11). Medicalmanagement is based on the administration of either systemic(1, 11–17) or intra-amniotic methotrexate (18), or instillationof hypertonic solutions (19, 20).Early diagnosis of cervical implantation is crucial for min-imizing risks. Nowadays, transvaginal echography allows thediagnosis of cervical implantation to be performed at veryearly stages of pregnancy, thus increasing chances of successwith medical treatment. However, the number of differenttreatments proposed suggests that none of them is alwayseffective, and endocervical pregnancy therefore remainsone of the most threatening obstetric pathologic conditions.Cervical twin pregnancy is an extremely rare conditionthat, because of the presence of two sacs and therefore ofa wide intracervical implantation area, even at a very earlystage of pregnancy is at high risk of hemorrhagic complicationand demolitive surgery. To the best of our knowledge onlya few cases have been reported previously in the literature(6, 21). In this article we describe the successful managementin terms of bleeding control and fertility preservation ofBecause of the risk of heavy bleeding, cervical pregnancy isa dangerous and life-threatening pathologic condition, whoseincidence is reported to vary between 1:18,000 to 1:1,000 ofall pregnancies (1). In many cases the only possibility tocontrol bleeding, which may occur spontaneously as wellas during the curettage, is hysterectomy.Up to now different therapeutic approaches have been pro-posed to avoid hysterectomy and preserve fertility. Surgicaloptions include intracervical balloon tamponade after cervi-cal curettage (2), cervical cerclage (3, 4), angioembolizationof the uterine arteries (5, 6), curettage and local prostaglandininjection (7), hysteroscopic resection (8, 9), and bilateral li-Received August 26, 2008; revised November 2, 2008; accepted Novem-ber 4, 2008.G.T. has nothing to disclose. G.C. has nothing to disclose. N.S. hasnothing to disclose. G.L. has nothing to disclose. E.C. has nothing todisclose.Reprint requests: Giuseppe Trojano, M.D., Department of Gynaecology,Obstetrics and Neonatology University, Hospital Bari, Policlinico ofBari, Piazza Giulio Cesare, 11, 70100 Bari, Italy (FAX: 39-080-5592230;E-mail: giutrojano@gmail.com).Fertility and Sterility Vol. 91, No. 3, March 2009 935.e17iety for Reproductive Medicine, Published by Elsevier Inc.a viable cervical twin gestation that infiltrated most of the cer-vix and that was based on a combination of medical and sur-gical treatment. The rationale was to interrupt the pregnancyto obtain a shrinkage of trophoblast and of vascular supplyby systemic methotrexate. Curettage was subsequently per-formed to reduce the patient’s bleeding after bilateral sutureof cervical arteries and placing a prophylactic high cervicalcerclage. Institutional Review Board approval was obtained.CASE REPORTA 36-year-old nulliparous woman referred to us complainingof heavy vaginal bleeding and pelvic pain. During clinical ex-amination a soft and bulky cervix was found. Surprisingly,a cervical ectopic twin pregnancy at the sixth week of gesta-tion was diagnosed by transvaginal ultrasound (Fig. 1). Car-diac activity in both embryos was present. Total diameter ofthe trophoblast was about 5 cm, and the muscular layer of thecervix was nearly completely infiltrated (Fig. 1); serumb-hCG levels were 52,000 mUI/mL.Because of the large size of the trophoblast and a highrisk of hemorrhagic complication, it was decided to avoidcurettage and to start with medical treatment. Methotrexate(50 mg/m2 of skin) 65 mg IM for 4 days every other dayalternated with folinic acid was given.After an expected rise in b-hCG serum levels up to 209,312mUI/mL, a progressive decrease was observed. However, thepatient continued to complain of bleeding of variable inten-sity. On day 22, although serum levels were decreased to16,242 mUI/mL, both cardiac activities had disappeared,and the total size of both gestational sacs was reduced to2.5 cm, significant vaginal bleeding was still present. There-fore, to stop bleeding, we decided to attempt a curettage ofthe cervical canal. Prophylactic bilateral ligature of the cervi-cal branches of the uterine artery was performed and a highcervical Shirodkar cerclage (Mersilene tape; Johnson andJohnson, New Brunswick, NJ) was placed but not tightenedto allow the curette to freely enter the entire uterine cavity.Notwithstanding methotrexate pretreatment, terminationof pregnancy, significant shrinkage of trophoblast, and theprophylactic suture of cervical branches of the uterine ar-teries, after the first passages of the curette heavy bleedingoccurred. Endocervical curettage was then completed, andcervical cerclage was tightened. The bleeding stopped imme-diately, and the patient was monitored strictly for the follow-ing hours.Seven days afterwards the cervical cerclage was removedand the patient discharged under good general conditions.b-Human chorionic gonadotropin titers were followed until7 days after the procedure when they became negative (<5mUI/mL).After 1 month the patient underwent ultrasound examina-tion that showed no abnormality; 3 months later hysteroscopyconfirmed a normal cervical and uterine cavity. Two yearslater the patient became pregnant again spontaneously. The935.e18 Trojano et al. Cervical twin pregnancypregnancy was uneventful, and she delivered a healthybaby vaginally at term.DISCUSSIONCervical pregnancy is a rare form of ectopic gestation thatrepresents a very engaging clinical situation. Even ifpromptly diagnosed and treated it often is associated with sig-nificant morbidity and deleterious effects for future fertility.At the moment no treatment is a priori effective, and the cor-rect management is still debated. In this article we describethe management of a very rare case of twin cervical preg-nancy at the sixth week of gestation.At the moment methotrexate treatment is used largely asfirst-line treatment worldwide. The success rate of metho-trexate treatment in cervical pregnancy has T:/PGN/DART-MOUTH/FNS/WEB/00024180/been reported to be as highas 81.3% (13). The combination of curettage after methotrex-ate administration increases the success rate to 94% even incase of viable cervical pregnancies (13). However, aftermethotrexate, as well as after combined surgical and neoad-juvant chemotherapy, the risk of severe bleeding is still high.The prophylactic cerclage may be an effective way to con-trol hemorrhagic complication in cervical pregnancy. In fact,in the present case, cervical cerclage showed to be the onlyeffective way to control bleeding. Our experience is in accor-dance with that of Mashiach et al. (4), who reported the effec-tiveness of cervical cerclage. We therefore decided to placebut not to tighten the cerclage to have the possibility ofFIGURE 1Transvaginal ultrasound scan at sixth week ofgestation showing the two gestational sacs at thecervical level. Total trophoblast diameter isapproximately 5 cm; trophoblast appears to infiltratemost of cervix so that the muscular layer in somepoints is totally infiltrated (white arrow).Trojano. Cervical twin pregnancy. Fertil Steril 2009.Vol. 91, No. 3, March 2009Fertility and Sterility9. Ash S, Farrell SA. Hysteroscopic resection of a cervical ectopic preg-nancy. Fertil Steril 1996;66:842–4.10. Nelson RM. Bilateral internal iliac artery ligation in cervical pregnancy:conservation of reproductive function. Am J Obstet Gynecol 1979;134:145–50.11. Lin H, Kung FT. Combination of laparoscopic bilateral uterine artery liga-tion and intraamniotic methotrexate injection for conservative manage-ment of cervical pregnancy. JAm Assoc Gynecol Laparosc 2003;10:215–8.12. Pascual MA, Ruiz J, Tresserra F, Sanuy C, Grases PJ, Tur R, et al.Cervical ectopic pregnancy: diagnosis and conservative treatment.Hum Reprod 2001;16:584–6.13. Kim TJ, Seong SJ, Lee KJ, Lee JH, Shin JS, Lim KT, et al. Clinical out-comes of patients treated for cervical pregnancy with or without metho-trexate. J Korean Med Sci 2004;19:848–52.14. Kirk E, Condous G, Haider Z, Syed A, Ojha K, Bourne T. The conserva-tive management of cervical ectopic pregnancies. Ultrasound ObstetGynecol 2006;27:430–7.15. Jurkovic D, Hacket E, Campbell S. Diagnosis and treatment of early cer-vical pregnancy: a review and report of two cases treated conservatively.Ultrasound Obstet Gynecol 1996;8:373–80.16. Kung FT, Chang SY. Efficacy of methotrexate treatment in viable andnonviable cervical pregnancies. Am J Obstet Gynecol 1999;181:1438–44.17. Balasch J, Pe~narrubia J, Ballesca JL, Creus M, Casamitjana R,Vanrell JA. Intra-uterine insemination, cervical pregnancy and success-ful treatment with methotrexate. Hum Reprod 1999;9:1580–3.18. Cerveira I, Costa C, Santos F, Santos L, Cabral F. Cervical ectopic preg-nancy successfully treated with local methotrexate injection. Fertil Steril2008;90:2005.e7–10.19. Prorocic M, Vasiljevic M. Treatment of heterotopic cervical pregnancyafter in vitro fertilization–embryo transfer by using transvaginal ultra-sound–guided aspiration and instillation of hypertonic solution ofsodium chloride. Fertil Steril 2007;88:969.e3–5.20. Suzuki M, Itakura A, Fukui R, Kikkawa F. Successful treatment of a het-erotopic cervical pregnancy and twin gestation by sonographicallyguided instillation of hyperosmolar glucose. Acta Obstet Gynecol Scand2007;86:381–3.21. Iloabachie GC, Igwegbe AO, Izuora KL. Cervico-isthmic twin preg-nancy carried to 37 weeks. Int J Gynaecol Obstet 1993;40:59–61.entering the entire uterine cavity and freely scraping the en-docervical canal.Placement of bilateral stitches in correspondence of thefornix to close cervical arteries showed not to be effectivein our hands. Presence of a wide vascular bed and anastomo-ses may account for the ineffectiveness of this procedure. Inconclusion, placing a nontightened high cerclage is an effec-tive maneuver for controlling bleeding and conservativelytreating cervical pregnancies.REFERENCES1. Yankowitz J, Leake J, Huggins G, Gazaway P, Gates E. Cervical ec-topic pregnancy: review of the literature and report of a case treatedby single-dose methotrexate therapy. Obstet Gynecol Surv 1990;45:405–14.2. Nolan TE, Chandler PE, Hess LW, Morrison JC. Cervical pregnancymanaged without hysterectomy. A case report. J Reprod Med 1989;34:241–3.3. Bachus KE, Stone D, Suh B, Thickman D. Conservative management ofcervical pregnancy with subsequent fertility. Am J Obstet Gynecol1990;162:450–1.4. Mashiach S, Admon D, Oelsner G, Paz B, Achiron R, Zalel Y. CervicalShirodkar cerclage may be the treatment modality of choice for cervicalpregnancy. Hum Reprod 2002;17:493–6.5. Lobel SM, Meyerovitz MF, Benson CC, Goff B, Bengtson JM. Preoper-ative angiographic uterine artery embolization in the management ofcervical pregnancy. Obstet Gynecol 1990;76:938–41.6. Nitke S, Horowitz E, Farhi J, Krissi H, Shalev J. Combined intrauterineand twin cervical pregnancy managed by a new conservative modality.Fertil Steril 2007;88:706.e1–3.7. Spitzer D, Steiner H, Graf A, Zajc M, Staudach A. Conservative treat-ment of cervical pregnancy by curettage and local prostaglandin injec-tion. Hum Reprod 1997;12:860–6.8. Hardy TJ. Hysteroscopic resection of a cervical ectopic pregnancy. AmAssoc Gynecol Laparosc 2002;9:370–1.935.e19

Succesful management of a cervical twin pregnancy: neoadjuvant systemic methotrexate and prophylactic high cervical cerclage before curettage. A case report

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Succesful management of a cervical twin pregnancy: neoadjuvant systemic methotrexate and prophylactic high cervical cerclage before curettage. A case report

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