Caserta, Luigi

Cignini, Pietro

Dominici, Luigi

Giorlandino, Claudio

Giorlandino, Maurizio

PubMed

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Journal of Prenatal Medicine 2007; 1 (2): 29-31 29Pietro Cignini1Maurizio Giorlandino2Luigi Caserta3Luigi Dominici2Claudio Giorlandino21 Institute of Gynecology, Perinatology and Child Health,University of Rome “La Sapienza”, Rome, Italy2 Prenatal Diagnosis Center, Rome, Italy3 Department of Gynecology, Obstetrics and Human Reproduction, 2nd University of Naples , ItalyReprint requests to: Pietro Cignini, MDInstitute of Gynecology, Perinatology and Child Health,University of Rome “La Sapienza”, Viale Regina Elena 324 - 00161 Rome - ItalyTel. +39 0645 448 318Fax. +39 0645498227E-mail: pietrocignini@fastwebnet.itCase reportCase #1A 36-year-old woman (gravida 2, para 1) come to thePrenatal Diagnosis Center, Artemisia, in Rome referredby another institution for evaluating the presence of aCesarean Scar Pregnancy (CSP). Woman came at ourinstitution with no symptoms. She had a caesareansection a term of gestation 3 years prior. She was at 9weeks gestation calculated from the first day of the lastmenstrual period. Physical examination was negativeand no bleeding from the vagina was observed. Serumhuman corionic gonadotropin level was 7500 mIU/ml.Both transabdominal and transvaginal ultrasound re-vealed a CSP according to the Jurkovic criteria (6).The gestational sac, containing a yolc sac and a em-bryonic pole with cardiac motion, was located in theanterior isthmus, in the location of the previous cae-sarean scar (Fig. 1A). Within the endometrial cavity,above the gestational sac, a fluid collection was ob-served, but no communications were observed be-tween this collection and the gestational sac. Alsosonographic examination with color-flow Doppler imag-ing was performed to determine whether the pregnan-cy was implanted in the uterus or was ectopic (Fig.1B). No normal myometrium was visualized betweenthe bladder and the gestational sac; only 3 mm ofthickness separated the sac from the urinary bladder(Fig. 1).Termination of pregnancy was suggested and the pa-tient was carefully counselled with the therapeutic op-tions, including laparotomy, laparoscopy, suction evac-uation and medical treatment with methotrexate. Aftera written informed consent was obtained an explorato-ry laparotomy was performed. When the peritonealcavity was opened after the mobilization of the bladder,the lower uterine myometrial implantation was con-firmed. A hysterotomy was performed but a severe he-morrage ensued and could not be contained by utero-tonics and conservative surgical measures; therefore,a decision was made to perform an emergency subto-tal hysterectomy. The estimated blood loss was 1500The importance of early diagnosis in cesareanscar pregnancyFigure 1 - A gestational sac (GS) within the fibroid tissue of aprevious caesarean scar in a sagittal view uf the uterus (U). Thecervix (C) is empty (Fig. 1A) and the Doppler imaging with theperipheral vascularization (white arrows) (Fig. 1B).ABTHE IMPORTANCE_Cignini  13-12-2007  15:06  Pagina 29FOR REVIEW ONLY © CIC EDIZIONI INTERNAZIONALIml; woman had an uneventful postoperative time andwas discharged from the hospital after 6 days withhaemoglobin level of 9.1 g/dl. Definitive histologic ex-amination revealed the presence of mature chorionicvilli infiltrating the myometrium. Endometriosis was al-so found in the superficial myometrium. No gestationaltissue was found in the entire uterine cavity.Case #2A 32-year-old woman coming to our emergency de-partment of Obstetric and Gynecology, at the 2nd Uni-versity of Naples with vaginal bleeding. She had twoprevious cesarean sections in 2000 and 2003. Shewas at 6 weeks gestation calculated from the first dayof the last menstrual period and the serum Beta-hCGlevel was 1800 mIU/ml. Physical examination was un-remarkable.A transvaginal ultrasound exam revealed a gestationalsac of 12 mm of diameter with an embryonic pole with-out cardiac activity implanted within the scar of the twoprevious cesarean sections. The entire cervical canalwas empty and the layer between the sac and the blad-der was diminished. After the confirmation of the CSP,woman was informed about the medical and surgicaloptions to treat this condition. After a written informedconsent was obtained an operative hysteroscopy wascarried out. The cervix was dilated by an Hegar dilatorup to number 12. Then the hysteroscope was intro-duced into the uterus and the entire cavity was visual-ized to exclude any type of gestational tissue. Then weproceeded to visualize the gestational tissue in the my-ometrium of the previous cesarean scar moving thehysteroscope outward in the cervix. The gestationaltissue was pull out using placenta forceps under sono-graphic guidance; a curettage of the uterine cavity wascarried-out and the bleeding points were stopped witha hysteroscopic rolling ball.The postoperative period was uneventful and follow-upincluded daily serial measurements of serum Beta-hCG; woman was discharged when two consecutivemeasurements fell or reached a level of 10mIU/ml.Then we seen woman every 48 hrs in an ambulatorywith serum Beta-hCG and transvaginal evaluation untillevels dropping below 5 mIU/ml.DiscussionEctopic pregnancy is one of the leading cause of mor-bidity and mortality among fertile women accounting of9% of the pregnancies-related deaths (1). Among ec-topic pregnancy there is a clinical entity called CSP inwhich the implantation of the pregnancy is within the fi-broid tissue of a previous caesarean scar (2); accord-ing some authors CSP is the rarest form of ectopicpregnancy but its incidence is not yet well established(3). From 1978 the incidence is increased probably re-lated to the high percentage of caesarean sections andthe use of transvaginal ultrasound early in pregnancy(3, 4). Anyway, because of the early onset in pregnan-cy of vaginal spotting or low abdominal painful, thiscondition could be exchanged at least for a sponta-neous miscarriage (5).Due to the limited number of women affected CSP, as re-ported in literature, information concerning both the nat-ural history and treatment are scarce. Decision concern-ing the possibility to perform a conservative treatment inwomen affected by CSP cannot rely on robust and evi-dencebased data and poses challenging problems to theobstetricians. Indeed, the mode of treatment of CSP isoften related to the severity of symptoms, the serum lev-els of free-Beta-hCG or surgical experience (3).Hence, in these women issues concerning early detec-tion is of utmost importance and it is the corner-stoneto reduce heavy complications related to the CSP .(4).Also in dedicated tertiary referral medical center forearly pregnancy, only 1-5 patients per year are diag-nosed making their management extremely complex(3, 4, 6). From 1966 to 2004 about 37 case reports andfour series of CSP were described in the literature (5).Although the pathophysiology of cesarean scar preg-nancy remains to be established, it can be supposedthat in the first days of gestation the blastocyst invadesthe myometrium through a microscopic lesion presentin the cesarean scar related to a previous trauma ofsurgical procedure such as cesarean section, my-omectomy, hysteroscopy and even manual removal ofthe placenta (7-10). Seow et al. (3) supposed also apossible correlation between intrauterine device, pelvicinflammatory disease and CSP. The pathological tro-phoblastic invasion associated with CSP can lead tosevere pregnancy complications such as massive he-morrage, placenta previa, placenta accreta and uterinerupture (11) and due to these problems the terminationof pregnancy is recommended (2, 12).The likelihood of fewer complications and of preservingthe reproductive function may assumed to be related tothe week of the diagnosis. The failure rate of unsuc-cessful in women ≥ 7 gestational week subjected toevacuation therapy was indeed 80%, while in cases <7 gestational week, such a rate was 11% (5). Thesetwo cases seem to confirm this hypothesis, but alonger series of cases are needed to validate these ob-servations.In case #1, a resection of the gestational sac via la-parotomy was performed because of the clinical andbiochemical features and also because the pregnancywas at an advanced stage. A recent review seems tojustify the use of laparotomy or laparoscopy in such se-lected cases instead of medical or other surgical treat-ments (3). Profuse hemorrhaging accounted for 12.5%of all complications in the laparotomy approach (3), butthis was the first case in which reproductive functionwas not preserved.Case #2 is the second case reported in the literature inwhich CSP was treated by hysteroscopy. In this case,low levels of beta-hCG, small gestational sac, the timeof gestation and the experience of the endoscopistsuggested the use of hysteroscopy. Moreover, hys-teroscopy was suggested because the ectopic gesta-tional sac grows toward the uterine cavity, not yetdeeply in the scar. In this case, local MTX would prob-ably also be effective in treatment, but mass regres-sion is very long, ranging from 2 months to 1 year.Due to the severity of complications, it is important dodiagnose scar pregnancy as early and accurately aspossible but it is very difficult because there are othersclinical entities could be exchanged for CSP (i.e. spon-P. Cignini et al.30 Journal of Prenatal Medicine 2007; 1 (2): 29-31THE IMPORTANCE_Cignini  13-12-2007  15:06  Pagina 30FOR REVIEW ONLY © CIC EDIZIONI INTERNAZIONALItaneous abortion and/or cervico-isthmic pregnancy)and diagnosis is often not made until uterine rupture(2). Transvaginal sonography is a useful tool for diag-nosing CSP; probably in woman whose underwent aprevious cesarean section an evaluation of the scarvery early in pregnancy, could make an early diagnosisof CSP; in this way a conservative treatment of theuterus and of the reproductive function could be feasi-ble by medical or surgical approach.In conclusion the management of CSP is not well es-tablished, but according our experience a conservativetreatment of the uterus is feasible early in pregnancy,probably before 7 weeks of gestation. However, a larg-er series of women are necessary to validate this hy-pothesis. An accurate selection of patients, an in-formed consent to the conservative treatment and astrict adherence to the follow-up program are manda-tory. We also suggest an evaluation of the scar inwomen who underwent a previous Caesarean sectionvery early on in the pregnancy. Due to the extreme rar-ity of disease in pregnant women, centralization of curein tertiary centers should be firmly pursued.References11. From the Centers for Disease Control and Prevention. Ec-topic pregnancy–United States, 1990-1992. JAMA 1995Feb 15;273(7):533.12. Fylstra DL, Pound-Chang T, Miller MG, Cooper A, MillerKM. Ectopic pregnancy within a cesarean delivery scar: acase report. Am J Obstet Gynecol 2002 Aug;187(2):302-4.13. Seow KM, Huang LW, Lin YH, Lin MY, Tsai YL, Hwang JL.Cesarean scar pregnancy: issues in management.Ultra-sound Obstet Gynecol 2004 Mar;23(3):247-53.14. Maymon R, Halperin R, Mendlovic S, Schneider D, VakninZ, Herman A, et al. Ectopic pregnancies in Caesarean sec-tion scars: the 8 year experience of one medical centre.Hum Reprod 2004 Feb;19(2):278-84.15. Wang CB, Tseng CJ. Primary evacuation therapy for Ce-sarean scar pregnancy: three new cases and review. Ul-trasound Obstet Gynecol 2006 Feb;27(2):222-6.16. Jurkovic D, Hillaby K, Woelfer B, Lawrence A, Salim R, El-son CJ. First-trimester diagnosis and management ofpregnancies implanted into the lower uterine segment Ce-sarean section scar. Ultrasound Obstet Gynecol 2003 Mar;21(3):220-7.17. Miller DA, Chollet JA, Goodwin TM. Clinical risk factors forplacenta previa-placenta accreta. Am J Obstet Gynecol1997 Jul;177(1):210-4.18. McGowan L. Intramural pregnancy. JAMA 1965 May 17;192:637-8.19. Chuang J, Seow KM, Cheng WC, Tsai YL, Hwang JL.Con-servative treatment of ectopic pregnancy in a caesareansection scar. BJOG 2003 Sep;110(9):869-70.10. Godin PA, Bassil S, Donnez J. An ectopic pregnancy de-veloping in a previous caesarian section scar. Fertil Steril1997 Feb;67(2):398-400.11. Clark SL, Koonings PP, Phelan JP Placenta previa/accre-ta and prior cesarean section Obstet Gynecol 1985 Jul;66(1):89-92.12. Lam PM, Lo KW, Lau TK. Unsuccessful medical treatmentof cesarean scar ectopic pregnancy with systemicmethotrexate: a report of two cases. Acta Obstet GynecolScand 2004 Jan;83(1):108-11.Early diagnosis of cesarean scar pregnancyJournal of Prenatal Medicine 2007; 1 (2): 29-31 31THE IMPORTANCE_Cignini  13-12-2007  15:06  Pagina 31FOR REVIEW ONLY © CIC EDIZIONI INTERNAZIONALI

The importance of early diagnosis in cesarean

scar pregnancy

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The importance of early diagnosis in cesarean scar pregnancy

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