{"paper_id":"8bba2c6f-393b-47f2-9777-afafdbbd5c81","body_text":"CASE REPORT\nSubserosal intramural ectopic pregnancy in an adenomyotic\narea following assisted reproduction treatment\nAhmed Abdel-Gadir & Kishor Shah &\nOluseye O. Oyawoye & Bina P. Chander\nReceived: 6 April 2009 / Revised: 29 April 2009 / Accepted: 30 April 2009 / Published online: 30 May 2009\n# Springer-V erlag 2009\nAbstract A 38-year-old woman presented for early preg-\nnancy ultrasound scanning 6 weeks and 4 days follow-\ning an assisted reproduction treatment cycle. She had ß\nhuman chorionic gonadotrophin (ßhCG) blood level of\n10,853 IU/L 2 weeks before presentation. She gave\nprevious history of termina tion of pregnancy, myomec-\ntomy and bilateral salpingectomy. The uterus was\nretroverted with multiple f ibroids and non-homogenous\nmyometrium in many areas. The endometrium was\n21.1 mm thick with no intrauterine pregnancy. An\ninitial diagnosis of cornual/interstitial ectopic pregnancy\nwas made. However, 3D images rendering and the\nmultiplanar technique showed a 27.5-mm gestation sac,\nmedial and above the interstitial part of the right tube,\nwith 7.6-mm-long foetal pole. ßhCG and progesterone\nblood levels on the same day were 19,551 IU/L and\n43.2 nmol/l, respectively. The patient opted against\nmethotrexate treatment. An ectopic pregnancy bulging\nout of the fundal area was excised laparoscopically.\nHistopathological assessment showed chorionic villi\nsurrounded by myometrium, as well as foci of adeno-\nmyosis, reaching the outer serosa. To our knowledge,\nthis is the second case of subserosal intramural ectopic\npregnancy to be reported and the first in a subserosal\narea of adenomyosis.\nKeywords Intramural pregnancy . Subserosal pregnancy .\nSubserosal adenomyosis\nIntroduction\nIntramural pregnancies are rare and have been described\nas conceptions within the uterine wall, surrounded by\nmyometrium without connection to the endometrial\ncavity, fallopian tubes or round ligaments [ 1, 2]. A\nmortality rate of 2.5% has been quoted with such\npregnancies [ 3]. Predisposing risk f actors include assisted\nreproduction treatment (ART), uterine surgery, salpingec-\ntomy and adenomyosis.\nMaking the right diagnosis could be difficult with\ntransvaginal ultrasound scanning, and cornual or interstitial\npregnancies might be diagnosed instead. In most cases, the\ncorrect diagnosis is made only intraoperatively [ 4]. A high\ndegree of suspicion is needed for all these varieties of\nectopic pregnancies; otherwise, a diagnosis of pregnancy of\nunknown location would be made. One report suggested\nthat an interstitial ectopic pregnancy s hould be suspected\nwhen the ß human chorionic gonadotrophin (ßhCG)\nlevel >2,000 IU/L with an empty uterus [ 5]. Further-\nmore, three criteria have been set to diagnose interstitial\npregnancies: The uterine cavity should be empty, with the\nectopic sac >1 cm from its most lateral edge, and it should\nbe surrounded by a thin myometrial layer [ 6]. Addition-\nally, the interstitial echogenic line was found to be 80%\nGynecol Surg (2009) 6:267 –271\nDOI 10.1007/s10397-009-0494-2\nA. Abdel-Gadir ( *) : B. P . Chander\nLondon Female and Male Fertility Centre, Highgate Hospital,\n17-19 View Road,\nLondon N6 4DJ, UK\ne-mail: AhmedAGadir@aol.com\nK. Shah\nIndependent Histopathology Services,\n142-144, New Cavendish Street,\nLondon WIW 6YF, UK\nO. O. Oyawoye\nDepartment of Obstetrics and Gynaecology,\nNewham University Hospital,\nGlen Road, Plaistow,\nLondon E13 8SL, UK\n\nsensitive and 98% specific in diagnosing interstitial\npregnancies [ 7]. Furthermore, the myometrium surround-\ning the cornual type has been described as abnormally thin\nand less than 5 mm thick [ 8]. However, in cases of\nintramural pregnancies, the t hickness of the myometrium\naround the sac depends on the degree of invasion by the\nectopic trophoblasts. The single published case of sub-\nserosal intramural ectopic pregnancy [ 9] is a good\nexample because of such deep implantation. Irrespective\nof all efforts, it might not be possible to differentiate\nbetween intramural and interstitial/cornual pregnancies.\nConservative medical treatment with systemic or local\nmethotrexate has been used for all sorts of ectopic\npregnancy including intramural ones. However, such\ntreatment is not always successful, and surgical intervention\nor selective uterine arteries embolisation [ 10] might be\nnecessary to deal with the persistent trophoblasts.\nCase report\nA 38-year-old woman with 4 years history of infertility\nattended our clinic for early pregnancy ultrasound\nscanning following an ART cycle performed overseas.\nTwo embryos were replaced in her uterus 6 weeks and\n4 days before. A ßhCG blood level 2 weeks before\npresentation was 10,853 IU/L. She gave history of\nprevious termination of pregnancy and two myomecto-\nmies, one laparoscopic and one through open surgery,\non two different occasions. A large 10- to 12-cm\nintramural anterior fundal fibroid was removed during\nopen surgery, and four tumours were removed during\nthe laparoscopic procedure followed by resuturing of the\nuterus laparoscopically. Bil ateral salpingectomy had also\nbeen performed, because of bilateral hydrosalpinges,\nfollowing three unsuccessful ART attempts. She was not\nin pain and had no vaginal bleeding. ßhCG and serum\nprogesterone levels were 19,551 IU/L and 43.2 nmol/l,\nrespectively, on the same day.\nTransvaginal scan examination showed a retroverted\nuterus with multiple small fibroids, the largest measuring\n4.6×3.8 cm. The myometrium was non-homogenous in\nmany areas suggestive of adenomyosis. The endometrium\nwas 21.1 mm thick. There was no intrauterine pregnancy.\nFibroid shadowing interfered with the quality of the\npictures during 2D scanning. However, a gestational sac\n27.5 mm in diameter was seen at the fundal area beside the\nlargest fibroid and 6.4 cm deep relative to the vaginal vault\n(Fig. 1). An initial diagnosis of cornual/interstitial pregnan-\ncy was suspected. However, image manipulation with 3D\nrendering and the multiplanar technique revealed a different\npicture. The sac was seen bulging out of the fundal area\nabove and medial to the echogenic interstitial part of the\nexcised right tube (Fig. 2), a picture we have not seen\nbefore. The foetal pole was 7.6 mm long, equivalent to\n6 weeks and 3 days pregnancy, and the yolk sac was\n4.9 mm in diameter. There was no free fluid in the pelvis.\nThe diagnosis, treatment options and risks involved were\ndiscussed with the couple. The possibility of ultrasound-\nguided methotrexate injection [ 11] was excluded because of\nthe inaccessibility of the sac. The patient was not agreeable\nFig. 1 An oblique transvaginal ultrasound picture showing a\nsubserosal gestational sac with a nearby intramural fibroid. The sac\nwas high in the fundal area and 6.4 cm deep relative to the vaginal\nvault. The body of the uterus and the empty uterine cavity are not\nshown in this view\nFig. 2 A rendered coronal view of the uterus with a right fundal\nectopic pregnancy above and medial to the echogenic interstitial part\nof the right tube with the same fibroid shown in Fig. 1 nearby. The\nempty triangular uterine cavity is marked with echogenic endometri-\num and is dented at the fundus by the same fibroid\n268 Gynecol Surg (2009) 6:267 –271\n\nto use systemic methotrexate and opted for laparoscopic\ntreatment instead. The risk of antenatal and intrapartum\nuterine rupture during any future pregnancy was thoroughly\nexplained. She was very keen not to lose her uterus. The\ncouple took 2 days to discuss their options and might have\nhad a second opinion as well. They opted for laparoscopic\nexcision of the ectopic pregnancy, with the risk of a\nhysterectomy explicitly explained. She consented for this\noption as a life-saving procedure only.\nLaparoscopy revealed a large ectopic pregnancy bulging\nfrom the fundal area of the uterus with a nearby fibroid\n(Fig. 3). Meticulous dissection and cauterisation of all the\nblood vessels leading into the mass (Fig. 4) was done\nbefore it was excised down to the surface of the fundus,\nwith minimal blood loss and minimal use of bipolar\nelectrocautery for haemostasis, to reduce the risk of\nmuscles necrosis (Fig. 5). Reapproximation of the edges\nwas not done as the scar was almost level with the surface\nof the fundus. She was discharged from hospital on the\nfollowing day. The risks involved with any further\npregnancy were reemphasised to the couple during the\npostoperative visit.\nHistological examination of the excised specimen\nshowed trophoblastic tissue with chorionic villi surrounded\nby thick myometrium (Fig. 6). In addition, the muscular\nwall showed many foci of adenomyosis, some near the\nouter serosa (Fig. 7).\nDiscussion\nThis case had many of the risk factors described before,\nwhich could lead to intramural ectopic pregnancies. The\nhuge fundal bulge of the ectopic mass, as confirmed by\nlaparoscopy, indicated a subserosal intramural location\nwhich has been reported only once before [ 9]. Such\ndeep intramuscular implantat ion was confirmed histopath-\nologically and might have gone through a sinus tract\ncreated during a previous myomectomy [ 9]. This is\nespecially so as the site of the ectopic pregnancy\ncorresponded to the site of the large intramural fibroid\nr e m o v e db yo p e ns u r g e r y ,a sd o c u m e n t e di nh e rp r e v i o u s\nnotes. The role of the concurrent adenomyosis is also\nimportant in this respect [ 1, 12] and could simulate the\nFig. 3 A laparoscopic view showing the subserosal ectopic pregnancy\nprotruding through the fundal area with the same fibroid shown in\nFigs. 1 and 2, as well as a neighbouring subserous fibroid\nFig. 5 A laparoscopic view of the uterus after excision of the ectopic\nmass down to the level of fundus\nFig. 4 A laparoscopic view following forced anteversion of the uterus\nwith an intrauterine canulla. The ectopic mass was made more\nprominent by forward pressure applied on its posterior aspect with\ngrasping forceps. Note the two prominent blood vessels marked with\narrows. Other vessels were seen and cauterised on the other side and\nduring dissection before excision of the ectopic pregnancy\nGynecol Surg (2009) 6:267 –271 269\n\nincreased myometrial invasion by FIGO grade 1 endome-\ntrial adenocarcinoma in cases with, compared to cases\nwithout, adenomyosis [ 13].\nMagnetic resonance imaging (MRI) has been used to\nhelp with the diagnosis of tubal and interstitial ectopic\npregnancies with great success. Criteria similar to those\nused during ultrasound examination for the diagnosis of\ninterstitial ectopic pregnancies [ 6, 7] have been suggested\nfor MRI as well [ 14, 15]. However, the statistics given for\naccuracy of transvaginal ultrasound scanning were not very\nmuch different from those quoted for MRI. The sensitivity\nand specificity of transvaginal ultrasonography in detecting\nectopic pregnancies was 90% and 99%, respectively, with\npositive and negative predictive values of 93.5% and\n99.8%, respectively. The diagnostic accuracy was 90.9%\n[16]. In comparison, a sensitivity of 95% and specificity of\n100% in diagnosing ectopic pregnancies was quoted,\nrespectively, for MRI with an accuracy of 96% [ 17].\nAccordingly, transvaginal ultrasound remains to be the\nfirst-line diagnostic imaging technique in the management\nof ectopic pregnancies. This is especially so because of its\neasy availability as an office diagnostic procedure. We see\nan important role for MRI in cases of pregnancies of\nunknown location when ultrasound fails to show intrauter-\nine or ectopic pregnancies in patients with high positive\nßhCG blood levels. It could also be helpful if the uterus\nis distorted with multiple fibroids and a suspected\ndiagnosis of an ectopic pregnancy is not possible to\nverify or exclude because of such anatomical changes\nand the shadowing caused by fibroids. In retrospect, we\nfeel that MRI would not have added further useful\ninformation to that already provided by 3D scanning, in\nour case. The sac was seen and its exact intramural\nlocation was clearly ascertained, relative to the uterine\ncavity and the interstitial part of the right tube. This\nconfirms the important role of 3D rendering in helping\nwith the diagnosis in such difficult cases as emphasised\nbefore [ 2]. It definitely changed our initial impression of a\ncornual pregnancy, but further clinical interpretation of the\nintramural site as a subserosal ectopic pregnancy was\nmissed as only one case has been published before, and\nwe were not aware of that article at that time. The final\ndiagnosis was made laparoscopically.\nTo the best of our knowledge, this is the second case of\nsubserosal intramural ectopic pregnancy to be reported.\nSimilar to the first case [ 9], there was previous history of\nmyomectomy. However, it is the first case of subserosal\nintramural ectopic pregnancy in a subserosal adenomyotic\narea. Treatment of intramural pregnancies is difficult and no\nconsensus has yet been agreed upon. The situation is even\nmore difficult with the subserosal type as this is only the\nsecond case to be reported. However, like cornual preg-\nnancies, laparoscopy could be the preferred surgical method\nof treatment of intramural pregnancies in experienced hands\n[18]. Reapproximation of the uterine scar would be\nnecessary when the incision involves the body of the uterus\nitself. However, with the subserosal type, the bulging mass\nwas excised down to the level of the fundus and\nreapproximation of the edges of a flat scar would be\ndifficult or even impossible. Accordingly, minimal use of\nthe electrocautery would be necessary to prevent muscle\nnecrosis and further weakening of the scar. The risk of\nbleeding could be reduced beforehand by occluding all the\nblood vessels connected to the mass first, as done in this\ncase, before excising the subserosal ectopic pregnancy\nitself.\nFig. 7 A histopathological low power (×40) haematoxylin and eosin-\nstained section showing subserosal myometrium with a large focus of\nendometrial glands and stroma, typical of adenomyosis\nFig. 6 A haematoxylin and eosin-stained histopathological photomi-\ncrograph (×100) showing chorionic villi at the centre surrounded by\nthick myometrium with invading cytotrophoblastic cells\n270 Gynecol Surg (2009) 6:267 –271\n\nConflict of interest There is no actual or potential conflict of\ninterest in relation to this article.\nReferences\n1. Ginsburg KA, Quereshi F, Thomas M, Snowman B (1989)\nIntramural ectopic pregnancy in adenomyosis. Fertil Steril 51\n(2):354–356\n2. Caliskan E, Cakiro ğlu Y , Coraqkci A (2008) Expectant manage-\nment of an intramural ectopic pregnancy in a primigravid woman.\nTurkish–German Gynecol Assoc 9(4):234 –236\n3. Neiger R, Weldon K, Means N (1998) Intramural pregnancy in a\ncaesarean section scar: a case report. J Reprod Med 43:999 –1001\n4. Lee GS, Hur SY , Kown I, Shin JC, Kim SP , Kim SJ (2005)\nDiagnosis of early intramural ectopic pregnancy. J Clin Ultra-\nsound 33(4):190 –192\n5. Timmerman E, Roovers JP , Ankrum WM, Hajenius P (2008)\nInterstitial pregnancy: a rare type of ectopic pregnancy. Ned\nTijdsch Geneesdk 152(14):787 –791\n6. Timor-Tritsch IE, Monteagudo A, Matera C, Weit CR (1992)\nSonographic evaluation of cornual pregnancies treated with\nsurgery. Obstet Gynecol 79:1044 –1049\n7. Ackerman TE, Levi CS, Dashefsky SM, Holt SC, Lindsay DJ\n(1993) Interstitial line: sonographic finding in interstitial (cornual\npregnancy) ectopic pregnancy. Radiology 189(1):83 –87\n8. Fleischer AC, Manning F A, Jeanty P , Romero R (1996)\nSonography in obstetrics and gyna ecology. Principles and\npractice, 5th edn. Appleton and Lange, East Norwalk, Connecticut\n9. Park WI, Jeon YM, Lee JY , Shin SY (2006) Subserosal pregnancy\nin a previous myomectomy site: a variant of intramural pregnancy.\nJ Minim Invasive Gynecol 13(3):242 –244\n10. Y ang SB, Lee SJ, Joe HS, Goo DE, Chang YW, Kim DH (2007)\nSelective uterine artery embolisation for management of intersti-\ntial ectopic pregnancy. Korean J Radiol 8(2):176 –179\n11. Oyawoye S, Chander B, Pavlovic B, Hunter J, Abdel Gadir A\n(2003) Heterotopic pregnancy: successful management with\naspiration of cornual/interstitial gestational sac and instillation of\nsmall dose of methotrexate. Fetal Diagn Ther 18:1 –4\n12. Karakök M, Balat O, Sari I, Kocer NE, Erdogan R (2002) Early\ndiagnosed intramural ectopic pregnancy associated with adeno-\nmyosis: report of an unusual case. Clin Exp Obstet Gynecol 29\n(3):217–218\n13. Ismiil ND, Rasty G, Ghorab Z, Nofech-Mozes S, Bernardini M,\nThomas G, Ackerman I, Covens Z, Khalifa MA (2007)\nAdenomyosis is associated with myometrial invasion by FIGO 1\nendometrial adenocarcinoma. In J Gynecol Pathol 26(3):278 –283\n14. Filhastre M, Dechaud H, Lesnik A, Taourel P (2005) Interstitial\npregnancy: role of MRI. Eur Radiol 15(1):93 –5\n15. Bourdel N, Roman H, Gallot D, Lenglet Y , Dieu V , Juillard D,\nLinstrument C, Botchorishvili R, Rabishong B, Mage G, Canis M\n(2007) Interstitial pregnancy. Ultrasonographic diagnosis and\ncontribution of MRI. A case report. Gynecol Obstet Fertil 35\n(2):121–124\n16. Condous G, Okaro E, Khalid A, Lu C, V an Huffel S, Timmerman\nD, Bourne T (2005) The accuracy of transvaginal ultrasonography\nfor the diagnosis of ectopic pregnancy prior to surgery. Hum\nReprod 20(5):1404 –9\n17. Y oshigi J, Yashiro N, Kinoshita T, O'uchi T, Kitagaki H (2006)\nDiagnosis of ectopic pregnancy with MRI: efficacy of T2*-\nweighted imaging. Magn Reson Med Sci 5(1):25 –32\n18. Soriano D, Vicus D, Mashiach R, Schiff E, Seidman D,\nGoldenberg M (2008) Laparoscopic treatment of cornual\npregnancy: a series of 20 consecutive cases. Fertil Steril 9\n(3):839–843\nGynecol Surg (2009) 6:267 –271 271","source_license":"CC0","license_restricted":false}