case report a case of recurrent rudimentary horn ectopic ... · veress needle approach with optical...

Case ReportA Case of Recurrent Rudimentary Horn EctopicPregnancies Managed by Methotrexate Therapy andLaparoscopic Excision of the Rudimentary Horn

Gaby N. Moawad and Elias D. Abi Khalil

Department of Obstetrics and Gynecology, George Washington University, Washington, DC, USA

Correspondence should be addressed to Elias D. Abi Khalil; [email protected]

Received 30 December 2015; Accepted 26 January 2016

Academic Editor: Michael Geary

Copyright © 2016 G. N. Moawad and E. D. Abi Khalil. This is an open access article distributed under the Creative CommonsAttribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work isproperly cited.

This report presents a case of a 31-year-oldwoman successfully treatedmedically for a noncommunicating rudimentary horn ectopicpregnancy who presented with a second, successive rudimentary horn pregnancy. Patient underwent laparoscopic excision of rightrudimentary horn and right salpingectomy after failedmethotrexate therapy. Given the potential for rupture and recurrence, seriousefforts should be made to excise a uterine rudimentary horn.

1. Introduction

A unicornuate uterus with or without a rudimentary horn isamong the rare forms of uterine anomalies. The incidence ofa pregnancy in a rudimentary horn is 1 in 76,000–150,000pregnancies [1]. We report a case involving consecutiverudimentary horn ectopic pregnancies ultimately managedby operative laparoscopy.

2. Case Presentation

A 31-year-old nulliparous presented at five weeks gestationwith severe abdominal cramping and nausea of one-dayduration. She had no prior history of abdominal surgery orpelvic inflammatory disease. At the time of presentation, herbeta-hCG was 45mIU/mL. A follow-up beta-hCG five dayslater was 657mIU/mL. A transvaginal ultrasound performedten days from her presentation demonstrated an endometrialstripe of 2.4 cm with no intrauterine pregnancy (IUP) seen.An elongated soft tissue structure suggestive of an ectopicpregnancy was noted in the right adnexa (Figure 1). At thistime, the patient was successfully treated with methotrexatewith a negative two-week B-HCG assay. Due to continuouspelvic pressure, a follow-up ultrasound was ordered a month

later and showed a persistent right adnexal mass. A hysteros-alpingogram (HSG) revealed a uterine cavity deviated to theleft with a patent ipsilateral tube suggestive of a unicornuateuterus (Figure 2). Follow-up Magnetic Resonance Imaging(MRI) confirmed a left unicornuate uterus with a noncom-municating right horn (Figure 3). Intravenous pyelogramrevealed normal upper and lower urinary tract system. Twomonths later, the patient noted a positive home pregnancytest. Her quantitative beta-hCG level was 199mIU/mL andan ultrasound revealed a 5-week rudimentary horn ectopicpregnancy with a yolk sac. Methotrexate was attempted butfailed as her follow-up day 4 and day 7 B-HCG levels wereon the rise. An MRI with contrast confirmed the diagnosisof a right rudimentary horn ectopic pregnancy. Patient wascounseled for laparoscopic excision of rudimentary horn andright ectopic pregnancy.

After placement of a Foley catheter, a V-Care uterinemanipulator was inserted in the uterus. A transumbilicalVeress needle approach with optical trocar placement wasutilized to gain peritoneal entry. Right and left lower quad-rant and suprapubic ports were then placed under directvisualization. With patient in Trendelenburg position, theright rudimentary horn ectopic pregnancy and unicornuateuterus were visualized (Figure 4). The right round ligamentwas transected. The right perirectal space was developed

Hindawi Publishing CorporationCase Reports in Obstetrics and GynecologyVolume 2016, Article ID 5747524, 3 pageshttp://dx.doi.org/10.1155/2016/5747524

2 Case Reports in Obstetrics and Gynecology

Figure 1: Transvaginal ultrasound. Red arrow: endometrial stripeof 2.4 cm and no visible intrauterine pregnancy. Yellow arrow: rightadnexal mass/ectopic pregnancy.

Figure 2: Hysterosalpingogram with unicornuate uterus pushed tothe left.

Figure 3:Magnetic Resonance Imaging. Red arrow: left unicornuateuterus. Yellow arrow: noncommunicating right horn with ectopicpregnancy.

Figure 4: Direct visualization of ectopic pregnancy in right rudi-mentary horn with left unicornuate uterus.

and a right ureterolysis was performed to the ureteric tun-nel. The uterine artery was skeletonized and ligated at itshypogastric origin. A window was opened in the posteriorleaf of the right broad ligament between the ureter and theright utero-ovarian ligament. The right fallopian tube wasreleased from the ovary and the right uteroovarian ligamentwas transected. The anterior leaf of the broad ligament wasopened to the level of the anterior vaginal fornix allowing thebladder to be reflected well away from the right rudimentaryhorn. The rudimentary horn was then transected from themain left uterine horn and the small serosal defect repairedintracorporeally with running 2.0 vicryl suture. The resectedrudimentary horn and ectopic pregnancy were placed in a10mm endobag and removed via the expanded suprapubicport site. After complete hemostasis was noted, the abdomenwas desufflated and the port sites closed.

Minimal blood loss was encountered during the case andthe patient was discharged home the same day. Pathologyconfirmed the findings of right rudimentary ectopic preg-nancy. A quantitative beta-hCG was negative two weeks afteroperation.

3. Discussion

A unicornuate uterus results from the arrested or defectivedevelopment of one of theMullerian ducts [1]. A rudimentaryhorn exists in seventy-five percent of cases. It results frompartial development of one Mullerian duct and incompletefusion with its contralateral counterpart [2]. In 1979, But-tram Jr. and Gibbons presented a classification system forunicornuate uteri that remain in use today. According to theirclassification, there are four types: A1a, A1b, A2, and B. TypeA is a unicornuate uterus with an associated rudimentaryhorn on the contralateral side (further subtyped accordingto the presence or absence of a cavity, A1 and A2, resp.).Type A1 is further subdivided into whether the rudimentaryhorn cavity is communicating with the uterus or not, A1aand A1b, respectively. Type B is a unicornuate uterus with nocontralateral rudimentary horn structure [3, 4].

Knowing that rudimentary horns are not contiguous withthe cervix, several theories have been proposed to explainthe mechanism of ectopic pregnancies. First theory describesthe retrograde entry of sperm into the fallopian tube of therudimentary horn. This would require sperm to navigatethrough the unicornuate uterus, the ipsilateral tube, andperitoneal cavity before entry into the rudimentary side.The rate of transperitoneal sperm migration is thought tobe as high as 51% in these pregnancies [5]. The secondtheory proposed by Latto and Norman in 1950 states thatmicroscopic channelsmay exist between the endometrial andrudimentary horn cavity.These would allow direct passage ofsperm from the unicornuate cavity to the rudimentary horn[6].

Fifty percent of unrecognized rudimentary horn ectopicpregnancies rupture before the third trimester [7]. Othercomplications include preterm labor, fetal malpresentation,and placenta accreta [8]. Early diagnosis is a crucial stepin preventing these complications. Combined quantitative

Case Reports in Obstetrics and Gynecology 3

hCG and sonographic assessments remain the mainstay fordiagnosis. Sonographic criteria for diagnosing rudimentaryhorn ectopic pregnancies have been described and theseincluded: a pseudopattern of an asymmetrical bicornuateuterus, absent visual continuity of tissue surrounding thegestational sac and the uterine cervix, and the presenceof myometrial tissue surrounding the gestational sac [9].Although not helpful during pregnancy, hysterosalpingogra-phy can assist in diagnosing a unicornuate uterus by showingfilling of a small, fusiform uterine cavity that tapers at theapex and is seen shifted to one side of the pelvis. It can alsodiagnose a rudimentary horn of the communicating type. Anoncommunicating rudimentary horn cannot be visualizedby HSG and these cases are usually diagnosed by MRI [10].

When diagnosed early, medical management withmethotrexate has been reported [11]; however, it does notprevent recurrence as demonstrated by the case presented.Surgical management allows for definitive treatment of theectopic pregnancy and prevents recurrence. Traditionally,rudimentary horn pregnancies have been surgically managedvia laparotomy with excision of the rudimentary horn andthe ipsilateral fallopian tube [2]. With current advancesin laparoscopic technique, it is possible to manage thesecases laparoscopically. Given the potential for rupture andrecurrence, serious efforts should be made to excise therudimentary horn. This can be done at the time of ectopicpregnancy diagnosis or few weeks after methotrexate therapywhen tissues are less fragile and have fewer tendencies tobleed.

Conflict of Interests

The authors declare that there is no conflict of interestregarding the publication of this paper.

References

[1] J. O. Schorge, J. I. Schaffer, J. Pietz et al., “Anatomic disorders,”in Williams Gynecology, chapter 18, 2008, http://www.access-medicine.com/.

[2] A. Shahid, O. Olowu, G. Kandasamy, C. O’Donnell, and F.Odejinmi, “Laparoscopic management of a 16-week rupturedrudimentary horn pregnancy: a case and literature review,”Archives of Gynecology and Obstetrics, vol. 282, no. 2, pp. 121–125, 2010.

[3] D. Reichman, M. R. Laufer, and B. K. Robinson, “Pregnancyoutcomes in unicornuate uteri: a review,” Fertility and Sterility,vol. 91, no. 5, pp. 1886–1894, 2009.

[4] V. C. Buttram Jr. and W. E. Gibbons, “Mullerian anomalies: aproposed classification (an analysis of 144 cases),” Fertility andSterility, vol. 32, no. 1, pp. 40–46, 1979.

[5] G. G. Nahum, H. Stanislaw, and C. McMahon, “Preventingectopic pregnancies: how often does transperitoneal transmi-gration of sperm occur in effecting human pregnancy?” BJOG:An International Journal of Obstetrics & Gynaecology, vol. 111,no. 7, pp. 706–714, 2004.

[6] D. Latto and R. Norman, “Pregnancy in a rudimentary horn ofa bicornuate uterus,” British Medical Journal, vol. 2, article 926,1950.

[7] G. G. Nahum, “Rudimentary uterine horn pregnancy. The20th century worldwide experience of 588 cases,” Journal ofReproductive Medicine, vol. 47, no. 2, pp. 151–163, 2002.

[8] L. Fedele, S. Bianchi, L. Tozzi, M. Marchini, and M. Busacca,“Fertility in women of unicornuate uterus,” British Journal ofObstetrics and Gynaecology, vol. 120, pp. 1007–1009, 1995.

[9] A. Tsafrir, N. Rojansky, H. Y. Sela, J. M. Gomori, and M.Nadjari, “Rudimentary horn pregnancy: first-trimester pre-rupture sonographic diagnosis and confirmation by magneticresonance imaging,” Journal of Ultrasound in Medicine, vol. 24,no. 2, pp. 219–223, 2005.

[10] T. M. Dykes, C. Siegel, and W. Dodson, “Imaging of congenitaluterine anomalies,”American Journal of Roentgenology, vol. 189,no. 3, pp. S1–S10, 2007.

[11] H. K. Sevtap, A. M. Aral, and B. Sertac, “An early diagnosisand successful local medical treatment of a rudimentary uterinehorn pregnancy: a case report,” Archives of Gynecology andObstetrics, vol. 275, no. 4, pp. 297–298, 2007.

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