European Journal of Obstetrics & Gynecology and Reproductive Biology 151 (2010) 199–202
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Laparoscopical management of cornual pregnancies: a report of three cases Andrea Tinelli a,*, Antonio Malvasi b, Marcello Pellegrino c, Giovanni Pontrelli b, Bruno Martulli b, Daniel Alberto Tsin d a Department of Obstetric & Gynecology, Division of Experimental Endoscopic Surgery, Imaging, Technology and Minimally Invasive Therapy, Vito Fazzi Hospital, P.zza Muratore, 73100 Lecce, Italy b Department of Obstetric & Gynecology, Santa Maria Hospital, Bari, Italy c Department of Pathology, Vito Fazzi Hospital, Lecce, Italy d Department of Gynecology, Division of Minimal Invasive Endoscopy, The Mount Sinai Hospital of Queens, NY, USA
A R T I C L E I N F O
A B S T R A C T
Article history: Received 26 November 2009 Received in revised form 18 March 2010 Accepted 31 March 2010
Objective: Cornual pregnancy refers to the implantation and development of a gestation in one of the upper and lateral portions of the uterus; authors report their experience in laparoscopic therapeutic procedures on three singleton cornual pregnancies. Study design: Three healthy women were admitted in General Hospitals with suspect of cornual pregnancies by clinical examination, increasing of b-hCG value and transvaginal ultrasonography. One of them had a haemoperitoneum. Surgeons performed all operative laparoscopies, by incision and enucleating of ectopic cornual mass, coagulating of its surrounding vessels and suturing of the uterine incision site. Results: Patients were successfully treated only by laparoscopy, post-operative recovery period was normal in all women, with no further therapeutically intervention in the follow-up course. The aftermath was uneventful at the follow-up of 2 years. Conclusion: In cornual pregnancies, the minimally invasive surgical treatment by salpingotomy or resection of the cornual region of the uterus and the suturing of the incision site, should be the option in women interested in future fertility. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Ectopic pregnancy Cornual pregnancy Angular pregnancy Interstitial pregnancy Extrauterine pregnancy Laparoscopy Gynaecological endoscopy Minimally invasive therapy Methotrexate
1. Introduction An extrauterine or ectopic pregnancy is defined as a pregnancy in which implantation occurs outside the uterine cavity: of all reported pregnancies, 0.4–2% are extrauterine [1]. A pregnancy implanted in the proximal portion of the fallopian tube, that is within the muscular wall of the uterus, is called an interstitial or cornual pregnancy. Anatomically, the interstitial part of the fallopian tube is 0.7 mm wide and approximately 1–2 cm long with a slightly tortuous course. The interstitial or cornual pregnancy is defined by its implantation site and is found in the interstitial region between the proximal portion of the fallopian tube and the musculature of the uterus. The interstitial ectopic pregnancy is located in the intramural portion of the tube covered by myometrium [2]. A cornual pregnancy can be further sub-classified as either ‘‘angular’’ or ‘‘interstitial’’: the former is one that implants medial to the insertion of the round ligament as it crosses the utero-tubal junction, while the latter is one that implants lateral to the round ligament at this junction [3]. Generally, authors
* Corresponding author. Tel.: +39 0339 2074078; fax: +39 0832661511. E-mail address:
[email protected] (A. Tinelli). 0301-2115/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ejogrb.2010.03.032
use the terms ‘‘cornual’’ and ‘‘interstitial’’ interchangeably with only rare mention of ‘‘angular’’ pregnancies. Due to its rarity, the available literature on cornual pregnancy is from case reports and small series. The general incidence of ectopic gestation is approximately 20 per 1000 pregnancies and cornual pregnancies account for 3% of ectopic gestations [3,4]. The mortality rate for interstitial pregnancies remains at 2.0–2.5%. A cornual gestation is an urgent and dangerous medical issue [4]. The authors describe a series of laparoscopic treatment of cornual pregnancies, followed by discussion regarding the diagnostic and treatment modalities of cornual pregnancies based on a review of the existing literature. 2. Materials and methods The authors report their experience of laparoscopic therapeutic procedures on three singleton cornual pregnancies in healthy Caucasian women. 2.1. Case 1 A 34-year-old woman with an uneventful past gynaecological history, and with multiple pharmacological allergies, was admitted for suspected ectopic pregnancy, with amenorrhoea for 10 weeks
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and irregular uterine bleeding. The clinical history showed a menarche at 12 years, oligoamenorrhea and estrogen-progestin contraception in the past; she had had a spontaneous abortion at 8 weeks’ gestation and a caesarean section at 40 weeks for cervical dystocia. On admission the beta human chorionic gonadotrophin (bhCG) was 8900 IU/ml. Ultrasonographic (US) examination revealed: a normal-volume retroverted uterus, with an endometrial thickness of 1.8 mm without a gestational sac, and a uterine fundal subserosal anterior ovoid mass, with disechoic density of 2.5 cm of diameter, highly vascularized, while the adnexa showed no substantial anomalies and no fluid in the pouch of Douglas. We monitored the patient daily by US and b-hCG measurement, till the serum value reached 11,600 IU/ml and a mass diameter of 3 cm. The patient refused medical treatment because of her multiple pharmacological allergies. After a signed detailed informed consent, laparoscopy was performed by direct visual access entry at the level of the umbilicus, with a 10 mm diameter optical trocar (Endopath Xcel Bladeless, Ethicon Endo-Surgery, Johnson & Johnson Company, USA) inserted through an intra-umbilical vertical incision, followed by the insufflation of a carbon dioxide pneumoperitoneum. Inspection of the abdominal cavity was made by a zerodegree laparoscope (Karl Storz, Tuttlingen, Germany), connected to a video monitor. One supra-pubic ancillary trocar of 10 mm diameter was inserted in the midline, and two others, each of 5 mm diameter, in each iliac fossa, laterally to the inferior epigastric vessels. All the pelvic structures were inspected and so was the abdomen. The pelvic inspection showed a high-volume hypervascularized mobile uterus, with a 3 cm subserosal fundal congested and soft left mass (Fig. 1), adherent to the left adnexa. To best perform cornual pregnancy removal, we placed a transvaginal uterine manipulator. In order to dissect the cornual pregnancy mass from the surrounding myometrium, we performed a dorsal incision with a monopolar crochet needle and removed it with a Collins forceps. For its enucleation and for the haemostasis of the small vessels of the vascular network around the cornual pregnancy, we used PK Dissecting Forceps (Gyrus PlasmaKinetic, AMS, USA). After washing and accurately checking the incision site the myometrium was sutured using 3 single intracorporeal ‘‘U’’ stitches, of 0 absorbable monofilament poliglecaprone, finally apposing an anti-adhesion barrier (Tabotamp, Ethicon, Norderstedt, Germany). 2.2. Case 2 A 37-year-old woman with an uneventful past gynaecological history was admitted for suspected ectopic pregnancy with
Fig. 2. Case 2: the ectopic pregnancy is detectable by a right highly vascularized uterine ovoid mass, in a fundal subserosal position.
amenorrhoea for 11 weeks. The clinical history showed a menarche at 11 years, and two spontaneous deliveries at term 5 and 7 years ago. The b-hCG was 9560 IU/ml. The US examination revealed: a normal-volume anteverted uterus, with an endometrial thickness of 1.5 mm without a gestational sac, and a uterine fundal subserosal anterior right ovoid mass, with disechoic density of 2.9 cm diameter, highly vascularized, no ovarian anomalies and no liquid in the pouch of Douglas. The patient was monitored daily by US and b-hCG measurement, till the serum value reached 12,500 IU/ml and a mass diameter of 3.3 cm. The patient refused the medical treatment with methotrexate and decided to receive a laparoscopy, signing a detailed informed consent. The laparoscopy was performed by Veress needle entry and pneumoperitoneum, at the level of the umbilicus, followed by a single-use, conical, blind blunt-tipped trocar (Ethicon, Johnson & Johnson, Somerville, NJ, USA) insertion. Inspection of the abdominal cavity was made by a zero-degree laparoscope (Karl Storz, Tuttlingen, Germany). The ancillary trocars were positioned similarly to the first case, and abdominal-pelvic inspection showed a high-volume hyper-vascularized mobile uterus, with a 3.3 cm right subserosal fundal congested and soft left mass (Fig. 2). After positioning a transvaginal uterine manipulator in order to dissect the cornual pregnancy, we performed a dorsal incision of the uterus by a monopolar crochet needle, removing the pregnancy with a Collins forceps, as in the first case. For the haemostasis of the small vessels we used bipolar dissecting forceps (BiClamp, Bipolar Electrosurgical Coagulation, ERBE, Germany). After washing and accurately checking the incision site, the myometrium was sutured by 4 single ‘‘U’’ stitches of 0 absorbable monofilament poliglecaprone. 2.3. Case 3
Fig. 1. Case 1: the ectopic pregnancy is detectable by a left highly vascularized uterine ovoid mass, in a fundal subserosal position.
A 31-year-old healthy woman was urgently admitted with a haemoperitoneum and amenorrhea of 7 weeks. The clinical history showed a menarche at 13 years and a caesarean section at 39 weeks. The b-hCG was 7600 IU/ml. The US examination revealed: a massive haemoperitoneum, and a normal-volume anteverted uterus, with an endometrial thickness of 1.2 mm without a gestational sac. Resuscitation was quickly performed using intravenous blood transfusion of more than 2 units of packed cells and fluids, and an urgent laparoscopy was performed, after signed detailed informed consent. Laparoscopy was performed by a direct visual access method, as in the first case. Pelvic inspection showed a massive haemoperitoneum, and a high-volume hyper-vascularized mobile uterus,
A. Tinelli et al. / European Journal of Obstetrics & Gynecology and Reproductive Biology 151 (2010) 199–202
Fig. 3. Case 3: the ectopic pregnancy is detectable by a left ruptured vascularized uterine mass, with a massive haemoperitoneum.
with a left ruptured subserosal cornual pregnancy (Fig. 3). In order to dissect the bleeding zone of cornual pregnancy, we performed its removal and diathermocoagulation of the ectopic pregnancy by bipolar clamp. Using PK Dissecting Forceps (Gyrus PlasmaKinetic, AMS, USA) we achieved haemostasis of the small vessels of the vascular network around the cornual pregnancy. After washing and accurately checking the incision site, we washed the abdominopelvic cavity with physiologic fluids and sucked up all the blood lost in the haemoperitoneum. 3. Results The operative time was, on average, 45 min and the blood loss during the procedure was 160 ml; the third patient had 650 ml of haemoperitoneum. Surgeons added an intramuscular methotrexate administration to women after the operation, to ensure complete resolution of the placenta. All patients had normal postoperative recovery and were discharged the day after laparoscopy, with no additional therapy. Biochemical follow-up showed a b-hCG measurement of 0 after 15 days. Histological examination confirmed the extrauterine pregnancy. Follow-up by ultrasound and hysteroscopy did not reveal anomalies after 2 years. During this time, patients chose to use an estrogen-progestin contraceptive. 4. Comment Cornual pregnancy remains one of the most difficult gestations to diagnose and treat. Although cornual pregnancies have reached term in some reports, most are diagnosed at much earlier gestational age, either by US evaluation or by b-hCG measurement and the classic triad of symptoms of ectopic pregnancy, including: amenorrhoea, vaginal bleeding, and abdominal pain [5]. The last two do not arise in some unruptured cornual pregnancies. When uterine rupture has been confirmed, it is important to act immediately, because of the risk of considerable haemorrhage. US findings consistent with cornual gestation include an eccentrically located gestational sac surrounded by asymmetrical myometrial tissue with a distinct and separate uterine cavity, and the ‘‘interstitial line sign’’, an echogenic line that extends into the upper regions of the uterine horn and borders the margin of the intramural gestational sac, representing either interstitial tube or endometrium, which depends on the age and size of the gestation; this sign had 80% sensitivity and 98% specificity for the sonographic diagnosis of interstitial pregnancy [6]. Timor-Tritsch et al. proposed ultrasound criteria for the diagnosis of interstitial ectopic pregnancy: an empty uterine cavity, a gestational sac located eccentrically and 5 mm)
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myometrial layer surrounding the gestational sac; when compared with the surgical/pathological diagnosis, application of these criteria yielded 90% specificity, but only 40% sensitivity [7]. Regarding the use of Magnetic Resonance Imaging (MRI) in cornual pregnancy diagnosis, reports affirm that this test allows for the differentiation of an intrauterine pregnancy implanted in a rudimentary horn from an interstitial pregnancy, because in the former case the gestational sac is situated outside the myometrium but in the latter case the gestational sac is surrounded by myometrium; it is appropriate for use in non-urgent cases and for evaluation of cases in which ultrasound scans have been inconclusive [8]. Concerning treatment, when the cornual pregnancy is correctly identified at an early stage and is unruptured, medical management can be attempted, with the potential advantage of avoiding surgery and with an increased likelihood of preserving fertility. Systemic injection of methotrexate (MTX) is the most extensively studied medical regimen: this drug has been administered either a single dose or in a multiple-dose fashion with some success, and also administered in a localized fashion, with both US and laparoscopic guidance [9]. In a review of 11 cases a 91% success rate was reported: systemic methotrexate achieves an 80–91% success rate. The overall failure rate for methotrexate treatment in ectopic pregnancy is also quite high, at 35% in the literature and another problem is possible adhesion formation after only MTX treatment, with long-term alteration of pelvic anatomy [10]. Other medical treatments showing some success include both etoposide and potassium chloride, both injected directly into the gestational sac during ultrasonographic visualization. With pharmacological treatments, neither gestational sac size nor serum b-hCG levels can be used to predict success for cornual gestations, but only to monitor the daily events [11]. Although medical management is a useful option for treating cornual pregnancies, it is not without its drawbacks [12]. In fact, because of the potential for catastrophic outcomes associated with failure of medical management, surgery remains the mainstay of treatment for cornual pregnancy; in the past, laparotomy with either hysterectomy or cornual resection was advocated. Recently, conservative options such as laparoscopic management or treatment by hysteroscopy have been studied. In women interested in future fertility, minimally invasive treatment by salpingotomy or resection of the cornual region of the uterus and the suturing of the incision site has been the option of choice; with the improvement of surgical skill these operations have been safely performed by laparoscopy, with good reproductive outcomes and no further therapies [13]. The surgical team and the operating room have to be ready for an emergency laparotomy since massive bleeding may occur during surgery, which must be resolved by laparotomy. Minimally invasive conservative treatment options reported in the literature include: cornuostomy, rather than cornuectomy [3,14,15], hysteroscopic treatment, either alone or under ultrasonographic guidance [16,17]; moreover, combinations of hysteroscopy under laparoscopic surveillance have also been used, followed by dilation and hysteroscopic final evacuation [18,19]. Generally, laparoscopy is performed to visually confirm the cornual gestation and to ensure immediate access to the pelvis in case of acute haemorrhage caused by uterine rupture [20], even if some expert laparoscopists have reported success with the laparoscopic suture loop or automatic stapling device to perform a cornual wedge resection [21]. One of the drawbacks to cornual resection and suturing is the use of a full-thickness uterine wall (as hysterotomy) and when electrosurgery is used, the depth of a thermal damage is difficult to assess. The surgeons’ attention is focused on limiting the lateral spread by the proper tool selection and its use in a limited and
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cautious manner, since this may prompt many surgeons to recommend caesarean delivery in subsequent pregnancies to decrease the known risk of uterine rupture [22]. Another drawback is the ultimate decrease in fertility rates for women undergoing this necessary procedure, even if the current trend is to use conservative surgical alternatives to cornual resection in an attempt to increase future fertility and decrease the risk of uterine rupture during a subsequent pregnancy. For women with unruptured cornual pregnancy who wish to preserve future fertility, conservative surgery by linear salpingotomy is considered the preferred option for the management of ectopic pregnancy. Conservative endosurgery has been reported to have advantages in terms of efficacy, safety, cost and subsequent reproductive outcome in comparison with laparotomy [23,24]. A conservative strategy that would address all of the concerns associated with the treatment of a cornual pregnancy must include: correct early diagnosis of cornual pregnancy, an unruptured gestational sac located near the uterine cavity, maternal desire for future childbearing, hemodynamic stability, and detailed informed consent to the significant risks associated with the attempt at conservation of the cornual region of the uterus. This conservative treatment in women with cornual pregnancy appears to be superior to the laparotomy option, either in unruptured cornual pregnancy, or in ruptured corneal pregnancy with haemoperitoneum. Considering the rarity of this condition, it may take some time for advocates of these new regimes to gather data to this effect and further technical evaluations of this modality of treatment will need large number of cases to establish such claims. Conflict of interest The authors certify that there is no actual or potential conflict of interest in relation to this article and no financial interests or connections, direct or indirect, or other situations that might raise the question of bias in the work reported or the conclusions, implications, or opinions stated – including pertinent commercial or other sources of funding for the individual author(s) or for the associated department(s) or organization(s), personal relationships, or direct academic competition. References [1] Goyaux N, Leke R, Keita N, Thonneau P. Ectopic pregnancy in African developing countries. Acta Obstet Gynecol Scand 2003;82:305–12.
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