Spontaneous Cornual Pregnancy after Homolateral Salpingectomy for an Earlier Tubal Pregnancy: A Case Report and Literature Review

Spontaneous Cornual Pregnancy after Homolateral Salpingectomy for an Earlier Tubal Pregnancy: A Case Report and Literature Review

Case Report Spontaneous Cornual Pregnancy after Homolateral Salpingectomy for an Earlier Tubal Pregnancy: A Case Report and Literature Review Nicola ...

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Case Report

Spontaneous Cornual Pregnancy after Homolateral Salpingectomy for an Earlier Tubal Pregnancy: A Case Report and Literature Review Nicola Pluchino, MD*, Filippo Ninni, MD, Stefano Angioni, MD, Arianna Carmignani, MD, Andrea Riccardo Genazzani, MD, PhD, and Vito Cela, MD, PhD From the Department of Reproductive Medicine and Child Development, Division of Gynecology and Obstetrics, University of Pisa (Drs. Pluchino, Ninni, Carmignani, Genazzani, and Cela), and Department of Gynecology and Obstetrics, University of Cagliari (Dr. Angioni), Italy.

ABSTRACT Cornual pregnancy is an infrequent pathological condition with severe prognosis if not adequately recognized. Ipsilateral salpingectomy represents a unique risk factor for this clinical entity. This article reports a laparoscopically treated spontaneous cornual pregnancy after homolateral salpingectomy for an earlier tubal pregnancy in a condition of hemodynamic instability as a result of cornual rupture. We include a review of the literature, underlining the feasibility of a laparoscopic approach and new treatment options combining medical and surgical tools with specific attention to their impact on future fertility and on risk of uterine rupture in a future pregnancy. Journal of Minimally Invasive Gynecology (2009) 16, 208–211 Ó 2009 AAGL. All rights reserved.

Spontaneous cornual pregnancy represents about 1% of the ectopic pregnancies (incidence of 1/2500–5000 live births) with severe prognosis if prompt medical intervention is not established (mortality of 2.5%–4.0%) [1]. This unique anatomic site of implantation may lead to a delay in diagnosis and rupture of uterus that may occur in 20% of cases that progress beyond 12 weeks of amenorrhea [2]. Risk factors for ectopic pregnancy include past pelvic inflammatory disease, previous pelvic surgery, uterine anomalies, the use of assisted reproductive techniques [3], and ipsilateral salpingectomy, which is a risk factor unique for cornual pregnancy [4]. With the advent of assisted reproductive technologies we observed a higher frequency of ectopic pregnancies than after natural conception, specifically increasing from 1.9% up to 7.3% for cornual/interstitial implantation [3].

The authors have no commercial, proprietary, or financial interest in the products or companies described in this article. Corresponding author: Nicola Pluchino, MD, Department of Reproductive Medicine and Child Development, Division of Gynecology and Obstetrics, University of Pisa, Via Roma 35, 56100 Pisa, Italy. E-mail: [email protected] Submitted August 26, 2008. Accepted for publication November 6, 2008. Available at www.sciencedirect.com and www.jmig.org 1553-4650/$ - see front matter Ó 2009 AAGL. All rights reserved. doi:10.1016/j.jmig.2008.11.008

However, the occurrence of this condition in the same side of a previous salpingectomy is a very intriguing pathological condition, in case of spontaneous pregnancy. In fact, to our knowledge few cases of spontaneous cornual pregnancies after homolateral salpingectomy are reported in the literature [4], whereas all the other case series are subsequent to embryo transfer techniques [5,6]. Most of them were treated laparatomically and only 1 previous case report describes a laparoscopically treated unruptured cornual pregnancy as a result of spontaneous conception after earlier salpingectomy for tubal pregnancy [7]. This article reports a laparoscopically treated spontaneous cornual pregnancy after homolateral salpingectomy for an earlier tubal pregnancy at 7 weeks of gestation in a condition of hemodynamic instability as a result of corneous rupture. In addition, we include a review of the literature, underlining the feasibility of a laparoscopic approach for patients with rupture of cornual pregnancy and subsequent hemodynamic instability. Case Report A 34-year-old woman (gravida 1, para 0) arrived at the emergency department with acute abdominal pain 7 weeks after her last menstrual period. She had been married for almost 2 years but used no contraception because she planned

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Spontaneous Cornual Pregnancy After Homolateral Salpingectom

on having children. Her medical history included a left laparoscopic salpingectomy 12 months before for a left tubal ectopic pregnancy. On initial physical examination her vital signs were: blood pressure 90/55 mm Hg; heart rate 105 beats/min; temperature 37.3 C; and oxygen saturation 98% in room air. She was in pain and anxious but conscious and well-oriented. On palpation her abdomen was tense and distended. Other physical examination findings were unremarkable. The patient’s hemoglobin level, white blood cell count, and platelet count on admission were 10.5 g/dL, 12.7 ! 103mL, and 198 ! 103mL, respectively. Electrocardiography and other coagulation markers revealed normal findings. Ultrasonography showed hemoperitoneum and the urine pregnancy test result was weakly positive. To preserve her future fertility a minimally invasive surgical approach was planned. Laparoscopic findings established a diagnosis of a ruptured 4- ! 2.5-cm left cornual pregnancy (Fig 1). Free blood in abdomen was accurately removed by vacuum (about 1500 mL) and repeated washings were made. Then we decided on a cornual incision followed by exposition and removal of the products of gestation using bipolar forceps and laparoscopic endoscissors. Subsequent hemostasis was maintained with bipolar electrocoagulation. To reduce coagulation to a minimum, an interrupted suture was also applied at the bleeding area. The resected cornual pregnancy was then removed with an Endopouch. We did not remove the contralateral tube to preserve the natural conception of this patient. The postoperative course was uneventful. She was discharged from hospital on postoperative day 2 and was followed up until her b-human chorionic gonadotropin (hCG) level decreased below 5 mIU/L. No complications were detected during the follow-up period. Discussion Cornual pregnancy is a challenging and potentially dangerous situation that may lead to massive hemoperitoneum and, in certain dramatic cases, rupture of uterus. Traditionally it was treated by cornual resection or even hysterectomy by laparotomy, probably as a result of the delayed diagnosis. Symptoms occur usually between 9 and 12 weeks of amenorrhea because of the distensibility of the myometrium in patients with abdominal pain or discomfort and abnormal vaginal bleeding [8]. Once the diagnosis is made, the literature reports a huge variety of treatment regimens, both medical and surgical, each of them exploring new protocols and experimenting with new solutions. Of interest, successful expectant management in a cornual pregnancy was described with conveniently decreasing ß-hCG blood levels [9]. Medical treatment for ectopic pregnancy seems to be more efficient if the ß-hCG level is less than 1000 mIU/mL; if the ectopic pregnancy is not symptomatic, less than 4 cm in diameter, and without cardiac activity; or if the ectopic

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Fig 1. Laparoscopic view of the left cornual pregnancy after the removal by vacuum cleaner of abdomen free blood. The absence of the ipsilateral tube and the anatomy of the pelvis is clearly visible.

pregnancy cannot be visualized by transvaginal ultrasonography [10–12]. Systemic administration of methotrexate given 1 mg/kg/ day intramuscularly or intravenously given on days 1, 3, 5, and 7 with 7 days between courses, with multiple doses being more efficient than a single dose [13], is the most studied medical approach with a regimen of either systemic or local injection or a combination of both. Furthermore, it seems that local injection is more efficient than systemic treatment [8]. Otherwise, we prefer surgical treatment if ß-hCG blood levels are greater than 10 000 IU/L (.5000 IU/L as a relative indication if they are increasing) and if the patient is hemodynamically unstable and feeling pain, it does not matter whether cardiac activity exists. Hysterectomy–performed laparotomically or vaginally via laparoscopic guidance–was proposed in the past as the surgical approach of choice to cornual pregnancy for women who are not interested in childbearing, leaving resection of cornual region as the surgical option for those who desire to preserve future fertility [14]. Laparoscopy–when feasible–has become the gold standard treatment for cornual pregnancies in the last few years [15] and generally has replaced laparotomic procedures [16]. Laparoscopic procedures can be implemented by different techniques to minimize blood loss, an event that can have catastrophic results because of the rich blood supply of the area: suture-loop tourniquets applied through the avascular area of the broad ligament [17] or temporary tourniquet around the cornual mass [18], pretied ligatures (Endoloop) [19], automatic staplers opportunely fixed [20], ligation of the ascending branches of the uterine vessels [21], apposition of fibrin glue [22], and mostly the use of diluted intramyometrial vasopressin injection [23]. Rupture and subsequent hemodynamic instability are no longer an absolute contraindication to laparoscopic approach for cornual pregnancy [24] and the always renewing interest laparoscopic field makes possible the management of dramatic conditions, such as hypovolemic shock [25] and/or

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massive hemoperitoneum [26], in a way that is simple, safe, and successful. In the last few years the need to increase future fertility and to decrease risk of uterine rupture introduced the use of more conservative surgical alternatives, such as cornuostomy rather than cornuectomy [27,28], and hysteroscopy alone or in association with ultrasonography [29,30]. However, no consensus exists regarding which is the optimal route of a future delivery [31–33]. Combination of surgical and medical treatment was also reported. Specifically, authors described a novel treatment regimen in which the first step consists of removal of most of the cornual pregnancy by dilatation and curettage via laparoscopic vision and the second step consists of a single methotrexate dose injection effective in terminating the pregnancy as evaluated by transvaginal ultrasonography and blood b-hCG levels [34]. Some authors developed and successfully performed a laparoscopic and ultrasound-guided vaginal evacuation [32] and a conservative treatment strategy is also reported via hysteroscopic suction [35]. In this case report, we highlight that cornual incision and subsequent pregnancy removal can be an effective treatment as previously described [36]; this surgical procedure could also reduce the hemorrhagic risk during a cornual resection. However, a hemostatic suture was deemed necessary to avoid excessive coagulation; therefore, in our opinion, knowledge of laparoscopic suturing is a prerequisite for the treatment of cornual pregnancies. Conclusions Previous salpingectomy is a risk factor for cornual pregnancy, although they are extremely rare in the case of spontaneous pregnancy. Because of limited data in the literature, recommendations about an ideal management option for cornual pregnancies are not possible and treatment should be individualized based on the clinical result and patient preference regarding future fertility, when feasible. References 1. Department of Health. Confidential enquiry into maternal health. Why mothers die 2000-2002? HMSO London. Available at: http://www. cemach.org.uk. Accessed January 22, 2009. 2. Felmus LB, Pedowitz P. Interstitial pregnancy: a survey of 45 cases. Am J Obstet Gynecol. 1953;66:1271–1279. 3. Pisarska MD, Carson SA. Incidence and risk factors for ectopic pregnancy. Clin Obstet Gynecol. 1999;42:2–8. 4. Simpson JW, Alford CD, Miller AC. Interstitial pregnancy following homolateral salpingectomy. Am J Obstet Gynecol. 1961;82:1173–1179. 5. Agarwal SK, Wisot AL, Garzo G, Meldrum DR. Cornual pregnancies in patients with prior salpingectomy undergoing in vitro fertilization and embryo transfer. Fertil Steril. 1996;65:659–660. 6. Sharif K, Kaufmann S, Sharma V. Heterotopic pregnancy obtained after in-vitro fertilization and embryo transfer following bilateral total salpingectomy: case report. Hum Reprod. 1994;9:1966–1967. 7. Trivedi AN, Roman J. A case report of a laparoscopically treated cornual pregnancy as a result of spontaneous conception after prior salpingectomy for tubal pregnancy. J Obstet Gynaecol. 1998;18:602–603.

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