doi:10.1111/j.1447-0756.2010.01361.

J. Obstet. Gynaecol. Res. Vol. 37, No. 5: 452457, May 2011

Spontaneous uterine rupture in the 33rd week of IVF pregnancy after laparoscopically assisted enucleation of uterine adenomatoid tumor
jog_1361 452..457

Hiroyuki Yazawa1, Sumiko Endo1, Syoutaro Hayashi1, Satoshi Suzuki2, Akiko Ito2 and Keiya Fujimori2
1

Department of Obstetrics and Gynecology, Fukushima Red Cross Hospital, and the 2Department of Obstetrics and Gynecology, School of Medicine, Fukushima Medical University, Fukushima, Japan

Abstract
Although a uterine leiomyomectomy or adenomyomectomy is an accepted procedure to treat symptoms such as dysmenorrhea or hypermenorrhea to enhance fertility, the risk of future uterine rupture is a major concern for patients who become pregnant following these surgery. Although uterine rupture very rarely occurs, this is the most feared complication in pregnancy and is associated with a high rate of maternal and fetal morbidity and mortality. A 37-year-old nulliparous woman had a 2-year history of infertility. A transvaginal ultrasound revealed multiple uterine tumors that resembled leiomyomas on the posterior and fundal walls of the uterine body. After the patient had three failed in vitro fertilization and embryo transfer treatments, the uterine tumor was enucleated, and pathologically diagnosed as an adenomatoid tumor. Five months after the operation, the patient became pregnant as a result of a fourth in vitro fertilization and embryo transfer. At the 33rd week of gestation, she complained of a sudden onset of abdominal pain. The patient was diagnosed with a ruptured uterus based on an ultrasound, and an emergency cesarean section was performed. The rupture occurred at the site of the operation scar on the posterior wall of the uterine body. The fetal legs extruded into the abdominal cavity from the uterine cavity but were enclosed within the unruptured chorioamniotic membrane. A male neonate (1956 g) was delivered without asphyxia and had Apgar scores of 8 and 9. The ruptured uterine wall could be repaired by suturing. To our knowledge, this is the rst case report of uterine rupture during pregnancy after resection of an adenomatoid tumor in the uterine body. Key words: adenomatoid tumor, enucleation, laparoscopy, pregnancy, uterine rupture. edly involved in rare instances, such as abnormal placentation, labor induction, and congenital uterine anomalies.

Introduction
Although uterine rupture rarely occurs during pregnancy, it is a catastrophic obstetric complication that is associated with a high rate of maternal and fetal morbidity and mortality. The most important factor affecting uterine rupture during pregnancy is a uterine scar created by a prior cesarean section or by myomectomy, adenomyomectomy or an operation to treat ectopic pregnancy, although other factors are report-

Case
A 37-year-old nulligravida woman presented to our hospital for having a 2-year history of infertility. One of the reasons for her infertility was that her husband failed to ejaculate during sexual intercourse. A

Received: January 28 2010. Accepted: April 28 2010. Reprint request to: Dr Hiroyuki Yazawa, Fukushima Red Cross Hospital, Department of Obstetrics and Gynecology, 11-31 Irie-cyo, Fukushima 960-8530, Japan. Email: ikyoku12@fukushima-med-jrc.jp

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Figure 1 A (a) transvaginal ultrasound, and (b) sagittal section and (c) coronal section MRI revealed multiple irregular-shaped nodular tumors on the posterior and fundal walls of the uterine body. The margin between the tumors and uterine muscle layers was unclear, with the largest tumor measuring approximately 5 cm on the posterior wall.

transvaginal ultrasound and MRI revealed multiple irregular-shaped nodular tumors on the posterior and fundal walls of the uterine body. These tumors phenotypically resembled leiomyoma nodules but the margin between tumors and uterine muscle layers was not clearly demarcated. The largest nodule was approximately 5 cm on the posterior wall, and the patient was preoperatively diagnosed with multiple leiomyomas of uterus (Fig. 1). An analysis of her husbands semen revealed mild asthenozoospermia. Because her husband could ejaculate with masturbation, AIH (articial insemination with husband semen) was performed 7 times, but this procedure did not result in pregnancy. Next, three courses of in vitro fertilization and embryo transfer (IVF-ET) with intracytoplasmic sperm injection were performed. Although good quality embryos were transferred into the uterus during these treatment cycles, she failed to become pregnant. Because it was suspected that the implantations had failed due to the uterine tumor, the next stage of treatments consisted of a tumor resection with a laparoscopic operation. As a preoperative management, gonadotropinreleasing hormone analogues (Leuplin, Takeda, Tokyo, Japan, 1.88 mg) were administered subcutaneously for 3 months. At the operation, the tumor was laparoscopically manipulated and incised longitudinally along the tumor of the posterior wall near the fundus with a monopolar needle. However, these tumor manipulations were difcult because there was no clear demarcation between the tumor and uterus (Fig. 2). A small vertical midline skin incision was made and these tumors were directly enucleated as much as possible (75 g in total) by laparotomy. The uterine wall was repaired by suturing two layers using 2-0 and 0 Vicryl. The postoperative recovery was uneventful and the patient was discharged on day 8. The nal histopathological diagnosis was a benign adenomatoid tumor. Five months after the operation, the forth IVF-ET (with intracytoplasmic sperm injection) was performed. Two 8-cell stage embryos were transferred on day 3 of fertilization into uterus, which resulted in pregnancy. Although two gestational sacs and fetal heart

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Figure 2 Laparoscopic operation. Some nodular intramural tumors were observed in the (a) posterior and (b) fundal and anterior walls of the uterus. (c) The tumors were laparoscopically extracted and incised longitudinally along the posterior wall with a monopolar needle. (d) The tumors were difcult to manipulate because there was no clear demarcation between the tumors and the uterus.

movements in each sac were recognized in the 6th week of gestation, one of the fetuses was subsequently aborted. Her singleton pregnancy was uneventful until the 33rd week of gestation. At 33.4 weeks of gestation, the patient was admitted to our hospital due to the sudden onset of intense abdominal pain that began 3 h earlier during an early morning walk with her pet dog. The pain was located in her upper abdomen. There were no signs of uterine contraction or vaginal bleeding, and her vital signs were stable. Although the abdominal ultrasound examination revealed that fetal growth, amniotic uid volume, placenta (located on the posterior wall) and fetal heart rate were all normal, the fetuss leg had extruded into the abdominal cavity beyond the muscle layer of uterine fundus (Fig. 3). She was diagnosed with uterine rupture and transported to Fukushima Medical University Hospital for an emergency cesarean section and intensive care of neonate. During the transport, ritodrine hydrochloride was administrated intravenously and the cardiotocography monitoring indicated a reassuring fetal status pattern. Immediately after arriving at Fukushima Medical University Hospital, emergency laparotomy was performed. It was determined that the uterus had ruptured on the right side of the posterior wall, at the site of the scar from the adenomatoid tumor enucleation. The unruptured chorioamniotic membrane had extruded into the abdominal cavity through a tear in the uterine

Figure 3 A transabdominal ultrasound revealed that the fetal leg was extruding into the abdominal cavity beyond the muscle layer of the uterine fundus. The patient was diagnosed with a uterine rupture.

muscle layer and contained the fetal leg inside the expanded intact amniotic bag. A cesarean section was performed with a lower uterine segment section and a male neonate weighting 1956 g was delivered. He appeared non-asphyxiated, and his Apgar scores at 1 and 5 min were 8 and 9, respectively. The placenta was located on the posterior wall and the upper edge was a

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respiratory distress syndrome occurred in the neonate. After the operation, the mother and neonate displayed uneventful puerperal and neonatal courses.

Discussion
Although uterine rupture during pregnancy is a very rare occurrence, it is a catastrophic obstetric complication that is associated with high maternal and fetal morbidity and mortality. One of the most important risk factors associated with uterine rupture in pregnancy are uterine scars caused by a previous cesarean section,1,2 or previous uterine surgeries, such as myomectomy,35 adenomyomectomy,68 hysteroscopic operation,9,10 or an operation to treat an ectopic pregnancy.11 In our case, the uterine tumor was pathologically diagnosed as an adenomatoid tumor, which is a very rare uterine tumor.12,13 To our knowledge, this is the rst case report of the uterine rupture during pregnancy after enucleation of an adenomatoid tumor in the uterine body. There are many reports concerning the relationship between uterine rupture and a previous cesarean section. In general, the rates of uterine rupture during vaginal delivery are less than 1% in women with previous low-transverse incisions, but the rates increase to 49% in women with classical or T-shaped incision.1,14 It is well known that the risks of uterine rupture in women with a previous cesarean section are signicantly reduced by a previous vaginal delivery,15 and the risk is inversely related to the length of the interdelivery intervals. The risk of women who attempt vaginal birth after cesarean section and have interdelivery intervals of less than 24 months increases 23-fold compared with women who have intervals greater than 24 months.16 In addition, the risk increases with oxytocin augmentation compared with spontaneous labors (1 vs 0.4%).17 In women with a prior cesarean section, most cases of uterine rupture occur during labor but the rupture may also occur in the antepartum period. Ramphel et al. reported seven cases of antepartum uterine rupture in women who had previous cesarean section.18 Vaknin et al. also reported seven cases of antepartum uterine rupture out of 120 636 singleton deliveries. Six of these events were associated with abnormal placentation (placenta previa and/or placenta percreta), and three events were associated with short interpregnancy intervals.19 According to a report of Gregory et al., the uterine rupture rates among 66 856 women with pervious cesarean deliveries were 0.43% (288 cases) during labor, and 0.12% (79 cases) in the antepartum period.20

Figure 4 (a,b) The rupture site was identied as the right side of the posterior uterine wall, where there was a scar from the previous adenomatoid tumor enucleation. (c) After the cesarean section was performed with a lower uterine segment section, the rupture could be repaired using 2 layers of sutures.

little apart from the ruptured site. After the cesarean section, the rupture could be successfully repaired using two layers of sutures (Fig. 4). Umbilical arterial gas analysis indicated no acidosis (pH 7.318) and no

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Rupture of an unscarred uterus during pregnancy is an exceedingly rare occurrence, with an estimated incidence of 1 in 800020 000 deliveries.16,2026 Although congenital uterine anomalies, adherent placenta, and induction or augmentation with oxytocin or prostaglandin have been listed as the main causes of the rupture of an unscarred uterus,22 a very small number of cases without discernable causes have also been reported.21,26,27 Dubuisson et al. performed a detailed analysis of several cases of uterine rupture after laparoscopic myomectomy (LM). Among the 100 patients who had delivered after a LM, there were three cases of spontaneous uterine rupture. However, only one of these ruptures occurred along the LM scar, so the authors concluded that the rate of uterine rupture after LM is 1.0%.4 In addition, 72 of these patients had trial of labor, 58 delivered vaginally, and no uterine rupture occurred during the trial labor.4 The authors commented that it may be useful to assess the quality of the scar postoperatively and to potentially monitor these patients for the management during future pregnancies, with second-look laparoscopies or MRIs in order to determine the best type of birth and delivery. It is unclear whether there is a greater risk of uterine rupture after LM than myomectomy by laparotomy. Many researchers have reported a signicant number of pregnancies without uterine rupture or only a few cases of uterine rupture after myomectomy by laparotomy.4,2830 However, Roopnarinesingh et al. reported a uterine rupture rate 5.3% for cases that delivered after myomectomy by laparotomy.31 Most cases of uterine rupture during pregnancy after myomectomy occurred early in the third trimester (2836 weeks of gestation).3 One suspected cause of uterine rupture after this operation is the excessive use of electrocautery, which results in poor vascularization and induces necrosis of the myometrium with adverse effects on scar healing.3,4,32 In addition, some case reports have indicated that uterine rupture occurred more frequently when the institute just started performing laparoscopic surgeries,3336 indicating that inexperience of the surgeons may have affected the outcomes.4 For uterine rupture after adenomyomectomy, only 22 pregnancies and three ruptured cases have been reported to date.6,7,37 In two of these three ruptured cases, an adenomyomectomy was performed by a laparoscopic operation; one patient was twin pregnancy by IVF-ET 12 months after the operation and ruptured at 30 weeks of gestation,6 and the other patient became pregnant only one month after the adenomyomectomy

and ruptured at 28 weeks of gestation.7 Therefore, a multiple pregnancy and a short interpregnancy interval seem to be risk factors for uterine rupture after adenomyomectomy. There are no systematic data concerning pregnancies after adenomyomectomy, such as a prior cesarean section or myomectomy. The uterine tumor in our patient was histologically diagnosed as an adenomatoid tumor, which is a rare type of uterine tumor. The tumor was originally described by Golden and Ash in 1945.38 It is a benign mesothelial neoplasm of the female genital tract with a relative incidence of approximately 1% of benign uterine tumors, and is most frequently observed in reproductive age.13,39,40 By ultrasound and MRI, it appeared as a solitary, circumscribed, nodular mass resembling a leiomyoma, but the edges were not as clearly demarcated as those of myoma. When the tumor was analyzed macroscopically during the operation, it appeared as a nodular tumor, but there was no clear demarcation between the tumor and uterus, such as adenomyosis of the uterus. Because our patient was 37 years old and she desired early restart of her infertility treatment, we preformed a 4th trial of IVF-ET 5 months after the operation, which resulted in pregnancy. The short interval between operation and pregnancy could be one cause of her uterine rupture during pregnancy. According to the literatures, the risk of uterine rupture seems to be higher in women with a previous myomectomy or adenomyomectomy than women with a prior cesarean section. Therefore, it is very important to seriously consider the timing of permission of pregnancy or re-initiation of fertility treatments after these procedures, taking the size, position, depth and number of the tumors and patients age into consideration, and to certainly avoid a multiple pregnancy. In cases of infertile women with uterine tumors as our patient, it is also important to obtain informed consent about the need for an adequate interval between operation and pregnancy and the risk of uterine rupture in a future pregnancy.

References
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