Abstract
INTRODUCTION
Uterine rupture after hysteroscopic septum resection is a rare complication, and its frequency is reported to be approximately 1–2.7%. Uterine perforation and monopolar resection during hysteroscopy are well-known risk factors for subsequent uterine rupture during pregnancy.
PRESENTATION OF CASE
We present a case of recurrent uterine ruptures during consecutive pregnancies in a patient who had undergone hysteroscopic septum resection for recurrent pregnancy loss.
DISCUSSION
Recurrent uterine rupture due to hysteroscopic septum resection in pregnancy is a very rare condition. In the present case we noted that the first two uterine ruptures resulted from uterine contractions; however, the third rupture occurred spontaneously and earlier in gestation. As each uterine rupture occurred earlier than the rupture in the previous gestation, a history of uterine rupture during pregnancy should raise provider suspicion about the possibility of earlier uterine rupture recurrence.
CONCLUSION
Uterine rupture may occur in pregnancies after hysteroscopic resection of the uterine septum. However, if a patient has a history of uterine rupture during previous pregnancies, the risk of uterine rupture may increase for earlier gestational ages in subsequent pregnancies. The patient must be informed about both the risks of uterine rupture during pregnancy after hysteroscopic septum resection and that recurrent ruptures may occur at earlier gestational weeks than during previous pregnancies.
Keywords: Uterine septum, Hysteroscopy, Recurrent uterine rupture
1. Introduction
Uterine septum is the most common type of Mullerian anomaly. It is related to recurrent pregnancy loss and infertility.1,2 Spontaneous abortion is the most frequent complication in patients with a uterine septum, and 60% of these cases are associated with pregnancy-related complications.3,4 Although variable consequences have been reported during pregnancy due to the morphology of the septum, hysteroscopic resection of an intrauterine septum has been reported to decrease the rate of miscarriage from 87.5% to 44.4%.5 Currently, optimum management of the uterine septum is achieved using hysteroscopy, which is effective, safe, and rapid. However, uterine rupture during pregnancy can occasionally be a devastating clinical situation after hysteroscopic septum resection due to myometrial damage.6
Herein, we present a case of recurrent uterine ruptures during consecutive pregnancies in a patient who had undergone hysteroscopic septum resection for recurrent pregnancy loss.
2. Case presentation
A 32-year-old multiparous woman presented at 23 weeks and 4 days (Gravida: 7, Para: 3). She was admitted to our clinic with abdominal pain. Her initial admission examination was unremarkable, and her vital signs were within normal limits. The uterine fundus was palpated 4 cm beyond the umbilicus, and neither tenderness nor rebound was observed during abdominal examination. All pelvic exam findings, including speculum application for amniotomy or cervical dilatation, were within normal limits. Obstetric ultrasonography to assess fetal measurement and amniotic volume were consistent with the last menstrual date, and the placenta was located on the posterior segment of the uterus. Abdominal sonography revealed that solid organs were intact, and free fluid that would suggest abdominal bleeding was not observed. Uterine contractions were evaluated through both palpation and abdominal tocography, but the uterine tonus was normal. Under these circumstances, the patient was hospitalized for observation.
Her medical history included 2 recurrent spontaneous abortions in the first trimester in 2003. The following year, a subsequent pregnancy resulted in preterm vaginal delivery at 30 weeks gestation. The infant died on the first postpartum day. After recurrent pregnancy loss, detailed examinations revealed that the uterus was septated, and a thrombophilia pattern was discovered; the patient was heterozygous for both the methylenetetrahydrofolate reductase enzyme (MTHFR) and prothrombin genes. The uterine septum was resected by monopolar diathermia using hysteroscopy in 2005 (Fig. 1). Two years after resection (2007), the patient was admitted to the obstetrics clinic for abdominal pain at 34 weeks gestation. After initial physical examination, sonography revealed fetal death. The patient received an immediate laparotomy, and uterine rupture was observed in the fundal area corresponding to the previously resected septum. The rupture was surgically repaired. The following year, the patient presented to the emergency department with similar symptoms at 28 weeks gestation. Abdominal inspection again revealed rupture at the same site and fetal death. The rupture site was surgically repaired again. In 2009, the patient was admitted to the hospital with a history of recurrent pregnancy loss and recurrent uterine rupture. She received both laparoscopy and hysteroscopy simultaneously in order to visualize the muscular defect and reinforce it using sutures. In 2011, her sixth pregnancy also ended in spontaneous abortion at 8 weeks gestation.
Fig. 1.
Left-sided hysterosalpingography reveals a massive uterine septum. Right-sided hysterosalpingography is present after hysteroscopic septum resection.
Six hours after hospitalization, the patient complained of abdominal pain. Her physical examination revealed abdominal tenderness and rebound. Her blood pressure and pulse were 80/50 mmHg and 130 beats/min, respectively. Fetal bradycardia and massive abdominal fluid collection were observed upon emergency sonography. Exploratory laparotomy and abdominal inspection revealed that the uterine fundus was ruptured over 7 cm through the transverse axis, and the amniotic sac remained with the fetus inside. Next, 1000 cc of coagulated blood was aspirated from the pelvis, and a 630 g viable fetus was delivered from the rupture site. After delivery of the placenta, the rupture site was evaluated and repaired using continuous 1/0 vicryl sutures. The patient was transfused with 2 units of packed red blood cells during the operation and one unit during the postoperative period. She was discharged on the fifth day after admission uneventfully, but the infant died in the neonatal intensive care unit on the first postpartum day (Fig. 2).
Fig. 2.
Third uterine rupture site extends from one cornu to the other.
3. Discussion
Uterine rupture after hysteroscopic septum resection is a rare complication, and its frequency is reported to be approximately 1–2.7%.7,8 Myometrial damage is believed to be the predisposing factor for uterine rupture (6). Another major factor is the frequency and intensity of uterine contractions during pregnancy after septum resection. A literature review reveals that uterine rupture cases after hysteroscopic septum resection are only documented in case reports.9 Recurrent uterine rupture due to hysteroscopic septum resection in pregnancy, as in our case, is a very rare condition.10 We noted that the first two uterine ruptures resulted from uterine contractions; however, the third rupture occurred spontaneously and earlier in gestation. Another important aspect of our case is that recurrent uterine rupture after hysteroscopic septum resection is very uncommon. To the best our knowledge, this is the first case to evaluate recurrent uterine rupture during pregnancy after hysteroscopic septum resection.
As each uterine rupture occurred earlier than the rupture in the previous gestation, a history of uterine rupture during pregnancy should raise provider suspicion about the possibility of earlier uterine rupture recurrence. This progression suggests that scar tissue formation after surgical repair of each rupture is weaker than that of the previous repair.
As mentioned previously, a uterine septum is the most common Mullerian anomaly 1. Although its frequency in the general population is not precise, it was reported to be as high as 15% in patients with recurrent pregnancy loss.11 Sparac et al. reported that a uterine septum consists of a similar amount of muscle tissue compared to the myometrial wall; however, these myometrial fibers are more prone to irregularity than the myometrial wall itself. Additionally, connective tissue in the septum is more flaccid.12
Complications of hysteroscopic septum resections include uterine perforation, excessive hemorrhage, air embolus, pulmonary edema, excessive glycine absorption, and infection.3,4 Uterine rupture can be evaluated as both an early and late complication. Uterine rupture can be visualized hysteroscopically. A deep incision during hysteroscopy is the most common predisposing factor for early rupture. Simultaneous application of laparoscopy or ultrasonography may prevent this acute complication. Uterine rupture during subsequent pregnancies is a late complication, which can be detrimental for both the mother and fetus. Sentilhes et al. reported that hysteroscopic metroplasty increases the risk of uterine rupture during subsequent pregnancies and that recent uterine perforation and the use of electrosurgery also increases this risk.13 However, these complications cannot be considered as independent risk factors. Electrosurgery increases the risk of thermal myometrial vascular damage and weakening of the tissue, which may cause deep tissue necrosis similar to what is observed during laparoscopic myomectomy. Therefore, the use of rigid scissors should be preferred, and hysteroscopic metroplasty should be stopped when a small (<1 cm) residual septum remains. The current patient had a history of monopolar electrocoagulation for septum resection and laparoscopic suspension of the fundus.
There is no consensus on the safe interval period between hysteroscopic septum resection and a subsequent pregnancy. The interval between operative hysteroscopy and subsequent pregnancies complicated by uterine rupture varies between 1 month and 5 years according the literature.13 In our case, the first rupture occurred 2 years after the initial operation, and subsequent ruptures occurred at 1- to 2-year intervals.
Management of subsequent pregnancies is controversial. Some investigators suggest close follow-up using sonography but have been unable predict uterine rupture.13–15 Prophylactic caesarean section would not have prevented the complications in this case. Therefore, until a reliable predictive method is introduced, patients must be informed about possible symptoms of uterine rupture, and the physician must be aware of fetal and maternal well-being.
4. Conclusion
Although hysteroscopic resection of an intrauterine septum decreases changes of pregnancy loss in women with recurrent abortions (87.5–44.4%), uterine rupture after operative hysteroscopy may have detrimental neonatal and maternal consequences. Uterine perforation and monopolar resection during hysteroscopy are well-known risk factors for subsequent uterine rupture during pregnancy. However, we observed that a history of previous uterine ruptures during pregnancy also increased the risk of ruptures that occur earlier in gestation in subsequent pregnancies. Currently, there is no uniform follow-up protocol that reduces perinatal risks in these patients; thus, patients must be informed about the symptomatology of uterine rupture during pregnancy, and clinicians must be proactive when it comes to detecting the associated clinical signs.
Conflict of interest statement
There is no conflict of interest.
Funding
None.
Ethical approval
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
Acknowledgements
Manuscript was edited by Elsevier Language Editing Service.
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