Case Report

Iatrogenic Uterine Diverticulum in Pregnancy After Robotic-assisted Myomectomy Christopher C. DeStephano, MD, MPH*, Amelia M. Jernigan, MD, and Linda M. Szymanski, MD, PhD From the Department of Gynecology and Obstetrics (Drs. DeStephano and Szymanski), Johns Hopkins University School of Medicine, Baltimore, Maryland, and Division of Gynecology Oncology, Department of OB/GYN (Dr. Jernigan), Cleveland Clinic, Cleveland, Ohio.

ABSTRACT Uterine diverticula are rare outpouchings of the uterus associated with abnormal uterine bleeding, pelvic pain, dysmenorrhea, and adverse obstetric events. At the time of cesarean delivery at 36 5/7 weeks’ gestation during the patient’s first pregnancy and 36 6/7 weeks during the second pregnancy, a fundal iatrogenic uterine diverticulum at the site of a prior robotic-assisted myomectomy was noted. The outpouching communicated with the endometrial cavity and was extremely attenuated, palpably 2 to 3 mm thick. Further research is needed to determine the incidence of iatrogenic uterine diverticulum after robotic myomectomy and whether these malformations increase the risk of adverse obstetric outcomes. Journal of Minimally Invasive Gynecology (2015) 22, 902–905 Ó 2015 AAGL. All rights reserved. Keywords:

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Robotic myomectomy; Uterine diverticulum; Pregnancy; Obstetric outcome

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Uterine diverticulum is a rare finding associated with abnormal uterine bleeding, pelvic pain, dysmenorrhea, and adverse obstetric events [1]. The diverticulum can result from a developmental malformation (true diverticulum) or weakening of the uterine wall from prior uterine surgery (iatrogenic or secondary diverticulum) [2]. Diagnosed at the time of cesarean section during the patient’s first pregnancy and again noted at cesarean section during the patient’s second pregnancy, we present a case of iatrogenic uterine diverticulum likely resulting from a prior robotic-assisted myomectomy. The Johns Hopkins University School of Medicine institutional review board states that case reports do not require review. The patient signed a consent form for publication of identifying material.

The authors declare no conflicts of interest. Corresponding author: Christopher C. DeStephano, MD, MPH, Department of Gynecology and Obstetrics, Phipps 279, 600 North Wolfe Street, Baltimore, MD 21287. E-mail: [email protected] Submitted January 22, 2015. Accepted for publication March 24, 2015. Available at www.sciencedirect.com and www.jmig.org 1553-4650/$ - see front matter Ó 2015 AAGL. All rights reserved. http://dx.doi.org/10.1016/j.jmig.2015.03.016

Case Report A 35-year-old gravida 1 para 0 presented for prenatal care with a history of a robotic fundal myomectomy 4 years before an outside hospital. Her surgeon noted a ‘‘large submucosal myoma bulging from the fundal to posterior region of her uterus’’ and otherwise normal anatomy. He also described cystic areas, consistent with a degenerated myoma, and remarked that it was ‘‘very difficult to appreciate the plane between the myoma and the myometrium.’’ After removing the myoma, a 2-cm defect in the endometrial cavity was noted and was subsequently closed with two 20 polysorb figure-of-eight stitches that incorporated both myometrium and endometrium. This was followed by 2 layers of 0 polysorb figure-of-eight stitches and an additional 2-0 polysorb subserosal baseball stitch. Given this history, she was counseled for delivery at 37 to 38 weeks’ gestation when she initiated obstetric care. On her earliest recorded ultrasound performed at an outside hospital (4 6/7 weeks’ gestation), the questionable presence of myomas with degenerative changes was noted. At our institution, a 19-week level 2 ultrasound noted a ‘‘non-distinct [6.4 ! 5.7 ! 4.1 cm] area of tissue in the

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fundal left anterior portion of the uterus that may represent a degenerating fibroid.’’ At 31 weeks she was sent for a growth ultrasound which noted polyhydramnios with an amniotic fluid index of 25.92 cm and a ‘‘non-distinct area of tissue in the fundal left portion of the uterus . [that] does not look like a fibroid today but rather thickened myometrium. In some views the uterine fundus appears to have a septum. This area may represent fibrous scar tissue from the patient’s reported myomectomy.’’ Shortly thereafter, she was admitted to an outside hospital for preterm labor and completed a course of betamethasone at 32 weeks’ and 4 days’ gestation. After discharge, follow-up sonograms demonstrated that her amniotic fluid was as high as 32.6 at 35 weeks and her contractions continued. Because of continued contractions and a prior myomectomy, she was counseled on the risk of uterine rupture with labor after myomectomy versus the risks of prematurity. She consented to proceed with a late preterm delivery and was scheduled for a cesarean delivery This pregnancy was otherwise notable for advanced maternal age, a resolved first trimester subchorionic hemorrhage, gestational thrombocytopenia, mild asthma, depression with sertraline use, and palpitations with a normal cardiac evaluation. She presented as scheduled at 36 weeks’ and 5 days’ gestation and underwent a primary low segment transverse cesarean section, delivering a live, female newborn from the frank breech presentation weighing 2570 g with Apgar scores of 1, 6, and 7 at 1, 5, and 10 minutes, respectively. After the uterus was exteriorized, a 5-cm diameter right posterior fundal outpouching was noted (Fig. 1). On internal exam of the uterus, this outpouching communicated with the endoFig. 1 A 5-cm diameter posterior fundal outpouching (white arrow) with adhesions to sigmoid colon noted during cesarean section for first pregnancy.

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metrial cavity and was extremely attenuated, palpably 2 to 3 mm thick in some areas. Dense adhesions between this outpouching and the sigmoid colon were noted. On examination of the placenta, a 4-cm outpouching was noted that filled this defect (Fig. 2). Because this was an unexpected finding, we did not have an opportunity to adequately consent the patient for uterine reconstruction. We decided against repairing the defect because it was hemostatic but had significant vasculature at the base of the outpouching that had the potential to bleed heavily and would likely contract postpartum, facilitating repair at a later time. The remainder of the cesarean section and her hospital stay were uncomplicated. After counseling regarding the finding and the recommendation for contraception and further evaluation and surgical closure of the diverticulum postpartum before subsequent conception attempts, she was discharged 4 days postoperatively. The patient presented 9 months later for prenatal care at 6 5/7 weeks pregnant. She was counseled on the risk of uterine rupture and was treated as if she had a history of classic cesarean section and the recommendation for delivery between 36 and 37 6/7 weeks’ gestation. Anatomy sonogram at 20 5/7 weeks showed no evidence of anomalies with no comments on the uterine diverticulum. An ultrasound at 31 5/7 weeks visualized the uterine diverticulum and showed an interface between the myometrium and the placenta. The patient received a complete course of betamethasone at 33 3/7 weeks and was scheduled for cesarean section at 36 6/7 weeks. A repeat low segment transverse cesarean section was performed, and the hysterotomy was closed in 2 layers. She delivered a male newborn weighing 2840 g with Apgar scores of 9 at 1 minute and 9 at 5 minutes. A larger, fundal, thin-walled uterine diverticulum was noted measuring 10.5 ! 9 ! 5.5 cm (Fig. 3). Because the area was hemostatic, the size of the diverticulum would make repair difficult, Fig. 2 A 4-cm placental outpouching (black arrow) was noted that filled the iatrogenic uterine diverticulum noted during cesarean section for first pregnancy.

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Fig. 3 Fundal, thin-walled iatrogenic uterine diverticulum measuring 10.5 ! 9 ! 5.5 cm noted during cesarean section for second pregnancy.

and the defect had the potential to bleed heavily at the time of cesarean, the decision was made to not excise the defect. The patient was counseled before the procedure that closure would not be attempted unless the diverticulum was immediately life-threatening. The patient previously expressed no interest in future fertility and planned to follow-up postpartum. The patient was consented preoperatively for a levonorgestrel-releasing intrauterine system; however, it was not placed because of the risk of migration into the large thin-walled diverticulum. The newborn was initially transferred to the neonatal intensive care unit for transitioning and was transferred to the newborn nursery on day of life 2 and discharged on day of life 3. The patient was discharged home on postoperative day 3 in good condition and planned for follow-up with the family planning clinic to discuss options for long-acting contraception. Comment True and secondary uterine diverticula are rarely reported in the literature. A PubMed search of ‘‘uterus and diverticulum’’ resulted in 11 cases of true uterine diverticulum and 1 case of uterine diverticulum after a myomectomy (unspecified surgical approach). Two cases described pregnancy outcomes in primigravida women diagnosed with uterine diverticula. One case described preterm rupture of mem-

Journal of Minimally Invasive Gynecology, Vol 22, No 5, July/August 2015

branes at 31 weeks delivered by cesarean section, whereas the other case reported uterine rupture at the site of the uterine diverticulum at 17 weeks’ gestation [2,3]. A PubMed search of ‘‘cesarean and diverticulum’’ resulted in 9 cases of cesarean scar diverticulum, but subsequent pregnancy outcomes were not described. A PubMed search of ‘‘(partial or incomplete) and (uterine) and (dehiscence or rupture)’’ resulted in 219 articles; however, only 4 articles were found when ‘‘myomectomy’’ was added to the search. Our case appears to be 1 of the only reported case reports of iatrogenic uterine diverticulum during subsequent pregnancies after a prior robotic-assisted myomectomy. This case was considered an iatrogenic uterine diverticulum or partial uterine dehiscence because of a narrow neck and persistent outpouching after contraction of the uterus. Uterine dehiscence would be expected to have a wider neck and thinner layer of tissue. However, the narrow neck can be explained by the placenta getting trapped in the defect and growing over the ensuing months. Uterine sacculation, on the other hand, is typically a larger outpouching that contracts after delivery. It occurs during pregnancy as the uterus is distended by the destruction of the uterine wall by trophoblastic tissue [2]. Because of the risk of potentially catastrophic dehiscence, repair of these defects is suggested. In this case, the decision was made not to excise the defect at the time of cesarean section because of lack of consent, risk of hemorrhage, and potential inadequate closure due to thick, vascular base of the defect arising from the fundal active segment. Definitive pathologic diagnosis therefore could not be obtained. The patient was instructed on the finding and its association with infertility and abnormal uterine bleeding. We recommended that the patient follow-up for further imaging and postpartum closure before subsequent conception attempts. It was expected that the defect and uterus would contract down postpartum, facilitating closure. However, the decision to close these defects should be individualized because successful closure of a thin anterior uterine wall with incomplete uterine rupture during the time of cesarean section was described by Matsubara et al [4]. On the other hand, in a case similar to our’s, Rajiah et al [2] did not attempt to excise a uterine diverticulum because of the surgical risks at the time of cesarean delivery and their expectation that the size of the diverticulum would be reduced postpartum. The possibility of a uterine diverticulum should be taken into account during the counseling of pregnant patients with a history of prior robotic myomectomy, especially in cases where the endometrial cavity was entered. Although recent evidence supports later cesarean delivery (38–39 weeks) for patients with a history of prior myomectomy, further research is needed regarding the optimal timing of delivery depending on the surgical approach to myomectomy and whether the endometrial cavity was entered. In a National Institute of Child Health and Human Development/Maternal-Fetal Medicine Units Network study, the frequency of uterine rupture in the prior myomectomy group was 0% (n 5 176) compared

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with .88% (n 5 455) for the classic cesarean delivery group and .41% (n 5 13 273) for the low segment transverse cesarean delivery group [5]. Given these findings, the authors advised against delivery before 37 weeks. However, the study was limited by the lack of information about the extent of the prior myomectomy or the type of myomectomy. In a cohort of 32 patients who underwent hysteroscopic or open abdominal myomectomy, 6 patients (18%) were found to have uterine diverticula on hysteronsalpingography [6]. It is unclear whether the incidence of uterine diverticula is similar after robotic or laparoscopic procedures. In the largest retrospective cohort of pregnancy outcomes after robotic myomectomy, 92 deliveries were documented subsequent to prior robotic myomectomy. Entry into the endometrial cavity occurred in 20.6% of cases, cesarean deliveries were performed in 95.7% of patients, and uterine rupture was documented in 1 case (1.1%). The uterine rupture occurred at 33 weeks’ gestation during precipitous labor [7]. This is lower than the 4% to 9% risk of uterine rupture quoted for patients after classic cesarean section who labor [8]. However, the mean gestational age of delivery in the study was 36 weeks 6 days, suggesting practitioners delivered patients earlier without labor similar to the management of women with prior classic cesarean section. In conclusion, we report a rare case of uterine diverticulum during subsequent pregnancies after a prior robotic

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myomectomy. Further research is needed to determine the incidence of uterine diverticulum after robotic myomectomy and whether these malformations increase the risk of adverse obstetric outcomes. Establishing a national robotic surgery database with subsequent obstetric outcomes is recommended to facilitate research on this topic. References 1. Schmidt AP, Glitz CL, Passos EP, Arbo E, Cunha-Filho JS. Hysteroscopy and three-dimensional ultrasonography in uterine diverticulum diagnosis: a case report. Am J Obstet Gynecol. 2004;190:561–562. 2. Rajiah P, Eastwood KL, Gunn ML, Dighe M. Uterine diverticulum. Obstet Gynecol. 2009;113(2 Pt 2):525–527. 3. Hassim AM. Rupture of a diverticulum of the uterus. Am J Obstet Gynecol. 1968;101:1132–1134. 4. Matsubara S, Shimada K, Kuwata T, Usui R, Suzuki M. Thin anterior uterine wall with incomplete uterine rupture in a primigravida detected by palpation and ultrasound: a case report. J Med Case Rep. 2011;5:14. 5. Gyamfi-Bannerman C, Gilbert S, Landon MB, et al. Risk of uterine rupture and placenta accreta with prior uterine surgery outside of the lower segment. Obstet Gynecol. 2012;120:1332–1337. 6. Lev-Toaff AS, Karasick S, Toaff ME. Hysterosalpingography before and after myomectomy: clinical value and imaging findings. AJR Am J Roentgenol. 1993;160:803–807. 7. Pitter MC, Gargiulo AR, Bonaventura LM, Lehman JS, Srouji SS. Pregnancy outcomes following robot-assisted myomectomy. Hum Reprod. 2013;28:99–108. 8. Cunningham G, Leveno K, Bloom S, Hauth J, Rouse D, Spong C. Williams Obstetrics. 23rd ed. New York: McGraw-Hill; 2010.

Iatrogenic Uterine Diverticulum in Pregnancy After Robotic-assisted Myomectomy.

Uterine diverticula are rare outpouchings of the uterus associated with abnormal uterine bleeding, pelvic pain, dysmenorrhea, and adverse obstetric ev...
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