Myomectomy scar pregnancy ‒ a serious, but scarcely reported entity: literature review and an instructive case

Marcela Toro-Bejarano; Robert Mora; Ilan E. Timor-Tritsch; Jessica Vernon; Ana Monteagudo; Francesco D’Antonio; Karen Duncan

doi:10.1515/crpm-2021-0071

Article Publicly Available

Myomectomy scar pregnancy ‒ a serious, but scarcely reported entity: literature review and an instructive case

Marcela Toro-Bejarano , Robert Mora , Ilan E. Timor-Tritsch , Jessica Vernon , Ana Monteagudo , Francesco D’Antonio and Karen Duncan

Published/Copyright: January 3, 2022

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From the journal Case Reports in Perinatal Medicine Volume 10 Issue 1

Abstract

Objectives

Uterine myomas are a frequent finding in reproductive age women with an estimated incidence 12–25%. 1. Treatment of uterine myomas to facilitate good pregnancy rates and outcome, such as hysteroscopic, laparoscopic, abdominal resection uterine artery embolization among others were evaluated in terms of pregnancy outcome. While the literature is replete of the pregnancy complication of uterine rupture after myomectomies, 2–4 there are very few publications evaluate a relatively rare pregnancy complication associated with placental implantation within the uterine cavity at the site of the previous myomectomy, namely the myomectomy scar pregnancy (MSP). Despite their relative rarity, this type of pathologically adherent placenta rightfully belongs to the well-known entity of placenta accreta spectrum (PAS).

Case presentation

We present a complicated case of MSP and review the available literature to raise attention to its clinical appearance, its prenatal diagnosis so appropriate intrapartum management can be planned.

Conclusions

Despite the rarity of MSP, continuous attention should be given at every single routinely scheduled or indication driven obstetrical US scan following myomectomies to evaluate the placental site implantation regardless of the route and technique of their initial surgical procedure.

Keywords: myomectomy; scar pregnancy; uterine rupture

Introduction

Uterine myomas are a frequent finding in reproductive age women with an estimated incidence 12–25% [1]. Treatments of uterine myomas to facilitate good pregnancy rates and outcome, such as hysteroscopic, laparoscopic, abdominal resection uterine artery embolization among others were evaluated in terms of pregnancy outcome. The pertinent literature is replete of the pregnancy complication of uterine rupture after surgeries to remove myomas [2], [3], [4].

However very few publications evaluate a relatively rare pregnancy complication associated with or reported to be seen after placental implantation within the uterine cavity at the site of the previous myomectomy, namely the myomectomy scar pregnancy. They attracted attention in pregnancies with no history of previous cesarean deliveries and without placenta previa, however all had histories of conservative surgery to remove uterine fibroids.

Despite their relative rarity, and due to their unpleasant surprise at the diagnosis as well as the associated complications at different stages of the pregnancy and delivery, this type of pathologically adherent placenta rightfully belongs to the well-known causes of placenta accreta spectrum (PAS).

We present a complicated case of myomectomy scar pregnancy and review the available literature to raise attention to its clinical appearance and emphasize that if kept in mind, it’s prenatal diagnosis can be established and appropriate intrapartum management can be planned.

We encountered a posterior uterine wall placenta accreta during the cesarean delivery at 36 weeks of a patient with prior myomectomies. While treating the resulting complications we reviewed the pertinent literature. By presenting the case, as well as the available literature on the subject of myomectomy scar pregnancy we considered that this may raise awareness of those who perform myomectomies and the obstetricians, who manage these patients during pregnancy and delivery.

Case presentation

The patient is a 44 year old G1P0 with history of a myomatous uterus, first diagnosed in 2007, when she presented with menorrhagia, dysmenorrhea, bladder pressure and constipation. Patient failed medical management and underwent robotic assisted, posterior wall myomectomy in 2017 followed by three hysteroscopies for resection of one submucous myoma in 2019 and an endometrial polyp in 2020. Following these procedures, she became pregnant in July 20 after in vitro fertilization (IVF). Her obstetrical course was unremarkable and, following the American College of Obstetrics and Gynecology guidelines. A series of ultrasound (US) images are presented (Figure 1), however never during her US scans was a morbidly adherent posterior placenta suspected. Even in retrospect it is hard to detect the pathology since no targeted attention using color Doppler was directed to the presumed site of the myomectomy scar. The patient was scheduled for primary cesarean delivery at 37 weeks 4 days for her history of prior myomectomies. The infant was delivered via low, transverse uterine incision without difficulty. Upon delivery of the placenta, it was noted that the placenta was adherent to the posterior wall of the uterus at one of the presumed myomectomy sites. The uterus was exteriorized for better visualization and manual extraction was attempted. The placental bed was noted to be bleeding profusely, and placenta accreta was diagnosed. Tranexamic acid, hemabate, methergine, and misoprostol were given to improve uterine tone and hemostasis. The area of the posterior uterus with adherent fragment of placenta was oversewen with multiple stitches with 0-vicryl until hemostasis was achieved. The hysterotomy was then repaired using 0-vicryl in a running locked fashion, in a single layer. A second, imbricating layer of the same suture was used to obtain excellent hemostasis. The fascia and skin were then closed in the usual fashion. Total estimated blood loss was 3740 mL and patient received 3 units of packed red blood cells, 1 unit of cryoprecipitate and 1 unit of fresh frozen plasma in the operating room.

Figure 1:

Myomectomy scar pregnancy. Prenatal ultrasound images. Retrospective review.

(A) Gestational sac in two views; No suspicion for any implantation pathology. (B) First sighting of the normal appearing placenta upon the presumed site of myomectomy (oval). (C) First hint of maybe vascularization below the placenta without lacunae. (D) The normal appearing placenta clearly implanted upon the presumed site of the myomectomy (oval).

In the immediate post-operative period, the patient developed a fever to 102.9 °F, and was diagnosed with suspected endometritis. Antibiotic treatment with ampicillin, gentamycin and clindamycin was initiated and continued until patient had been afebrile for more than 24 h. On postpartum day 1, the patient had the expected scant, normal vaginal bleeding remaining hemodynamically stable, but noted to have persistent low-grade fevers and tachycardia. Chest and abdominal X-ray and CT PE were performed which were negative for pulmonary artery embolism and pelvic abscess. After blood transfusion, the hemoglobin level was 8.8 gm/dL decreased from the preoperative level of 10.7 gm/dL. The laboratory results were within normal limits throughout her hospital stay. A transvaginal ultrasound (TVUS) scan on post-partum day 3 revealed echogenic tissue at the fundal aspect measuring 4.0 × 3.6 × 3.3 cm. Color Doppler revealed increased flow within this area arising from the posterior fundal aspect of the cavity. The rest of patient’s post-partum course was unremarkable and she was then discharged home with precautions and close follow-up. Repeat TVUS 18 days after her surgery which revealed an area of echogenic tissue at the posterior aspect of the cavity measuring 5.6 × 1.7 × 4.2 cm (Figure 2A) with two foci of low velocity venous flow with PSV of 8 cm/s. Despite the low vascularity noted on ultrasonography, to prevent bleeding at the planned removal of the placental remnant the decision was made to proceed with bilateral uterine artery embolization followed by hysteroscopy with dilation and curettage for removal of the remaining retained fragments of the placenta. Both procedures were performed without complications.

Figure 2:

Myomectomy scar pregnancy. Postnatal ultrasound images. Prospective studies.

(A) Postpartum day 18, a 3.0 × 2.9 cm placental fragment attached to the posterior wall of the uterine cavity. The small shadowing, hyperechoic, submillimeter structures are generated by the suture material. (B) Image of the uterine artery embolization on postpartum day 24. On the same day after the uterine artery embolization a D&C was also performed. (C) Six days after embolization (postpartum day 40) part of the retained placenta was still present. (D) and (E). On days 30 and 41 after embolization (postpartum days 54 and 65) the retained placental fragment is still present, however no pathological vascularity is seen. (E) Finally, 75 days after the embolization (postpartum day 100!) the uterus appears normal again.

The post UAE and D&C ultrasound images (Figure 2B–E) revealed an extremely slow resolution of the still clearly seen residual fragments of the slow placental involution. Finally, on a rescan 3 and a half months after the delivery a normal uterus was seen (Figure 2F).

The literature

The literature is replete with reports evaluating the outcome of pregnancies following the three most used surgical techniques of myomectomies. Their main focus is to review uterine rupture in pregnancy as their most consequential complication. There is scant data about complications resulting from pregnancies implanted in the site or on the scar of a myomectomy and their outcome. There is even confusion about the term to be used in cases of placental implantation onto the site of a surgically removed fibroid creating a scar facing the uterine cavity. In the literature it is referred to as “intramural pregnancy”, “subserosal pregnancy” and “myomectomy scar pregnancy”. The latter seems to be the most used term of this rare, nevertheless complication ridden, faulty implantation of a pregnancy in or on a scar created by a previous myomectomy.

Our case of the myomectomy scar pregnancy following a series of myomectomies, was detected, immediately after the delivery of the baby, it created an instant havoc in the operating room requiring urgent containment of the bleeding placental site. it is striking that despite the many US scans there was no conscious scrutiny directed to the site of the previous myomectomy which was below the posterior placenta (Figure 2B). The evolving case and its long and convoluted process of managing the case speaks for itself. The judicious use of the uterus saving uterine artery embolization was indicated and allowed the safe removal of the retained and still adhered placental remnant.

The present case prompted us to do a detailed literature search to find similar documented cases and be able to cite the literature to raise the awareness our department (and maybe of others at lage) to the existence, the possible predelivery diagnosis and management of this relatively rare but far-reaching entity. The result of our review is summarized in Table 1. However below we detail some of the salient and relevant observations of the authors publishing on this entity.

Table 1:

Literature review of published article about myomectomy scar pregnancies.

#	Author/year	GA, weeks	Location of myomectomy scar and pregnancy implantation	Myomectomy performed	Single or multiple myomas removed	Management of myomectomy scar pregnancy	Histology
1	Seinera P et al. [5] 2000	N/A	N/A	Laparoscopic	1	At CD “slightly adherent placenta removed”. No surgical treatment reported.	N/A
2	Warshak CR et al. [7] 2006	36	Posterior	N/A	N/A	Cesarean delivery hysterectomy	Accreta
3	Park WI et al. [6] 2006	4	Posterior	Abdominal	1	Laparoscopic removal	N/A-
4	Al-Serehi A et al. [8] (2008)	Two cases:	Both adherent placenta to posterior-fundal aspect. No placenta previa.	Unknown x2	Unknown X2	a. Emergent CD @ 34 weeks due to maternal hemorrhage, subtotal hysterectomy.	a. Placenta accrete.
	Al-Serehi A et al. [8] (2008)	a. 12 w 4 d					a. Placenta accrete.
	Two cases	b. 18 w				b. Vaginal delivery @ 39 weeks.	b. Placenta delivered with the myoma. Histology: placenta accreta
5	Wong KS et al. [9] 2010	N/A	Posterior	Abdominal	Multiple	US guided intra-gestational and systemic MTX injection	N/A
6	Tagore S et al. [18] 172010	5	Fundal serosa	Laparoscopic	Unknown	US-guided intra-gestational injection of 50 mg MTX	N/A
	Li M et al. [16] 2011	1st trimester	Cornual area	Hysteroscopic	1	Hysteroscopy & laparoscopy	Smooth muscle with hemorrhage, necrotic tissue & villous tissue& few trophoblasts
	Li M et al. [16] 2011	1st trimester	Misdiagnosed as “cornual” pregnancy	Hysteroscopic	1	Hysteroscopy & laparoscopy
7	Mathiesen E et al. [14] 2013	26	Posterior	Hysteroscopic	1	Cesarean hysterectomy at 34 weeks	Inctreta
8	Banon K et al. [10] 2013	10	Posterior	Abdominal	1	Systemic MTX; Suction curettage and laparoscopic removal	N/A
9	Lo T-K et al. [17] 2015	38	Anterior	Laparoscopic	Multiple (12)	Scheduled CD with diagnosis of placenta accreta. Bilateral UAE and post-partum follow-up.	N/A
10	Tanaka M et al. [11] 2016	26	Posterior	Hysteroscopic	1	C/D at 39 weeks and hysterectomy for profuse bleeding	Accreta
11	Ishiguro T et al. [12] 2018	8	Multiple sites in two separate surgeries	Both laparoscopic	Multiple myomas during two separate surgeries	Emergent laparoscopic removal under transvaginal US guidance	N/A
13	Vagg D et al. [13] 2018	12	1 right/fundal-posterior	Abdominally	1	Hysterectomy	N/A
14	Zhu L et al. [25] 2020	6	Left/posterior fundus	Abdominal	1	Combined hysteroscopic and laparoscopic excision	N/A
15	Liu D et al. [15] 2019	Unknown	Right cornual area	Unknown	1	Excision	N/A
16	Dutta I et al. [19] 2020	Unknown	Anterior	Unknown	1	Laparotomy, removal of scar pregnancy	N/A
17	Mohr-Sasson A et al. [3] 2020	Three cases GA N/A	N/A	Three cases: one case each in the abdominal laparotomy, laparoscopic & hysteroscopic group	N/A	All three delivered.	N/A
17	Mohr-Sasson A et al. [3] 2020	Three cases GA N/A	N/A		N/A	In all three cases of PAS cases manual lysis was sufficient to remove the placenta and no postpartum hemorrhage control was needed	N/A
18	Saleh MM et al. [26] 2021	21w 4d	Posterior	N/A	1	Emergency CD at 28 weeks 5 days.	Placental tissue attached to resected intestinal was as indirect proof of placenta percreta
18	Saleh MM et al. [26] 2021	21w 4d	Posterior	N/A	1	Small bowel at placental invasion site resected. With side-to-side reanastomosis. Uterine back wall repaired saving hysterectomy.
19	Our case 2021	34	Posterior	Laparoscopic	Multiple, twice, 2 years apart	CD. Focal posterior percreta. Bleeding. Sutured posterior uterine wall to stop bleeding. 1-week later bleeding. UAE & same day D&C RPOCs removal	Accreta

CD, cesarean delivery; UAE, uterine artery embolization; RPOC, retained products of conception; N/A, not available; D&C, dilatation and Curettage; MTX, methotrexate.

Seinera et al. [5] evaluated 65 patients in 54 patients who became pregnant after laparoscopic myomectomy reporting only one case of adherent placenta at the surgery site.

Park et al. [6] claim the first documented publication of an “in-vitro” “sub serosal pregnancy” in a 35-year-old woman with a history of myomectomy diagnosed by transvaginal US revealing a gestational sac within the sub serosal area of the posterior uterine wall. A successful treatment was achieved with conservative surgery, and the pathology of the excised mass demonstrated chorionic villi involving myometrium. Early in a subsequent pregnancy, placental invasion through the sinus tract was detected which suggested, that the probable pathogenesis of this rare variant of intramural pregnancy is implantation through a sinus tract made during a previous uterine surgery.

In an article directed towards evaluating the accuracy of ultrasound and MRI in the diagnosis of placenta accrete Warshak et al. [7] found only one case of myomectomy related placenta accrete.

Myomectomy has been generally considered as a risk factor for placenta accreta [8] however Gyamfi-Bannerman et al. [3] analyzed subsequent pregnancies of 176 women who had undergone myomectomy and found no cases of placenta accreta; therefore, they suggested that myomectomy is associated with a low risk for placenta accreta.

In 2010, Wong et al. [9] reported on a patient who had open myomectomy and presented with a 1st trimester pregnancy implanted on the site of the myomectomy. She was treated with systemic MTX and serial ultrasound exams revealed a slowly resolving gestational sac over 15 weeks. Understandably, there was no histology proof in this case.

Banon et al. [10] called the MSP “intramural pregnancy” publishing an asymptomatic 6 week (probably) missed abortion diagnosed by routine US with a history of an open myomectomy for a large, left-sided, posterior leiomyoma. There was no information if the uterine cavity was entered. Suction curettage was performed approximately 1 month after the initial diagnosis. Pathology revealed decidua with foci of necrosis and portions of gestational endometrium, but no placental villi were identified. Ultimately, the diagnosis of intramural pregnancy was made via US and CT. After systemic MTX treatment the intramural pregnancy failed to resolve completely, and it was removed using robotic laparoscopic resection. Her only predisposing factor to this rare type of pregnancy was the history of myomectomy.

Because of the small number of reported cases, the influence of hysteroscopic myomectomy on placenta accreta is largely unknown. Tanaka et al. [11] propose that any patient with previous hysteroscopic myomectomy should be considered to be at high risk for placenta accreta, even if she does not develop placenta previa.

Ishiguro et al. [12] described one of the most interesting MSP cases of an MCP following a cryopreserved embryo transfer. After laparoscopic assisted myomectomy with removal of as many as 15 fibroids after 2 years of infertility the patient has undergone a second laparotomy assisted myomectomy of 19 fibroids. Then a pregnancy was achieved by in-vitro fertilization in which the gestation seemed to be implanted on one or maybe in one of the many scars left behind the myomectomies. One of their theories was that the embryo transfer procedure that used a hard-tipped ET catheter penetrated the myometrium causing an intramural or subserosal pregnancy. This group also suggested that if an intramural or subserosal pregnancy is suspected before surgery, a myometrial repair should be considered after removing the products of contraception in order to prevent a repeat intramural pregnancy.

Vagg et al. [13] reported on a “intramural ectopic pregnancy” at 12 weeks gestation in a woman 1 year post open myomectomy. Both transvaginal US and MRI were utilized as diagnostic aids. Due to the size and location of the gestational sac, hysterectomy was deemed to be the safest treatment modality in their case. A MSP misdiagnosed as a cornual pregnancy was presented by Liu et al. [14] to exemplify the possibility to misdiagnose cases of pregnancy implanted at previous myomectomy sites. Several other authors published single cases of MCP after myomectomy [14], [15], [16], [17], [18], [19] and are included in Table 1.

Larger series were studied by Mohr-Sasson et al. [20] and evaluating the occurrence of PAS in post-myomectomy pregnancies of 241 women based upon literature search. They found only one single case of PAS in each of the laparoscopic, laparotomic and hysteroscopic surgery groups. Placenta previa was low and comparable between groups. The author’s conclusion was that: (1) Prevalence of PAS during pregnancy post myomectomy performed by any kind of approach is low; (2) Surgical approach was non-discriminatory; (3) Manual lysis of the placenta was lower following hysteroscopic myomectomy, as compared to laparotomic or laparoscopic myomectomy; (4) There was no need for additional postpartum hemorrhage control in any of their study groups. Interesting is at this center that following myomectomy done by laparoscopy or by laparotomy, it is contra-indicated to deliver vaginally, whereas after hysteroscopic myomectomy vaginal delivery is recommended when possible.

Gyamfy-Bannerman et al. [3] studied 47,112 women in their Cesarean Registry identifying 176 women who had myomectomies. When they analyzed their obstetrical performance, no cases of PAS were recorded.

Fukuda et al. [21] reported no significant differences in the perinatal outcomes between 105 females who delivered after myomectomy. Forty-eight had laparoscopic and 57 had abdominal myomectomy. Moreover, both groups had a high rate of successful transvaginal delivery after selecting the appropriate candidates.

Kasuga et al. [22] reported no cases of PAS in their 34 cases of pregnancies after hysteroscopic myomectomies.

Similarly, Lebowitz et al. [23] studied a cohort of 127 patients to predict outcomes following myomectomy for intramural fibroids of whom 74 achieved pregnancy and delivered without any cases of pathological.

An even larger number of pregnant patients, who delivered after laparoscopic myomectomies, were reported by Koo et al. [24]. In their 523 deliveries after myomectomies there was no mention about cases of PAS.

The first review of the literature was published by Zhu et al. in 2020 [25] adding their own case of a 28-year-old pregnant woman with vaginal bleeding diagnosed with MSP by US and MRI and then underwent excision via laparotomiy.

Saleh et al. [26] published probably the most bizarre case of a placenta invading the site of a myomectomy scar and infiltrating the adjacent small bowel. In this case the authors suggest, that this may have been a placenta percreta grade 3 by FIGO due to the invasion of the small intestine. However as the authors subsequently concede: the small amount of myometrium resected alongside the placenta was not sufficient to diagnose placenta percreta. Although highly indicative, PAS cannot be ruled in or ruled out, since no hysterectomy specimen was available.

Regardless of its actual pathogenesis, we consider this case as further supporting the dangerous and consequential nature of placental implantation on myomectomy scars.

An important issue is to understand the available diagnostic tools to enable the best detection rate for this pathology. It should be clear at the outset that both MSP and CSP are precursors of PAS.

In early 1st trimester of the pregnancy transvaginal probes provide clear picture imaging the anterior, fundal or posterior aspects of the uterus. The focal depth has to be adjusted to the level of a posterior placenta. Detecting lacunar spaces in the placenta could be helpful since it is a useful marker of PAS. The use of color and power Doppler is also beneficial. This should be considered as a “must”, since lowering the pulse repetition frequency may reveal a diagnostic clue in the form of increased vascularity. The introduction of microvascular imaging (MVI) can aid in the detection of sub-placental vascular spaces between the myometrium and the placenta (similar to cases of cesarean scar pregnancies) [27, 28]. MVI can detect deeper and smaller vessels as opposed to regular color Doppler.

Similar suggestions are now considered for patients conceiving after previous cesarean deliveries for early detection of cesarean scar pregnancies as the precursor of PAS [29].

Corresponding author: Ilan E. Timor-Tritsch, MD, Department of Obstetrics and Gynecology, New York University School of Medicine, New York, NY, USA, Phone: +1 917 520 7177, E-mail: Ilan.timor@nyulangone.org

Research funding: None declared.
Author contributions: All authors have accepted responsibility for the entire content of this manuscript and approved its submission.
Competing interests: Authors state no conflict of interest.
Informed consent: Informed consent was obtained from all individuals included in this study.

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Received: 2021-09-15

Accepted: 2021-12-02

Published Online: 2022-01-03

Articles in the same Issue

https://doi.org/10.1515/crpm-2021-0071

Keywords for this article

myomectomy; scar pregnancy; uterine rupture