Disseminated Peritoneal Leiomyomatosis of the Peritoneum Following Laparoscopic Supracervical Hysterectomy with Morcellation: A Case Report

Introduction

According to World Health Organization (WHO) data, hysterectomy is one of the most commonly performed surgical procedures in women [1]. Hysterectomy can be carried out using various approaches, including the vaginal route [either traditional or via vaginal Natural Orifice Transluminal Endoscopic Surgery (vNOTES)], laparoscopy, or open laparotomy. In laparoscopic surgery, one option for women with non-oncological indications is laparoscopic supracervical hysterectomy [1]. The main advantages of this technique include shorter operative time and reduced complexity, particularly in patients with a history of cesarean section [2]. However, disadvantages include future oncologic risks associated with the retained cervical stump and the possibility of abnormal bleeding from the cervical remnant [3].

It is also widely believed that subtotal hysterectomy lowers the risk of pelvic floor dysfunction and vaginal prolapse [4]. This belief is related to the preservation of the sacrospinous ligaments, which remain intact during subtotal hysterectomy. However, objective studies have not confirmed significant benefits of this technique in reducing the risk of pelvic organ prolapse or urinary incontinence [5].

An advantage of retaining the cervix is its potential role as an anchoring point for procedures such as sacrocolpopexy or laparoscopic lateral suspension [6]. A challenge associated with subtotal hysterectomy, however, is the extraction of the removed uterine body through a 10-mm trocar. In such cases, morcellation is used to reduce the volume of tissue for removal. This technique carries the risk of dispersing viable uterine cells or tissue fragments throughout the abdominal cavity, potentially leading to complications such as disseminated peritoneal leiomyomatosis (DPL) [7].

The dissemination of myometrial cells is one of the leading theories explaining the pathogenesis of DPL. An alternative theory, proposed by Morgan et al in 2022, suggests that DPL may originate from the metaplasia of stem cells [8]. The present report describes the case of a 40-year-old woman presenting with abdominal pain due to DPL, 5 years after undergoing a supracervical hysterectomy with morcellation.

Case Report

A 40-year-old woman was referred to a gynecologist by her general practitioner due to intermittent lower abdominal pain without additional symptoms, and a suspected solid ovarian mass was detected during an abdominal ultrasound. Her medical history was unremarkable except for a laparoscopic supracervical hysterectomy with salpingectomy performed 5 years earlier due to abnormal uterine bleeding. During the initial laparoscopic procedure, mechanical morcellation was carried out without the use of a containment system. Postoperative histopathological analysis confirmed the presence of uterine leiomyomas (2 mitotic figures per 10 high-power fields).

During the consultation, gynecological examination with a speculum revealed a cervical stump with no suspicious oncological findings. No palpable adnexal abnormalities were detected. Transvaginal ultrasound showed a solid pelvic mass with irregular structure and low vascularity (color Doppler score=1). The echogenicity of the lesion was similar to that of the cervical stump (Figure 1). With the bladder filled, the mass appeared to be in contact with the bladder dome. When the bladder was emptied, the lesion was located approximately 1.5 cm from the bladder mucosa (Figure 2). The uterine body was not visualized, and the cervical stump appeared normal. Both ovaries were visualized separately from the lesion. Laboratory results showed CA-125=23 U/mL and HE4=50.5 pmol/L. Further imaging diagnostics were deemed necessary.

Pelvic and abdominal magnetic resonance imaging (MRI) revealed a polycyclic mass of intermediate signal intensity on T2-weighted images, located above and slightly to the left of the bladder, measuring 45×36×30 mm (right-left×anterior-posterior×head-foot). Two additional foci with similar signal characteristics were identified: one adherent to the sigmoid colon (approximately 23×19 mm) and a smaller lesion (approximately 7 mm in diameter) located near the superior right side of the bladder wall. On Liver Acquisition with Volume Acceleration (LAVA) sequences, all lesions showed contrast enhancement comparable to that of the cervical stump (Figures 3–6).

Based on the imaging findings, residual uterine fragments from prior morcellation (ie, DPL) were suspected. The patient was scheduled for laparoscopic excision of the lesions.

During laparoscopy, after establishing pneumoperitoneum, a 5-cm mass was identified on the anterior abdominal wall in the midline above the bladder dome, covered by parietal peritoneum. Morphologically, the lesion resembled a leiomyoma. It was adherent to omental appendages and the sigmoid colon. Additionally, a 2-cm lesion was found on the surface of the sigmoid colon adventitia (Figure 7). Adhesions involving the anterior abdominal wall lesion were dissected, and the mass was excised with a margin of subperitoneal adipose tissue. The pouch of Douglas and the vesicouterine pouch were inspected.

A third lesion, approximately 1 cm in diameter and resembling a leiomyoma, was identified subperitoneally near the bladder dome (Figure 8). The mass was excised in its entirety without breaching the bladder wall. A methylene blue test confirmed bladder wall integrity. The lesion on the sigmoid colon surface was excised using an EndoGIA 60-mm stapler (Figure 9). No additional pathological changes were found on inspection of the abdominal cavity.

All excised specimens were retrieved from the abdominal cavity using a laparoscopic endobag. The patient had an uneventful recovery and was discharged on postoperative day 3 in good general condition. Histopathological analysis confirmed leiomyomas of the abdominal soft tissue. Immunohistochemistry demonstrated positivity for estrogen receptor, progesterone receptor, desmin, and smooth muscle actin (SMA). CD117 and CD34 were negative. The Ki-67 index was 1%, and the mitotic index was 0 per 10 high-power fields (Figure 10).

Discussion

This case illustrates a rare but significant complication that should be considered in the differential diagnosis of multiple peritoneal masses, particularly in patients with a history of surgery for uterine fibroids.

In laparoscopic procedures such as myomectomy, total laparoscopic hysterectomy, or supracervical hysterectomy – especially when the uterus is too large to remove vaginally – morcellation is often employed to fragment and extract tissue. However, morcellation carries several inherent risks. The literature describes cases of organ injury caused by morcellator blades, most commonly affecting the colon, abdominal vessels, kidneys, ureters, bladder, and diaphragm [9].

A major concern is the dissemination of uterine tissue fragments into the abdominal cavity, particularly when malignancy is diagnosed postoperatively, such as endometrial carcinoma or sarcoma. Adequate preoperative evaluation is crucial to minimize this risk, including cervical cytology, high-risk human papillomavirus testing, endometrial biopsy, and ultrasound assessment of uterine fibroids. However, ultrasonography remains a weak point, as no definitive imaging markers currently exist to distinguish between benign fibroids and uterine sarcomas. The risk of an unexpected sarcoma diagnosis following presumed benign uterine surgery is estimated at approximately 0.2% [10].

If malignancy is identified postoperatively, morcellation significantly worsens the prognosis. It increases the recurrence rate at 1 year (RR=2.38; 95% CI: 1.05–5.38) and at 3 years (RR=1.88; 95% CI: 1.50–2.37), as well as mortality within the first year (RR=4.75; 95% CI: 1.03–22.0) [11]. Additionally, it impairs accurate staging in cases of incidental endometrial carcinoma.

Another important risk is the unintentional spread of benign fibroids or smooth muscle tumors of uncertain malignant potential, which may result in the development of peritoneal leiomyomas [12]. These lesions derive their blood supply from non-uterine sources and are a more common complication than morcellation-associated uterine sarcoma. The first reported case of DPL following laparoscopy – without morcellation – was documented in 1997 at a trocar site [13].

The incidence of DPL ranges between 0.12% and 0.95%, depending on the study, and may be slightly higher in patients undergoing morcellation during myomectomy vs patients undergoing morcellation during supracervical hysterectomy [14]. Its true incidence remains difficult to establish, especially given evolving surgical techniques [8]. In a study by Cucinella et al, 50% of patients with DPL presented with symptoms, although the sample size was limited to 4 cases [15]. In a larger analysis of nearly 2500 laparoscopic supracervical hysterectomies and myomectomies involving morcellation, 3 cases of DPL were reported (0.12%) [16].

A systematic review of 21 studies on DPL found the mean age at diagnosis to be 38.5 years [8]. In another review of 53 DPL cases, the mean age was 40 years (range 24–57 years), with an average interval of 57 months between surgery and symptom onset [16]. Abdominal pain was reported in 53% of cases [16], making it the most common presenting symptom [8]. This age range suggests a possible role of sex hormones in DPL pathogenesis, supported by findings of estrogen and progesterone receptor expression in resected lesions, as in the present case [9].

The most common sites for DPL include the peritoneum, omentum, and mesentery [8]. Another study identified frequent involvement of trocar sites, the broad ligament, pouch of Douglas, sigmoid colon, and omentum [16]. Intestinal lesions are particularly problematic due to risks of obstruction and the technical difficulties of excision. Severe complications such as bowel obstruction and sepsis requiring hemicolectomy have been described [17].

Interestingly, DPL has also been reported in patients without prior uterine surgery or with an intact uterus [18,19]. This phenomenon has been linked to the hormonal theory, suggesting metaplasia of stem cells under the influence of sex hormones [9].

Other benign complications associated with morcellation include de novo endometriosis and adenomyomas. The risk of developing de novo endometriosis after subtotal hysterectomy or myomectomy is estimated at approximately 1.4%, which is similar to the risk in control groups not exposed to morcellation [20].

DPL presents significant diagnostic challenges. Imaging studies such as transvaginal ultrasound and MRI play a central role. The presence of T2 hypointensity in extrauterine masses – similar to that of uterine fibroids – in patients with a history of fibroids or prior morcellation should raise suspicion for DPL [19]. Tumor markers have limited value, although elevated CA-125 has been reported in some DPL cases [21]. Histopathological examination remains the cornerstone of diagnosis. The presence of spindle-shaped smooth muscle cells and immunohistochemical positivity for markers such as SMA and desmin confirm the diagnosis [8]. Histopathology is especially critical, as the endoscopic appearance of the lesions can mimic peritoneal carcinomatosis [22].

Due to these risks, the U.S. Food and Drug Administration (FDA) has addressed the safety of power morcellation since 2014 [23]. In its updated 2020 guidelines [24], the FDA recommends the use of containment systems and advises against morcellation in postmenopausal women, women over the age of 50, or when the uterus can be removed intact via mini-laparotomy or vaginally [22].

Containment systems are designed to prevent tissue dissemination into the peritoneal cavity. However, their use presents several limitations, including poor visibility within the containment bag, longer operative times, risk of injury to adjacent structures (due to blind manipulation), and the possibility of the bag becoming trapped in the morcellation device [6]. Although they theoretically reduce tissue spread, evidence is limited due to the rarity of related complications. Experimental studies have demonstrated that even with intact bags, tissue fragments stained with blue dye were identified in the peritoneal cavity in 9.2% of cases [25].

Conclusions

This case highlights the importance of considering DPL in the differential diagnosis of abdominal or pelvic pain following laparoscopic hysterectomy with morcellation. The condition may mimic malignancy on imaging and underscores the critical role of histopathological evaluation in confirming this benign diagnosis.