Review
Parasitic leiomyomas: a systematic review

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Abstract

Background

Parasitic leiomyomas were first described as early as 1909 but are a rare condition. In recent years, due to the rise of laparoscopic surgery and power morcellation, several cases of parasitic leiomyomas associated with this surgical procedure have been reported.

Methods

A literature search was performed using PubMed, Embase and Google Scholar with the following combination of keywords: leiomyoma OR uterine neoplasms OR uterine myomectomy OR laparoscopy OR hysterectomy OR peritoneal neoplasms AND parasitic. Papers describing parasitic leiomyomas were included. The results of these studies are summarized herein.

Results

We retrieved abstracts of 756 papers. Of these, 591 were excluded for not fulfilling the inclusion criteria and 54 were removed as duplicates; after full-text assessment, 8 were rejected for presenting cases of malignancy and finally 103 were included in our systematic review. From these, we present information about 274 patients with parasitic leiomyomas. The mean age of women was 40 years (range 18–79 years); and 154 (56%) had no history of uterine surgery, the others (120, 44%) having had a previous myomectomy or hysterectomy. Of the total, 106 (39%) women had a history of power morcellation. The most frequent clinical symptom was abdominal pain (49%) and the most frequent presentation was disseminated peritoneal leiomyomatosis.

Conclusions

While parasitic leiomyoma was first described a century ago, the recent introduction of laparoscopic power morcellation has increased the number of reported cases.

Introduction

Uterine leiomyomas are the most common benign pelvic tumors in women; they occur in approximately 25% of reproductive-aged women and are noted on pathologic examination in approximately 80% of surgically removed uteri [1]. Surgery is recommended for symptomatic patients. Although there have been changes in technology and technique, the objectives remain the same: removing disease, minimizing morbidity and preserving fertility.

Leiomyomas are classified according to their location within the myometrium. The International Federation of Gynaecology and Obstetrics (FIGO) identifies eight different types of leiomyoma [2], Group 8 corresponding to those with atypical presentation, including parasitic leiomyoma.

Classically, parasitic leiomyomas have been defined as unusual variants of pedunculated leiomyomas that, for some reason, lie free from the uterus in the abdominal cavity, surviving by obtaining a blood supply from neighboring structures [3]. They may originate from leiomyomas that detach from the uterus and receive a blood supply from adjacent organs or from myoma fragments left in the peritoneal cavity after morcellation of a leiomyoma or the uterus, subsequently taking a blood supply from other organs and growing [4].

The first description of a parasitic leiomyoma was made by Kelly and Cullen in 1909 [5] and in 1951 a case was reported of parasitic leiomyomas in the bladder [6]. In recent years, with the rise of laparoscopic surgery [7], [8], a new type of parasitic leiomyoma has emerged, that with an iatrogenic origin, in particular, as a complication of laparoscopic myomectomy or hysterectomy that require morcellation of the surgical piece to be extracted through a small incision. After morcellation, small fragments of the leiomyoma may remain unnoticed in the abdominal cavity and become implanted and take a blood supply from neighboring structures. They can grow and cause symptoms, but may also be asymptomatic and be diagnosed as an incidental finding on clinical examination. The growth of parasitic leiomyomas seems to be influenced by sex hormones and growth factors, although more research is required to improve our understanding of their development [9].

Despite the attention currently being paid to iatrogenic parasitic leiomyomas, we thought it would be interesting to review all cases of parasitic leiomyomas published to date and assess their association with laparoscopic surgery and morcellation.

Section snippets

Methods

We carried out a systematic review of parasitic leiomyomas, and report it in accordance with the recommendations of the PRISMA statement for systematic reviews and meta-analyses [10].

The studies identified were considered to be valid if they included case reports or series of case reports of parasitic leiomyoma, or were systematic reviews. We did not consider valid publications that did not report any specific cases of parasitic leiomyoma, or in which histopathological findings did not reveal a

Results

We identified 482 publications through PubMed and 274 through Embase. After reading the abstracts of these 756 papers, we excluded 591 for not focusing on the subject of our study, parasitic leiomyoma, while a further 54 were removed for being duplicates or for being included in the two systematic reviews included [10], [11]. Reading the full text of the remaining 111 papers in detail, we found that just 103 presented cases of parasitic leiomyoma; the 8 other papers presented cases of

Discussion

The first laparoscopic hysterectomy was performed by Reich in 1989 [15], though the technique for carrying out laparoscopic myomectomies with manual morcellation with a scalpel had been described earlier, in 1980 [16]. Indeed, a series of cases of laparoscopic myomectomies was published in 1990 [17], electric power morcellation for extracting large fragments being first described 3 years later [18]. The use of laparoscopic surgery and morcellation for the surgical treatment of some

Location

Parasitic leiomyoma may occur as single or multiple leiomyomas or disseminated leiomyomatosis throughout the abdominal cavity. In our review, 151 cases (55%) corresponded to DPL.

Generally, parasitic leiomyomas are located in the pelvis, although due to the use of power morcellators, small fragments of tissue may be disseminated throughout the abdominal cavity and adhere to any organ they encounter, giving rise to parasitic leiomyomas. The most common presentation of this type of leiomyoma after

Treatment

In the majority of cases we have reviewed, parasitic leiomyomas were resected by laparoscopy, results implying that the technique is both feasible and safe [108], [109]. On the other hand, an abdominal approach is indicated if the location or size mean that a laparoscopic approach is not suitable, or if malignancy is suspected. When a parasitic leiomyoma is found in neighboring organs, a multidisciplinary team should be involved to ensure the effective removal of all diseased tissue.

Various

Long-term implications

Given that growth in the number of cases of parasitic leiomyomas is attributable to wider use of endoscopic techniques for uterine surgery, it is very important to be careful using this technique during the first surgery. Moreover, it is known that morcellation is not free of potentially serious complications [113]. One of the currently recognized problems is the risk of morcellation of an undiagnosed sarcoma during laparoscopic myomectomy or hysterectomy [114], and hence, various scientific

Conclusions

First reported more than 100 years ago, parasitic leiomyoma is a pathological entity that may occur spontaneously or iatrogenically. Recently, the number of cases has grown due to widespread use of laparoscopic surgery and power morcellation. One option to minimize the risk of parasitic leiomyoma following these procedures could be to perform morcellation in a containment bag.

Conflict of interest

The authors do not report conflict of interest.

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      Frequently, PLs present as pelvic or abdominal tumors separated from the uterus after a history of surgical morcellation.1–2 Myomatous fragments inadvertently spread within the peritoneal cavity are able to establish a new blood supply from adjacent structures and develop into PLs.3 Mainly owing to the growing diffusion of morcellation in minimally invasive hysterectomy or myomectomy, the incidence of PLs is increasing, and it is likely to be even higher than believed because most PLs may remain unsuspected until particular conditions lead to clinically relevant manifestations.1,2 No grant or other sources of funding have been received for the drawing up of this article.

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