Akihiro Takeda, MD, Masahiko Mori, MD, Kotaro Sakai, MD, Takashi Mitsui, MD, and Hiromi Nakamura, MD

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1 Journal of Minimally Invasive Gynecology (2007) 14, Parasitic peritoneal leiomyomatosis diagnosed 6 years after laparoscopic myomectomy with electric tissue morcellation: Report of a case and review of the literature Akihiro Takeda, MD, Masahiko Mori, MD, Kotaro Sakai, MD, Takashi Mitsui, MD, and Hiromi Nakamura, MD From the Department of Obstetrics and Gynecology, Gifu Prefectural Tajimi Hospital, Tajimi, Japan (all authors). KEYWORDS: Parasitic peritoneal leiomyomatosis; myomectomy; Electric tissue morcellation Abstract. A 33-year-old woman, gravid 2 para 2, underwent laparoscopic myomectomy with electric tissue morcellation for intraligamental myoma. Six years later, asymptomatic pelvic tumor was found during a routine checkup. Under laparoscopic observation, multiple soft tumors were detected in the peritoneal cavity and these tumors were successfully excised by a laparoscopic-assisted procedure. Pathological examination demonstrated that these tumors were progesterone receptor-positive leio that were almost identical histologically to the myoma tissue excised 6 years earlier AAGL. All rights reserved. Uterine leio occur in approximately 20% of women aged 35 years or older. 1 Although they are frequently asymptomatic, it is well recognized that uterine leiomyoma may cause metrorrhagia, infertility, abdominal distention, pelvic pain, miscarriage, or premature delivery. For symptomatic patients, surgical interventions are usually considered. In addition to laparotomic procedures, recent advances in surgical equipment and techniques have facilitated minimally invasive endoscopic surgical treatment. 2,3 In these endoscopic procedures, irrespective of 2 or myomectomy, 3 retrieval of myoma tissue through minimal abdominal-wall incision requires fragmentation in the abdominal cavity 4,5 if the myoma tissue can not be extracted The authors have no commercial, proprietary, or financial interest in the products or companies described in this article. Corresponding author: Akihiro Takeda, MD, Department of Obstetrics and Gynecology, Gifu Prefectural Tajimi Hospital, Maebata-cho, Tajimi, Gifu gyendoscopy@gmail.com Submitted May 9, Accepted for publication July 11, through vagina or posterior colpotomy. Although the fragmentation process of myoma tissue may cause dissemination of viable myoma particles in the abdominal cavity and, in rare occasions, these particles may survive and become implanted onto tissue such as the peritoneum and omentum, reports of such cases are limited. 6,7 In this report, a case of peritoneal leiomyomatosis found 6 years after laparoscopic myomectomy with electric tissue morcellation is described with a review of the literature. Case report A 33-year-old woman, gravida 2 para 2, with no history of disease, underwent laparoscopic myomectomy after administration of gonadotropin-releasing hormone agonist at our hospital in Gasless laparoscopic surgery was performed by subcutaneous abdominal wall lift method. 8 A supraumbilical 5-mm port was established for laparo /$ -see front matter 2007 AAGL. All rights reserved. doi: /j.jmig

Takeda et al Parasitic peritoneal leiomyomatosis 771 Figure 1 findings at initial surgery. (A) Subserosal myoma located in left intraligamental portion.

An additional 5-mm port left lateral to the umbilicus, a 12-mm port in the right lower abdomen, and suprapubic 12-mm port were established for the surgical procedure.

2 Takeda et al Parasitic peritoneal leiomyomatosis 771 Figure 1 findings at initial surgery. (A) Subserosal myoma located in left intraligamental portion. (B) Electromechanical tissue morcellator was used to downsize myoma node before enucleation. scopic observation. An additional 5-mm port left lateral to the umbilicus, a 12-mm port in the right lower abdomen, and suprapubic 12-mm port were established for the surgical procedure. Under laparoscopic observation, a subserosal myoma node measuring 8 7 cm was identified in the left intraligamental portion (Figure 1A), and the broad ligament was opened. Because mobility of myoma node was limited and identification of the myoma bed was difficult, this myoma node was partially downsized by fragmentation with a Steiner electromechanical morcellator 4 (Karl Storz Endoscopy Japan, Tokyo, Japan) (Figure 1B). Then after achieving mobility, the myoma bed was dissected, and myomectomy was completed. Myoma tissue was removed from the abdominal cavity through the 12-mm port. Excised tissue weight was 215 g. No other myoma nodes were identified. After extensive washing of the peritoneal cavity, the surgical wound was closed. Surgical duration was 96 minutes, and blood loss was less than 20 ml. Postoperative course was uneventful, and the patient was discharged 3 days after surgery. Without any significant pelvic findings, her follow-up at the outpatient clinic was terminated 10 months after surgery. Six years after the initial surgery, at 39 years of age, she was referred to our outpatient clinic because of an asymptomatic pelvic tumor found during routine checkup. On magnetic resonance imaging, the bilateral ovaries and uterus appeared normal in shape, and multiple tumors ranging from 1 to 6 cm with unknown origin were identified in the lower abdomen (Figure 2). Blood cell counts and C-reactive protein showed normal values. Serum lactate dehydrogenase (192 IU/L) and tumor marker values (alpha fetoprotein: 3.28 ng/ml; carcinoembryonic antigen: 1.52 ng/ ml; CA19-9: U/mL; CA125: U/mL; squamos cell carcinoma-related antigen: 0.8 ng/ml) were within the normal range. Gasless laparoscopic-assisted surgery was performed by the subcutaneous abdominal wall lift method with an Alexis wound retractor (Applied Medical, Rancho Santa Margarita, CA) as previously described. 9 Under laparoscopic observation, Figure 2 Magnetic resonance images of peritoneal leiomyomatosis found 6 years after laparoscopic myomectomy with electric tissue morcellation. (A) Axial T2-weighted magnetic resonance image of (arrows) located in abdominal cavity. Uterus showed normal shape. (B) Sagittal T2-weighted magnetic resonance image of (arrows) located in abdominal cavity.

772 Journal of Minimally Invasive Gynecology, Vol 14, No 6, November/December 2007 Figure 3 findings of peritoneal leiomyomatosis found 6 years after laparoscopic myomectomy with electric tissue

3 772 Journal of Minimally Invasive Gynecology, Vol 14, No 6, November/December 2007 Figure 3 findings of peritoneal leiomyomatosis found 6 years after laparoscopic myomectomy with electric tissue morcellation. Parasitic myoma attached to: (A) omentum; (B) round ligament; (C) peritoneum of left pelvic sidewall adjacent to left ureter; and (D) vesicouterine peritoneum. multiple soft tumors ranging from 1 to 6 cm were identified in the lower abdomen. Tumors were firmly attached to the omentum (Figure 3A), right round ligament (Figure 3B), left pelvic sidewall peritoneum (Figure 3C), peritoneum of the vesicouterine pouch (Figure 3D), and peritoneum of pouch of Douglas. There were no direct communications between the uterus and these nodules, and the major blood supply came from either omentum or peritoneum. Uterus, bilateral ovaries, and fallopian tubes appeared normal. These nodules were dissected and excised at the base of the tumor by Ligasure Atlas (Tyco Healthcare, Tokyo, Japan). A tumor on the left pelvic sidewall was firmly attached to peritoneal tissue adjacent to the ureter. Because there was concern about the risk of ureteral injury, a ureteral stent was placed in the left ureter under cystoscopic observation before blunt dissection, then the nodule was removed. After fragmentation by surgical scalpel through the suprapubic port, 9 the excised tissue was removed from the abdominal cavity. Five tumors were resected and excised tissue weight was 105 g. Surgical duration was 79 minutes, and blood loss was less than 20 ml. There were no intraoperative or postoperative complications. The patient was discharged 3 days after surgery. Histopathologic findings of the tumor tissues obtained from the initial and second surgeries were compared. Tumors obtained from second surgery showed a histologic appearance of leiomyoma (Figure 4B) that was almost identical to the leiomyoma tissue excised 6 years earlier (Figure 4A). By using sections taken from formalin-fixed, paraffinembedded blocks, immunohistochemical staining for estrogen and progesterone receptors was carried out. Although progesterone receptor was positive in the nuclei of leiomyoma cells obtained from the initial (Figure 4C) and second (Figure 4D) surgery, estrogen receptor- was not detected under the current experimental conditions. Discussion Parasitic myoma is classically known as the rare variant of pedunculated subserosal myoma that has for some reason become partially or completely separated from the uterus and started to receive an alternative blood supply from another source such as the omentum and mesenteric vessels. 10 In contrast to such spontaneously occurring, the recent prevalence of minimally invasive endoscopic procedures 2,3 may promote a new type of myoma 6,7 that can be called an iatrogenic myoma. According to our experience, even with correct use, careful observation has shown that the electric tissue morcellator produces minute fragments of myoma tissue. There is a possibility that these fragments will survive and become implanted onto peritoneal or omental tissue. With the inclusion of the current case, there have been 4 cases of myoma after laparoscopic surgery reported to date under various diagnostic names in the literature (Table 1). 6,7 In 2

4 Takeda et al Parasitic peritoneal leiomyomatosis 773 Figure 4 Histologic findings of myoma tissue. (A) Myoma tissue resected at initial laparoscopic myomectomy after administration of gonadotropin-releasing hormone agonist. (Hematoxylin-eosin staining, scale bar: 50 m.) (B) Recurrent myoma tissue resected at second surgery. (Hematoxylin-eosin staining, scale bar: 50 m.) (C) Immunohistochemical localization of progesterone receptor in nuclei of myoma cells resected at initial surgery. (Immunoperoxidase staining, scale bar: 50 m.) (D) Immunohistochemical localization of progesterone receptor in nuclei of myoma cells resected at second surgery. (Immunoperoxidase staining, scale bar: 50 m.) of these cases, the initial surgery was laparoscopic myomectomy, whereas laparoscopic myomectomy and second total laparoscopic, and laparoscopic myomectomy and second laparoscopic supracervical, were performed in 1case each. Because an electric tissue morcellator was used for fragmentation of myoma tissue in these cases, it is assumed that production of tiny remnant fragments of myoma tissue had a close relationship to the later occurrence of myoma. Further, when diagnosing the iatrogenic myoma, it is important to differentiate from spontaneously occurring rare condition called disseminated leiomyomatosis peritonei 11 by carefully obtaining the patient s surgical history. Symptoms of are not specific. The palpable mass was noted in 2 cases, whereas in our case, asymptomatic tumors were found at regular checkup. The interval between the initial surgery and the second surgery ranged from 8 to 72 months. Parasitic occurred at various sites involving the peritoneum and omentum in the abdominal cavity. These facts indicate that possible occurrence of myoma, as a rare complication of myoma tissue morcellation, should be explained to the patient and family before surgery when the use of an electromechanical morcellator for fragmentation of myoma tissue is planned. During surgery, presence of myoma remnants in the abdominal cavity should be carefully inspected, and even small fragments should be recovered for prevention. At the end of surgery, vigorous irrigation should be performed during procedures in which the potential for spillage exists. Further, long-term follow-up is recommended to detect the occurrence of myoma at least until the patient becomes postmenopausal. On histologic examination of excised tissue, 1 case involved cellular leiomyoma, whereas 3 cases were diagnosed as common leiomyoma. Except for our case that showed a close similarity of histologic appearance between tissues obtained from the initial and second surgeries, histologic comparison between tissues obtained from initial and second surgeries were not available in the other 3 cases. Because this comparison is thought to be important to investigate the developmental mechanism of, histologic study of the original myoma tissue and myoma should be described in future case reports whenever possible. Presence of steroid hormone receptors in myoma tissue was investigated in 2 cases including the current case. In the case of cellular leiomyoma, estrogen and progesterone receptors were positive, 7 whereas, in our case, only the progesterone receptor was detected. Although it is not known whether steroid hormone receptor status 12 plays a significant role in the development and growth of, further accumulation of cases is required to assess the developmental mechanism of myoma.

5 Table 1 Study Paul et al. 6 Sinha et al. 7 Current report Parasitic after laparoscopic surgery reported in the literature Diagnosis Multiple peritoneal Postlaparoscopic Postlaparoscopic Parasitic peritoneal leiomyomatosis Previous surgery myomectomy myomectomy, total laparoscopic myomectomy, laparoscopic supracervical myomectomy Age at previous surgery, yr Use of electric tissue morcellator at previous surgery Signs and symptoms caused by Interval between first and second surgery, mo Second surgical procedure 28 Yes N/A 30 41, 45 Yes Pain, mass in abdomen 34, 40 Yes Mass in abdomen 33 Yes Asymptomatic, found at regular checkup ER estrogen receptor; N/A not available; PgR progesterone receptor assisted Location of myoma nodules Parietal peritoneum at previous port site, superior portion to fundus of uterus, right paracolic gutter Sigmoid colon serosa, lateral pelvic wall, pouch of Douglas Sigmoid colon serosa Omentum, right round ligament, left pelvic sidewall peritoneum of vesicouterine pouch, peritoneum of pouch of Douglas Size of myoma nodules, cm No. of myoma nodules Histopathologic findings N/A 3 Leiomyoma 15, 7, 8 (total weight: 2020 g) 10 (total weight: 360 g) 6, 4, 4.5, 3.5, 1 (total weight: 105 g) 3 Cellular leiomyoma (ER positive, PgR positive) 1 Leiomyoma 5 Leiomyoma (ER negative, PgR positive) 774 Journal of Minimally Invasive Gynecology, Vol 14, No 6, November/December 2007

6 Takeda et al Parasitic peritoneal leiomyomatosis 775 After encountering this case, records of 38 patients who underwent laparoscopic myomectomy using an electric tissue morcellator between 1999 and 2000 at our hospital were reviewed. Seven patients were lost to follow-up within 6 months, whereas the other 31 patients were followed up for a median of 60 months (range: months). Eleven cases showed myoma recurrence in the uterine musculature. Although 5 patients underwent a second surgical intervention (laparoscopic-assisted vaginal : 2, laparoscopic-assisted myomectomy: 2, and myomectomy at cesarean section: 1), the other 6 patients did not receive further treatment. At this time, there have not been any noted in these patients. Conclusion Parasitic peritoneal leiomyomatosis could be an extremely rare complication of endoscopic surgery when myoma tissues are removed through minimal incision after fragmentation. Further accumulation of cases are required to assess actual incidence and precise developmental mechanism of this rare disorder. References 1. Buttram VC, Reiter RC. Uterine leiomyomata: etiology, symptomatology and management. Fertil Steril. 1981;36: Istre O, Langebrekke A, Qvigstad E. Changing technique from open abdominal to laparoscopic: new trend in Oslo, Norway. J Minim Invasive Gynecol. 2007;14: Hurst BS, Matthews ML, Marshburn PB. myomectomy for symptomatic uterine. Fertil Steril. 2005;83: Steiner RA, Wight E, Tadir Y, et al. Electrical cutting device for laparoscopic of tissue from the abdominal cavity. Obstet Gynecol. 1993;812: Carter JE, McCarus SD. myomectomy: time and cost analysis of power vs manual morcellation. J Reprod Med. 1997;42: Paul PG, Koshy AK. Multiple peritoneal after laparoscopic myomectomy and morcellation. Fertil Steril. 2006;85: Sinha R, Sundaram M, Mahajan C, et al. Multiple leio after laparoscopic : report of two cases. J Minim Invasive Gynecol. 2007;14: Takeda A, Manabe S, Hosono S, et al. A case of a mature cystic teratoma of the uterosacral ligament. J Minim Invasive Gynecol. 2005; 12: Takeda A, Sakai K, Mitsui T, et al. Wound retraction system for gasless laparoscopic-assisted myomectomy with a subcutaneous abdominal wall-lift method. J Minim Invasive Gynecol. 2007;14: Brody S. Parasitic fibroid. Am J Obstet Gynecol. 1953;65: Advincula AP, Hernandez JC, Lieberman R. Disseminated leiomyomatosis peritonei. Fertil Steril. 2005;84: Nisolle M, Gillerot S, Casanas-Roux F, et al. Immunohistochemical study of the proliferation index, estrogen receptors and progesterone receptors A and B in leiomyomata and normal myometrium during the menstrual cycle and under gonadotrophin-releasing hormone agonist therapy. Hum Reprod. 1999;14:

Case Report Torsion of a Subserosal Myoma Managed by Gasless Laparoendoscopic Single-Site Myomectomy with In-Bag Manual Extraction

Case Reports in Obstetrics and Gynecology Volume 2016, Article ID 7831270, 4 pages http://dx.doi.org/10.1155/2016/7831270 Case Report Torsion of a Subserosal Myoma Managed by Gasless Laparoendoscopic Single-Site