Case Report

Surgical Cytoreduction for Disseminated Benign Disease After Open Power Uterine Morcellation Amanda Ramos, MD, Amanda N. Fader, MD, and Kara Long Roche, MD BACKGROUND: Uterine morcellation is a technique used in gynecologic surgery to facilitate a laparoscopic approach to the removal of an enlarged uterus. The safety of this technique has been a source of recent debate, as uterine morcellation can result in the intraperitoneal dissemination of undiagnosed uterine malignancies. CASES: We report on three women who previously underwent minimally invasive hysterectomy and open power morcellation for benign disease, who subsequently presented with peritoneal implants highly suspicious for malignancy. Each woman required a laparotomy and extensive, multiorgan resection to clear the disease. Benign pathology was diagnosed in all cases. CONCLUSION: Even in the setting of benign conditions, open power morcellation of the uterus may be associated with clinically significant dissemination of uterine disease. (Obstet Gynecol 2015;125:99–102) DOI: 10.1097/AOG.0000000000000549

Teaching Point 1. Open power morcellation of the uterus is capable of not only spreading malignant disease, but also disseminating benign disease that may cause significant morbidity to our patients.

From the Kelly Gynecologic Oncology Service, Department of Gynecology and Obstetrics, Johns Hopkins Hospital, Baltimore, Maryland. Corresponding author: Kara Long Roche, MD, 600 N. Wolfe St, Ste 281, Baltimore, MD 21287; e-mail: [email protected]. Financial Disclosure The authors did not report any potential conflicts of interest. © 2014 by The American College of Obstetricians and Gynecologists. Published by Lippincott Williams & Wilkins. ISSN: 0029-7844/15

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he most common gynecologic surgery performed in the United States is the hysterectomy.1 Minimally invasive procedures, specifically laparoscopic hysterectomy, result in decreased blood loss, shorter hospital stays, a faster return to normal activities, and fewer complications when compared with total abdominal hysterectomy.1 Various minimally invasive tools, including open power morcellation devices, allow surgeons to extract enlarged leiomyoma or uteri through laparoscopic incisions. Open power morcellation involves fragmenting the tissue specimen within the intraperitoneal cavity so that it can be removed through a small abdominal incision.2 The velocity of the power morcellator used to cut the uterus or leiomyoma into fragments has the potential to spread the morcellated tissue throughout the peritoneal cavity, which can result in seeding of the benign or malignant tumor. There have been numerous reports of peritoneal tumor dissemination after open power uterine morcellation in cases of unexpected malignancy, leading to increasing concerns about its routine use.3 Additionally, benign disseminated leiomyomatosis has been described after the morcellation of benign uterine leiomyomas. The exact incidence of this phenomenon is unknown. In this report, we present three cases of women treated at Johns Hopkins Hospital (Baltimore, MD) over a 3-month period in 2013 who previously underwent laparoscopic hysterectomy and open power morcellation of the uterus for benign conditions. They subsequently presented with intraperitoneal disease highly suggestive of a gynecologic malignancy. Each required an extensive surgical cytoreduction with a gynecologic oncologist. The final pathology revealed disseminated benign uterine pathology in all cases.

CASE 1 A 38-year-old woman, gravida 4 para 4, presented to our institution with left-sided abdominal pain and constipation. Eight months before presentation, she underwent a robotic-assisted total laparoscopic hysterectomy with a right salpingo-oophorectomy and left salpingectomy for a symptomatic leiomyomatous uterus the size of a pregnancy at 24 weeks of gestation. An open power morcellator was used to facilitate removal of the leiomyomatous uterus. Final pathology demonstrated proliferative endometrium and myometrium with multiple benign leiomyoma. A computed tomography (CT) scan revealed multiple soft tissue implants scattered throughout the abdomen and pelvis (Fig. 1). Implants were noted in the left adnexa,

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middle of the abdomen, pericolic gutters, left upper quadrant, splenic hilum, gastric cardia, and abdominal wall; the largest measured 9 cm. Her CA 125 level was elevated at 937 units/mL (normal range less than 35 units/mL). Exploratory laparotomy with radical resection of abdominal and pelvic tumors was performed. Mobilization of the splenic flexure, splenectomy with en bloc resection of tumor and portion of the left diaphragm, (Fig. 2) left colon resection with primary side-to-side anastomosis, total omentectomy, left oophorectomy, and bilateral ureterolysis was required to completely excise the tumor. Intraoperative findings revealed multiple large, firm, wellcircumscribed tumor masses throughout the abdomen and pelvis (Fig. 3). Intraoperative blood loss was 500 cm3. At the conclusion of the procedure, there was no gross residual disease. The postoperative course was complicated by hemoperitoneum from a bleeding accessory splenic artery requiring splenic artery embolization and transfusion of 2 units of packed red blood cells. She was discharged from the hospital on postoperative day 5. Final pathology revealed benign adenomyomatosis.

CASE 2 A 51-year-old woman, gravida 2 para 2, presented with abdominal pain, dysuria, and pelvic pressure. A transvaginal ultrasonogram revealed a heterogeneous soft tissue lesion in the pelvis measuring 6 cm. A follow-up magnetic resonance image (MRI) showed a 6.8-cm peripherally enhancing mass with significant fat stranding, inflammation, and enhancement of the surrounding tissues including the rectosigmoid colon, anterior vaginal cuff, and superior margin of the bladder. Six months before presentation, she had undergone a robotic-assisted total laparoscopic hysterectomy with open power morcellation and right salpingo-oophorectomy with left salpingectomy and extensive enterolysis. Pathology of

the morcellated uterus revealed secretory endometrial tissue with fragments of uterine leiomyoma. Our patient underwent an exploratory laparotomy, radical debulking of the pelvic mass, bilateral ureterolysis, left salpingo-oophorectomy, partial bladder cystectomy, rectosigmoid resection with functional side-to-side and end-to-end anastomosis, infracolic omentectomy, placement of omental J flap in the pelvis, and mobilization of hepatic and splenic flexure with extensive enterolysis. Intraoperative findings revealed a 15-cm pelvic mass, densely adherent to the anterior abdominal wall, the bladder, the bilateral ureters, and the rectosigmoid colon. Her postoperative course was complicated by a wound infection requiring intravenous antibiotics. She was discharged home on postoperative day 12. Final pathology revealed fibrous tissue with marked acute and chronic inflammation and endometriosis.

CASE 3 A 51-year-old woman, gravida 2 para 2, presented to the emergency department with nausea, vomiting, and abdominal pain. A CT scan showed a small bowel obstruction, a heterogeneous and irregular solid lesion measuring approximately 5.734.5 cm in size in the central pelvis, pelvic ascites, and slight peritoneal nodularity measuring approximately 2.331 cm in the left pelvis. Physical examination at that time was notable for a large, immobile pelvic mass filling the posterior cul-de-sac. Her CA 125 level was 17.3 units/mL. Approximately 1 year before presentation, she underwent a total laparoscopic hysterectomy with open power morcellation for a leiomyomatous uterus. Pathology at that time was consistent with benign leiomyoma.

Fig. 1. Abdominal computed tomography scan performed on patient 1, axial image. The perisplenic (yellow arrow) and subdiaphragmatic (red arrow) masses are appreciated. These masses caused the patient a sensation of discomfort and bloating.

Fig. 2. Gross pathology from the exploratory laparotomy performed on patient 1. This is the spleen with three attached “morcellomas.”

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Our patient underwent an exploratory laparotomy with extensive lysis of adhesions, radical resection of the pelvic mass, and right salpingo-oophorectomy. Intraoperative findings were notable for extensive adhesions from the sigmoid colon and small bowel to the pelvic mass, resulting in a small bowel obstruction. Her postoperative course was uneventful, and she was discharged to her home on postoperative day 6 in good condition. The final pathology of the resected mass was a benign leiomyoma with ischemic changes.

DISCUSSION This series highlights three cases of disseminated, benign uterine pathology presenting between 6 and 12 months after minimally invasive hysterectomy with open power morcellation. In each case, an advanced gynecologic malignancy was suspected and radical surgical resection by a gynecologic oncologist was required to clear the disease. While there is currently much debate regarding the safety of morcellation techniques in the setting of an undiagnosed malignancy, this series demonstrates the potential risks even in the setting of benign uterine pathology. Iatrogenic endometriosis is a rare occurrence that may be observed after morcellation of the uterus during laparoscopic hysterectomy.4–6 Radiologically, disseminated endometriosis may appear malignant and therefore surgical intervention with definitive pathologic diagnosis is generally required. While this phenomenon has been previously reported, the extent of disease and radical resection required in the patients presented in the current case report is unique. One patient in this series was diagnosed

Fig. 3. Disseminated adenomyoma in situ adherent to the diaphragm and spleen in patient 1. Ramos. Cytoreduction After Uterine Morcellation. Obstet Gynecol 2015.

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with disseminated endometriosis and one with disseminated adenomyosis; interestingly, neither patient had this respective pathology identified at the time of hysterectomy. The current body of literature regarding the risk of dissemination of disease after uterine open power morcellation describes a broad spectrum of patient outcomes. In a case series performed by Shuster et al, the incidence of new-onset endometriosis between women undergoing a supracervical hysterectomy with uterine morcellation compared with those women undergoing traditional vaginal or abdominal hysterectomy was not significantly different regardless of whether endometriosis was present at the time of hysterectomy. Three cases of endometriosis after uterine morcellation were reported, all of which were managed with a minimally invasive approach.6 In a retrospective review by Donnez et al, eight cases of disseminated adenomyosis were reported among 1,405 cases of women undergoing laparoscopic subtotal hysterectomy with open uterine morcellation. 4 These women presented with pelvic masses ranging from 2 to 8 cm and mildly elevated CA 125 levels (19.4–128 units/mL), and all were managed with minimally invasive surgical resections.4 A case report by Hilger et al describes a patient who presented 1 year after initial laparoscopic supracervical hysterectomy and uterine morcellation with pelvic pain and a CA 125 level of 95 units/mL. Diagnostic imaging revealed a complex right adnexal mass later diagnosed as endometrial and myometrial tissue. Several pelvic masses were identified during repeat robotic-assisted surgery consistent with a diagnosis of disseminated endometrial and myometrial tissue. Our case series describes a much more extreme presentation of disseminated benign disease, with each patient requiring a radical dissection or multiorgan resection to clear her disease. Overall, the incidence of disseminated malignant or benign uterine pathology after morcellation is poorly understood and is likely underreported in the literature.7 However, as minimally invasive techniques and the use of open power morcellators have become more widespread, it is possible that these undesirable outcomes will increase. In contemporary practice, power morcellators are usually operated intraperitoneally, but not necessarily within an enclosed environment, such as a bag or device that contains the tissue fragments. Thus, open power morcellators have an inherent design flaw, as the device allows for bits of tissue to spread throughout the peritoneal cavity.

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Minimally invasive hysterectomy facilitated by open power morcellation has benefited thousands of women, by allowing those women to forego the risks and longer recovery periods associated with laparotomy.3 However, while morcellation has the potential to benefit many women, recent reports and a cautionary safety announcement by the Food and Drug Administration, suggest that the incidence of undetected uterine sarcoma may be higher than previously recognized in this population, and that women with an undetected malignancy may become upstaged by iatrogenic dissemination or spread requiring further treatment and resulting in poorer oncologic outcomes. Approximately 15% of patients will be upstaged by re-exploration, especially patients with leiomyosarcoma. These patients will also experience decreased disease-free survival and overall survival.3 Any gynecologic surgeon considering performance of uterine morcellation should perform a thorough preoperative workup to rule out uterine malignancy and to assess a patient’s candidacy for this procedure, including endometrial biopsy, cervical cytology, and consideration of MRI. Additionally, the patient should be appropriately counseled regarding the risk of an occult malignancy and seeding of either benign or malignant tissue within the abdomen and pelvis. Further investigation into the risks of malignant and benign disease dissemination after uterine mor-

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cellation is warranted. Moreover, as concerns increase regarding this practice, there is an urgent need for continued professional dialogue, innovation, and technologic advancements to facilitate the safe, minimally invasive removal of enlarged uterine pathology. REFERENCES 1. Jonsdottir GM, Jorgensen S, Cohen SL, Wright KN, Shah NT, Chavan N, et al. Increasing minimally invasive hysterectomy: effect on cost and complications. Obstet Gynecol 2011;117: 1142–9. 2. Steiner RA, Wight E, Tadir Y, Haller U. Electrical cutting device for laparoscopic removal of tissue from the abdominal cavity. Obstet Gynecol 1993;81:471–4. 3. Odebuyo T, Rauh-Hain AJ, Meserve EE, Seidman MA, Hinchcliff E, George S, et al. The value of re-exploration in patients with inadvertently morcellated uterine sarcoma. Gynecol Oncol 2014;132:360–5. 4. Donnez O, Squifflet J, Leconte I, Jadoul P, Donnez J. Posthysterectomy pelvic adenomyotic masses observed in 8 cases out of a series of 1405 laparoscopic subtotal hysterectomies. J Minim Invasive Gynecol 2007;14:156–60. 5. Hilger WS, Magrina JF. Removal of pelvic leiomyomata and endometriosis five years and supracervical hysterectomy. Obstet Gynecol 2006;108:772–4. 6. Schuster MW, Wheeler TL II, Richter HE. Endometriosis after laparoscopic supracervical hysterectomy with uterine morcellation: a case control study. J Minim Invasive Gynecol 2012;19: 183–7. 7. Milad MP, Milad EA. Laparoscopic morcellator-related complications. J Minim Invasive Gynecol 2014;21:486–91.

OBSTETRICS & GYNECOLOGY

Surgical cytoreduction for disseminated benign disease after open power uterine morcellation.

Uterine morcellation is a technique used in gynecologic surgery to facilitate a laparoscopic approach to the removal of an enlarged uterus. The safety...
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