De Novo Endometrial Implant Into the Colon After Uterine Morcellation

Instances of iatrogenic endometriosis after laparoscopic supracervical hysterectomy are uncommon but have been reported in the literature in women undergoing hysterectomy for pelvic pain. A 41-year-old woman with no history of endometriosis developed cyclic pain and a mass in her lower abdominal wall after uterine morcellation for a laparoscopic supracervical hysterectomy and sacrocolpopexy for uterocervical prolapse. Exploratory surgery revealed an endometrial implant involving the peritoneum and omentum, with transmural involvement of the cecum at a site separate from the trocar site. Aberrant endometrial cell implantation after morcellation of the uterine corpus has been reported; however, this was a rare instance of transmural large bowel iatrogenic endometriosis, necessitating a partial bowel resection, in a patient without a history of endometriosis or pelvic pain, at a site remote from where the uterus was morcellated and removed. In light of recent concerns over dissemination of occult cancerous cells, this case illustrates that dissemination of noncancerous cells can similarly have detrimental outcomes for patients. Key Words: Endometriosis, Laparoscopy, Supracervical hysterectomy, Uterine morcellation.

Endometriosis is defined by the presence of endometrial glands and stroma outside the lining of the endometrial cavity. Sites that may be seeded with these endometrial glands include the ovaries, peritoneum, rectovaginal septum, ab- dominal wall, bowel, ureter, and even lung paren- chyma. 1,2 Endometriosis affects approximately 6% to 10% of all reproductive-aged women and accounts for 35% to 50% of women who experience cyclic chronic pelvic pain and/or have complications with fertility. The severity of the symptoms differs greatly among these women and has no correlation with the extent of disease. 2 Cases of postoperative iatrogenic endometriosis after pel- vic surgery are rare. In women with known histories of endometriosis, surgical scar endometriomas have been documented after surgeries that involve the uterus, such as a Cesarean section or hysterectomy. 3–5 The proposed mechanism for trocar-site or surgical-scar endometriosis is spillage of endometrial cells at the time of surgery, im- plantation of these cells, and proliferation to form endo- metriomas. 6 More than 200,000 women undergo surgical correction for pelvic organ prolapse each year. Advances in minimally invasive surgical techniques facilitate the ability of sur- geons to perform sacrocolpopexy procedures for vault prolapse with decreased morbidity. 7,8 For women who have uteri at the time of the procedure, a supracervical hysterectomy is generally performed to allow a durable repair, while minimizing the risk for mesh complications. As the number of laparoscopic sacrocolpopexy proce- dures increases, uterine morcellation will be performed more frequently. Uterine morcellation may potentiate dissemination of en- dometrial cells in the peritoneal cavity. In reproductive- aged women, abnormal seeding of endometrial cells may respond to hormonal fluxes and produce new-onset symptoms of endometriosis in women with histories of it. Evidence of implantation of endometriomas after laparo- Citation Mellano EM, Anthony CM, Grisales T, Tarnay CM. De novo endometrial implant into the colon after uterine morcellation. CRSLS e2014.00403. DOI: 10.4293/CRSLS.2014.00403. Copyright © 2014 SLS This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-ShareAlike 3.0 Unported license, which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original author and source are c redited. Address correspondence to: Erin M. Mellano, MD, University of California, Los Angeles, David Geffen School of Medicine, Department of Obstetrics an d Gynecology, Division of Female Pelvic Medicine and Reconstructive Surgery, 10833 Le Conte Avenie, Box 951740, Los Angeles, CA 90095-1740, USA. Telephone: 310-2 67-5918, Fax: 310-794-6635; E-mail: [email protected] 1e2014.00403 CRSLS MIS Case Reports from SLS.org CASE REPORT scopic supracervical hysterectomy has been documented in women without evidence of the disease before surgery. In these cases, the patients undergoing hysterectomy pre- operatively reported dysmenorrhea or pelvic pain sug- gesting preexisting endometriosis. 9 –13 The present case report is unique in that an endometrial implant developed well away from the site of morcellation, involving the large bowel and peritoneum, in a woman with no history of pelvic pain or signs of endometriosis at the time of her index operation. CASE A 41-year-old, parous 2 woman was referred to our office for symptomatic stage 3 pelvic organ prolapse and urinary stress incontinence for which she desired surgical correc- tion. She underwent a laparoscopic supracervical hyster- ectomy, sacrocolpopexy, posterior colporrhaphy, perine- orrhaphy, retropubic midurethral sling, and cystoscopy. The procedure was uncomplicated, and the pelvic anat- omy was visually normal, including the ovaries, which were left in situ. The uterus was morcellated intraperi- toneally with a power morcellator and was removed through a 12-mm umbilical incision under direct visu- alization with the camera in the 12-mm right lower quadrant assistant port. Her postoperative course was uncomplicated, and she was discharged home on post- operative day 2. Approximately 5 months after her surgery, the patient began to experience cyclic pain and vaginal spotting. The vaginal spotting was attributed to residual endometrial glands in the endocervical canal. She had no history of dysmenorrhea and no overt endometriosis noted at the time of her surgery. The results of physical examination were unremarkable, and an in-office ultrasound study revealed normal-appearing adnexal structures. She was treated with nonsteroidal anti-inflammatory medication for her pain. Over the next year, she continued to be vexed with right low abdominal wall pain at the site of a self-reported abdominal firmness that expanded and regressed in size cyclically with menstruation. Interval imaging studies, in- cluding ultrasound and computed tomography (CT), were performed and were unable to locate an abnormality in the anterior abdominal wall. Of note, an inflammatory focus along the lower margin of the cecum in the right lower quadrant, 15 mm in size with some fat stranding and trace fluid, interpreted as postsurgical changes, was seen on CT /H1102212 months after her surgery. Attempts to treat her pain with local anesthetic and oral analgesics were largely unsuccessful. Given the cyclic nature of the pain, a 1-month trial of a gonadotropin-releasing hormone ago- nist was administered. The patient noticed a dramatic improvement of her pain with hormonal suppression. Eighteen months after surgery, she was examined while on her menstrual cycle, and a palpable mass was appre- ciated, medial and inferior to the right lower quadrant assistant laparoscopic port site in the anterior abdominal wall. Her symptoms and the response to hormonal sup- pression were suggestive of endometriosis, despite un- characteristic location, distant from the umbilical port where the morcellation and uterine tissue had been re- moved. Magnetic resonance imaging (MRI) performed during menstruation did not demonstrate any visually enhancing lesions in the abdominal wall. Despite nondi- agnostic imaging results, the presumptive clinical conclu- sion was an endometriotic implant. Given the debilitating nature of this pain, the patient elected to undergo an exploration and excision of this mass. On examination under anesthesia, a 3 /H110034 cm palpable mass in the anterior abdominal wall was identified. The palpable mass was below the level of the fascia, and upon entry into the peritoneum, a lobulated 3 /H110035 cm mass was encountered. This was adherent to the peritoneum and omentum, with a gross appearance consistent with endo- metriosis. The conglomerate involved the nearby viscera, with transmural involvement of the cecum ( Figure 1 ). The mass, a portion of the cecum, and the adjacent ap- pendix were resected with the assistance of a general surgeon ( Figure 2 ). Intraoperative frozen section was consistent with endo- metriosis. No additional lesions or masses were visualized in the peritoneal cavity. The patient had an uneventful recovery and was discharged home 2 days postopera- tively. Final pathology confirmed the mass to be an irreg- ular 5 /H110033.4 /H110032.0 cm fragment of yellow-gray, fibrofatty, indurated tissue with focal cavities filled with blood, sug- Figure 1. Cecal involvement of endometriotic implant. De Novo Endometrial Implant Into the Colon After Uterine Morcellation, Mellano EM et al. 2e2014.00403 CRSLS MIS Case Reports from SLS.org gestive of endometrioma. The cecum was noted to have transmural infiltration of the endometrioma.

Our patient presented postoperatively with a clinical pic- ture classic for endometriosis, with cyclic pain and a palpable, tender mass on examination. Although cases of iatrogenic endometriotic implants have been reported af- ter pelvic surgery, this case is unique in that the patient had no history suggestive of endometriosis, nor were there any intraoperative findings of endometriosis at the time of her index surgery. Case reports of de novo endo- metriosis after uterine morcellation have been notable in patients with preoperative symptoms of pelvic pain, sug- gestive of preexisting endometriosis. 9 –13 The absence of preoperative pelvic pain or intraoperative signs of preexisting endometriosis implies that the most likely etiology of our patient’s endometrioma was direct seeding of endometrial cells after morcellation. What is interesting about this particular case is that the endometri- otic implant did not involve a laparoscopic port site. Furthermore, it was just inferior to the right lateral trocar site, where the laparoscope was used during morcellation through the umbilical port. It is possible that the viewing angle in relationship to the right lower quadrant port site precluded the surgeons from recognizing a fragment of retained uterine tissue. Traditional imagining techniques used to diagnose endo- metriosis include ultrasound, MRI, and CT. It is unusual to be unable to identify abdominal wall endometriotic im- plants with MRI. The sensitivity of MRI in detecting endo- metriosis ranges from 78% to 96.3%, with specificity of 73% to 100%. 14 –16 Although the palpable lesion in this patient was thought to be in the anterior abdominal wall, the true transcecal location made it significantly more challenging to identify on imaging. Ultrasound, double- contrast barium enema, CT, and MRI have all been used to diagnose bowel endometriosis, but they have limita- tions. 17 The sensitivity of these techniques can be reduced by bowel peristalsis, even after bowel preparation. An antispasmodic may be helpful in visualizing intestinal en- dometriosis by preventing artifacts from peristalsis. 15 Gad- olinium may help enhance a study, but there is no con- sensus of benefit. Recent studies advocate for the use of 3.0-T MRI to increase sensitivity and specificity in detect- ing deep infiltrating endometriosis, compared with the routine 1.5-T pelvic MRI protocol. 14 Regardless of imaging modality, high clinical suspicion should not be dissuaded by negative imaging. A laparoscopic supracervical hysterectomy performed at the time of colpopexy mandates that the uterus be mor- cellated to remove the specimen. With advances in mini- mally invasive endoscopic surgery, surgeons should be aware of the potential complications, although the overall incidence of these complications is difficult to determine. Clearly, morcellating devices have the potential for great visceral injury, but the risks associated with dissemination of small tissue fragments are more difficult to delineate. Case repots of ectopic leiomyoma, endometriosis, adeno- myosis, ovarian tissue, and sarcoma exist in the literature, but the overall incidence is unknown. 18 In a case-control study comparing the rate of de novo endometriosis after hysterectomy with and without morcellation, the overall incidence was found to be 1% in both groups, suggesting no additional risk with morcellation. 6 Instances of atypical endometrial implants found years after supracervical hys- terectomy with benign pathology demonstrate the poten- tial capability of transformation into abnormal histology. 2 Even more rare are cases of leiomyomatosis peritonealis disseminata after morcellation of leiomyoma for benign indications, quoted at a rate of 0.1%. 19 The risk for disseminating occult cancerous cells with morcellation is low, but the morbidity of such an occur- rence is significant. The baseline rate of port-site metas- tases in laparoscopic gynecologic oncology surgery is 1% to 2%, which is similar to the incisional metastatic rate for equivalent exploratory laparotomy procedures. 20 The American College of Obstetricians and Gynecologists rec- ommends that adequate assessment of the endometrial cavity be performed before morcellation; however, this is not routinely done for asymptomatic women who are undergoing surgery for prolapse. 21,22 Endometrial biopsy is a sensitive and specific method for identifying endome- trial abnormalities, but there are no reliable diagnostic Figure 2. Gross specimen of endometrial implant sent to pa- thology. 3e2014.00403 CRSLS MIS Case Reports from SLS.org tests to identify rare instances of leiomyosarcoma (LMS).18,23 LMS is typically diagnosed after surgery for a presumed leiomyoma. The incidence of unexpectedly di- agnosing a leiomyoma variant, leiomyoma atypia, or a malignant sarcoma after morcellation is estimated at 0.09% to 1%. 18,19,24 The Society of Gynecologic Oncology re- leased a statement in December 2013 that morcellation is contraindicated in the presence of documented or sus- pected malignancy and may be inadvisable in premalig- nant conditions or risk-reducing surgery. 25 Morcellation of LMS increases the rate of abdominopelvic dissemination and adversely affects disease-free and overall survival. 23 In cases of occult sarcoma after morcellation, evidence of peritoneal dissemination was seen up to 64% of the time on subsequent evaluation. 19,24 Regardless of whether the tumor is removed intact or morcellated, the Society of Gynecologic Oncology statement emphasizes that LMS has an extremely poor prognosis. 25 Current power morcellation devices do not provide a fail-safe method for preventing intraperitoneal spread of morcellated tissue. Ultimately, the only way to avoid the spread of cells of the morcellated tissue would be to have an enclosed morcellation system. Some have suggested that specimens should be placed in endoscopic bags and brought up to the port site to be manually morcellated for removal. 18 This is a cumbersome technique that may not be feasible for large or partially calcified specimens. In- creased interest in developing devices to prevent spill during morcellation have been reported, such as power morcellation within a bag or special enclosed wire-mesh morcellators. 18,26 As of yet, these devices are experimen- tal, and until contained morcellation devices become available, precautions should be taken to prevent perito- neal spread or seeding of morcellated tissues. We suggest irrigation of the peritoneal cavity, along with careful in- spection through an additional port site to ensure that no fragments of tissue are missed. In this particular case, reevaluating the peritoneal cavity from the umbilical cen- tral port site might have identified retained tissue previ- ously unrecognized. As we grapple with the ramifications of morcellation in the rare cases of cancer, this case highlights the sequelae of morcellation even in benign disease. In concordance with the recommendations of the Society of Gynecologic Oncology and the American Col- lege of Obstetricians and Gynecologists, surgeons should have transparent discussions with their patients regarding risks, benefits, and alternatives of all procedures so that patients can make informed decisions in their medical care. 22,25

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