Journal of Obstetrics and Gynaecology
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Endometrial stromal sarcoma in the abdominal wall arising from scar endometriosis T. A. Usta, S. E. Sonmez, A. Oztarhan & T. Karacan To cite this article: T. A. Usta, S. E. Sonmez, A. Oztarhan & T. Karacan (2014) Endometrial stromal sarcoma in the abdominal wall arising from scar endometriosis, Journal of Obstetrics and Gynaecology, 34:6, 541-541 To link to this article: http://dx.doi.org/10.3109/01443615.2014.914476
Published online: 15 May 2014.
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Endometrial stromal sarcoma in the abdominal wall arising from scar endometriosis T. A. Usta1, S. E. Sonmez2, A. Oztarhan3 & T. Karacan1 Departments of 1Obstetrics and Gynecology, Bagcilar Training and Research Hospital, 2Plastic and Reconstructive Surgery, Bezmialem Foundation University and 3Obstetrics and Gynecology, Sisli Etfal Training and Research Hospital, Istanbul, Turkey DOI: 10.3109/01443615.2014.914476 Correspondence: T. A. Usta, Department of Obstetrics and Gynaecology, Bagcilar Training and Research Hospital Bagcilar, 34200, Istanbul, Turkey. E-mail:
[email protected] Introduction Scar endometriosis (SE) is a rare entity reported in 0.03–1.08% of women following obstetric or gynaecological surgeries (Veda and Srinivasaiah 2010). Malignant transformation in endometriotic deposits was first described in the literature as early as 1925 (Sampson 1925). Endometriosis in association with malignancy is being reported with increasing frequency (Higashiura et al. 2012). A preoperative diagnosis is difficult, and in most of the cases, the diagnosis is confirmed after surgery. Endometrial stromal sarcoma (ESS) is the least common (⬍ 10%) type of uterine sarcoma, and furthermore, extrauterine endometrial stromal sarcoma is extremely rare (Higashiura
Figure 1. Wide excision of abdominal wall endometriosis with normal tissue around the margins (nodular, pigmented, non-mobile mass lesion was located on the left side of the anterior abdominal wall and close by, the previous Pfannenstiel incision scar).
et al. 2012). We hereby report a case of ESS of the abdominal wall arising from endometriosis in a caesarean section scar in a 41-yearold woman, and how we surgically managed this case.
Case report A 41-year-old woman presented with a mass lesion on the left side of a Pfannenstiel incision, that was painful during menstruation (Figure 1). The patient described that the lesion had gradually enlarged, causing exacerbating pain and tenderness during her menstruation periods. She had undergone a myomectomy operation 10 years ago and a caesarean section operation 19 years ago. On vaginal examination, her uterus was bigger, myomatous, irregular, fixed and immobile. An endometrioma of approximately 30 ⫻ 27 mm size was determined in transvaginal examination in the left ovary, concordant with multiple leiomyomata. Magnetic resonance imaging (MRI) demonstrated leiomyomata at uterine corpus and endometrioma at the ovary. An endometriotic mass lesion involving the skin and rectus abdominis muscle was also identified by MRI. Fine needle aspiration cytology (FNAC) performed on the mass lesion in subcutaneous tissue showed endometriosis. Based on the typical history, clinical and cytological features, a diagnosis of scar endometriosis with involvement of the abdominal wall was established. The parietal peritoneum and rectus abdominis muscle were infiltrated, and a 3 ⫻ 4 cm chocolate cyst was encountered intraoperatively on the left ovary, with multiple leiomyomata and diffuse adhesions. Wide surgical excision was performed removing a wide margin on all sides (Figure 1). Additionally, total abdominal hysterectomy and bilateral salpingo-oophorectomy were performed. Microscopic examination revealed low-grade endometrial stromal sarcoma at samples obtained from rectus muscles (Figure 2). Recovery was uneventful.
Discussion Cutaneous localisation of endometriosis is unusual and appears most frequently in surgical scars from obstetric or gynaecological interventions. In clinically doubtful cases, FNAC is one of the first diagnostic tools in the evaluation of tumours. Sometimes, the FNAC smears can be haemorrhagic, showing only a few macrophages and inflammatory cells, in which case the diagnosis of SE might be missed, as in our case. The mass lesion should be excised and evaluated histologically to rule out malignancy (Veda and Srinivasaiah 2010). Extragenital ESS is an extremely uncommon pathology with very few cases reported to date. Extragenital ESS is thought to occur more often in premenopausal women (Sofoudis et al. 2012). The ovary is the primary site of malignancy in 76% of the cases; however, 24% of the tumours arise from extragonadal sites (Lauslahti 1972; Brooks and Wheeler 1977). The origin of ESS has been explained by at least two hypotheses; first, is the malignant transformation of endometriosis, and second is the de novo tumour formation, potentially derived from Müllerian cells (Jung et al. 2009). The pluripotential Müllerian epithelium is considered to be widely distributed in the abdominal and pelvic cavities. Therefore, ESS may originate from endometriosis
Figure 2. (a) Endometrial glands (asterisk) with neoplastic cells suggestive of low-grade endometrial stromal sarcoma (arrow) (H&E, ⫻ 40). (b) Histopathological examination showing characteristically uniform oval and spindle-shaped cells, suggestive of low-grade endometrial stromal sarcoma (H&E, ⫻ 100).
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or from metaplasia of the pelvic Müllerian cells (Khan et al. 2002). Most cases reported in literature have appeared after caesarean sections. It is believed that scar endometriosis results from iatrogenic inoculation of endometrial cells into the fascia or subcutaneous tissues during invasive abdominopelvic procedures. Therefore, it is strongly recommended that before closure, the abdominal wound is thoroughly cleaned and irrigated vigorously with saline (Teng et al. 2008). There is insufficient information in the literature about the optimal management of uterine or extragenital ESS. As in other sarcomas, surgery is the most effective treatment for ESS. The efficacy of adjuvant therapy has not been proven (Puliyath and Nair 2012). In this paper, we have presented a rare case of malignancy in extragenital endometriosis. The case demonstrates that the endometrial stromal sarcoma might originate from a pre-existing endometriosis in the absence of any primary foci in the uterus or ovaries. Malignant transformation of endometriosis should be considered in the differential diagnosis of any new pelvic lesion in a patient with a history of endometriosis. Clinicians should keep in mind that ESS derived from scar endometriosis might be seen in the abdominal wall. To the best of our knowledge, this is the first case of endometrial stromal sarcoma arising from rectus abdominis muscle described in the literature. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Brooks JJ, Wheeler JE. 1977. Malignancy arising in extragonadal endometriosis: a case report and summary of the world literature. Cancer 40:3065–3073. Higashiura Y, Kajihara H, Shigetomi H et al. 2012. Identification of multiple pathways involved in the malignant transformation of endometriosis. Oncology Letters 4:3–9. Jung SI, Shin SS, Choi C et al. 2009. Endometrial stromal sarcoma presenting as prevesical mass mimicking urachal tumor. Journal of Korean Medical Science 24:529–531. Khan AW, Craig M, Jarmulowicz M et al. 2002. Liver tumours due to endometriosis and endometrial stromal sarcoma. HPB (Oxford) 4:43–45. Lauslahti K. 1972. Malignant external endometriosis. Acta Pathologica Microbiologica Scandinavica 223:98–102. Puliyath G, Nair MK. 2012. Endometrial stromal sarcoma: A review of the literature. Indian Journal of Medical and Paediatric Oncology 33:1–6. Sampson J. 1925. Endometrial carcinoma of the ovary, arising in endometrial tissue in that organ. Archives of Surgery 10:1–72. Sofoudis C, Kalampokas T, Grigoriadis C et al. 2012. Endometrial stromal sarcoma in a 29-year-old patient. Case report and review of the literature. European Journal of Gynaecological Oncology 33:328–330. Teng CC, Yang HM, Chen KF et al. 2008. Abdominal wall endometriosis: An overlooked but possibly preventable complication. Taiwanese Journal of Obstetrics and Gynecology 47:42–48. Veda P, Srinivasaiah M. 2010. Incisional endometriosis: diagnosed by fine needle aspiration cytology. Journal of Laboratory Physicians 2:117–120.
Recurrent urethro-vaginal fistula following urethral injury and repair at the time of transobturator tape sling insertion M. F. Aslam, J. Krashin & M. A. Denman From the Division of Urogynecology and Reconstructive Pelvic Surgery, Department of Obstetrics and Gynecology, Oregon Health and Science University, Portland, Oregon, USA DOI: 10.3109/01443615.2014.914477 Correspondence: M. F. Aslam, Department of Obstetrics and Gynecology, Mail Code L466, OHSU, SW Sam Jackson Park Road, Portland, Oregon 97239, USA. E-mail:
[email protected] Case report A 48-year-old patient was referred to us at the Department of Female Pelvic Medicine and Reconstructive Surgery at the University Hospital, due to continuous urinary leakage for the past 8 months. At that time (8 months earlier), she had a transobturator tape (TOT) sling inserted for stress incontinence of urine, that was not proven by preoperative urodynamic assessment, in another hospital. The urethra was injured at the time and repaired in a single layer with interrupted chromic catgut sutures. Check cystoscopy was carried out at the time. The bladder was drained with Foley’s catheter for 14 days afterwards. However, she suffered from uncontrollable leakage of urine following removal of the catheter. Urethro-vaginal fistula was confirmed on examination, with methylene blue test, as well as voiding cystourethrography. The patient was counselled about the findings and agreed to examination under anaesthesia, with a view towards excision of the tape and repair of the fistula. Initial cystoscopy revealed a normal bladder with transurethral erosion of clear mesh material at the level of the bladder neck and a 1 cm urethral defect distal to this. She underwent transvaginal excision of the tape at the level of the bladder neck and repair of the urethro-vaginal fistula. The repair necessitated re-attachment of the inferior aspect of the urethra to the bladder base following separation of these structures with mesh removal. The defect was repaired in a two-layer fashion with 4-0 Vicryl suture. A total of 300 ml of diluted methylene blue was instilled into the bladder, confirming watertight repair with no leak seen after catheter removal and manual occlusion of the external urethral meatus. A bulbocavernosus fat pad (Martius flap) was developed and brought into the field to cover the defect. A 2 ⫻ 3 cm piece of small intestine submucosa (SIS) graft was placed, as there was limited tissue to bring into the suture area for reinforcement. The graft was used to bridge the vaginal mucosa. The patient was discharged home with a transurethral Foley’s catheter. She was advised to stop smoking to facilitate wound healing. The Foley’s catheter was removed 3 weeks postoperatively, following a voiding cystourethrogram demonstrating no recurrent fistula. She did well for 4 weeks and then reported a return of large volume leakage. Examination demonstrated recurrent fistula at the bladder neck. The patient was counselled regarding the findings and underwent a second transvaginal, multilayered repair of a 3 cm bladder neck fistula, with the same techniques as used previously. Increased tissue mobilisation was achieved. Tissue mobilisation was achieved for at least 2 cm circumferentially around the fistula. The fistula edges were excised prior to closure. An additional midurethral approximation of the lateral edges of the pubococcygeus muscle was performed to facilitate bladder neck support, following watertight repair. A transurethral Foley’s catheter was again placed for 3 weeks and removed following a voiding cystourethrogram demonstrating no recurrence of the fistula. Postoperatively, she developed stress urinary incontinence, which responded to a transurethral bulking agent (Macroplastique®, Uroplasty) 4 months following her second repair. She was seen in follow-up 1 month following injection, with no complaints of urinary leakage.
Conclusion Synthetic midurethral slings are commonly performed for stress urinary incontinence. Recent studies have shown the transobturator method has fewer bladder complications or other adverse events compared with the retropubic approach, concluding that the transobturator approach is safe and effective for the treatment of stress incontinence due to fewer complications (Cheng and Liu 2012). The risk of urethral injury remains, regardless of approach. Fistula formation however, is exceedingly rare. Of the reported cases of urogenital fistula after synthetic slings for incontinence, none relate to the placement of tape after urethral injury, which is unique in our case (Morton and Hilton 2009). Our presented case combines two uncommon long-term complications: urethral mesh erosion and recurrent fistula. We posit the unusual combination is related to the urethral injury, repair and placement of foreign body over the repair site. Increased inflammation in a single layer repair