Journal of Obstetrics and Gynaecology, 2015; Early Online: 1–2 © 2015 Informa UK, Ltd. ISSN 0144-3615 print/ISSN 1364-6893 online

CASE REPORT

Polypoid endometriosis presenting as a mass at the pouch of Douglas N. S. Gezer1, M. Seçil1, E. Ç. Ulukuş2 & M. Celiloğlu3

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1Department of Radiology, Faculty of Medicine, Dokuz Eylul University, Izmir, Turkey, 2Department of Pathology, Faculty of Medicine, Dokuz Eylul University, Izmir, Turkey, and 3Department of Obstetrics and Gyneacology, Faculty of Medicine, Dokuz Eylul University, Izmir, Turkey

DOI: 10.3109/01443615.2015.1014327 Correspondence: Dr Naciye Sinem Gezer, Department of Radiology, Faculty of Medicine, Dokuz Eylul University Hospital, Mithatpasa Cad. 35340 Inciralti-Izmir, Turkey. E-mail: [email protected]

Introduction Polypoid endometriosis is a rare variant endometriosis that may simulate malignant tumours on clinical and intra-operative examinations and forms large and often multiple polypoid masses with the appearance of endometrial polyps (Medeiros et al. 2011). The disease may involve various pelvic and peritoneal sites (Parker et al. 2004). The rectosigmoid colon has been reported to be the most frequent site of involvement followed by the ovary (Parker et al. 2004). The literature on magnetic resonance imaging (MRI) characteristics of polypoid endometriosis is scarce and confined to a few case reports. To our knowledge, only seven cases of polypoid endometriosis have been reported previously in the English literature using MRI findings.

Case report A 25-year-old woman who had given birth before 10 months was referred to our hospital with an incidentally detected left adnexal

mass. On physical examination, no abdominal tenderness was present. Laboratory examination revealed a slight elevation of the serum CA-125 level – 86.6 (normal, ⬍ 16.3) IU/mL. Ultrasonography of the abdomen detected a 10-cm, multiloculated cystic pelvic mass with solid components at the pouch of Douglas. The uterus and ovaries were morphologically normal, although they were adjacent to the large mass. The patient was further evaluated by MRI of the pelvis that showed a 10 ⫻ 11⫻ 13 cm multicystic mass in the pouch of Douglas. Cystic components of the mass were hyperintense on T2-weighted (T2W) and mostly hypointense on T1-weighted (T1W) images (Figure 1a and b). A few of the cysts were slightly hyperintense on T1W images, and one cyst showed a fluid–fluid level on T2W images. Heterogeneously hyperintense solid areas with irregular polypoid extensions and small high-intensity spots within solid components were also detected on T2-weighted images. There were hypointense rim-like structures surrounding the mass on T2W images (Figure 1b). On fat-saturated post-contrast T1W images, both the solid components and surrounding rim-like structures showed intense enhancement similar to that of the uterine endometrium (Figure 1b). There were no detectable distant metastases, ascites or enlarged lymph nodes. Based on the imaging and clinical findings, the patient underwent surgery with a pre-operative diagnosis of a pelvic malignancy of unknown origin. During surgery, there were no ascites in the pelvic cavity, no evidence of a peritoneal metastatic lesion or lymphadenomegaly, and the ovaries and uterus were found to be normal and retained. Gross and microscopic examination findings of the specimen led to the diagnosis of polypoid endometriosis.

Discussion The pre-operative radiological differential diagnosis of a multiloculated solid-cystic pelvic mass in such a case should include ovarian and extra-ovarian neoplasms. Based on serum CA-125 elevation and

Figure 1. Axial T1W (a) and T2W (b) images show a multiloculated, cystic, pelvic mass with solid components at the pouch of Douglas. A few of the cysts demonstrate slight hyperintensity on T1W imaging and marked hyperintensity on fat-saturated T1W imaging (arrow heads, a, c). Both the solid and cystic components are surrounded by hypointense rim-like structures shown on T2W imaging (arrows, b). The solid components have irregular polypoid extensions (dashed arrows, b). On fat-saturated T1W imaging with gadolinium, both the solid components and surrounding rim-like structures demonstrate intense enhancement (arrows, d).

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the imaging findings, surface epithelial ovarian tumours, including serous or mucinous cystadenocarcinomas and endometrioid adenocarcinoma, might be suspected. However, the involvement of extraovarian sites other than the ovary itself might suggest primary peritoneal tumours, particularly serous carcinoma. The solid components within the mass reduced the possibility of multicystic mesothelioma. Pseudomyxoma peritonei should also be encountered in the differential diagnosis of multicystic pelvic tumours but was excluded because there were no peritoneal implants or ascites. The differential diagnosis of a solid-cystic mass with papillary projections of unknown origin should also include primary fallopian tube carcinoma. Nevertheless, associated findings such as peritumoural ascites, intrauterine fluid collection or hydrosalpinx did not exist. The MRI findings of our patient were correlated with the characteristics described in the literature. The appearance of a mass with a cystic component, containing heterogeneous hyperintense polypoidal solid areas on T2W images that homogenously enhance on contrast study, as in our case, has previously been described as a diagnostic clue (Kozawa et al. 2012; Ozaki et al. 2008; Takeuchi et al. 2008; Lacroix-Triki et al. 2004). It has been reported that T2 hyperintensity of endometrial polyps similar to that of the endometrium was probably based on their internal structures, such as endometrial glands, stroma, fibrous core and cysts (Ozaki et al. 2008; Takeuchi et al. 2008). In the present case, hypointense rim-like structures surrounding the mass on T2W images were also described as in previous case reports. Takeuchi et al. have demonstrated that linear areas of hypointensity surrounding the polypoid mass on T2W images that showed intense contrast enhancement, similar to the adjacent uterus on post-contrast T1W images, were fibrous tissue associated with endometriosis. Furthermore, Kozawa et al. reported hypointense rim and linear areas surrounding the hyperintense solid component on T2W images and suggested that these findings could be a diagnostic clue to this rare entity (Kozawa et al. 2012). The present case demonstrates that polypoid endometriosis presenting as multicystic large mass with solid polypoid components may mimic malignancy with imaging characteristics. Clinical features such as elevation of CA-125 level may lead to confusion because non-tumoural conditions such as endometriosis can also be associ-

ated with increased CA-125 levels. The MRI characteristics of the mass, such as containing heterogeneous hyperintense polypoidal solid areas on T2W images and hypointense rim-like structures surrounding the mass, may suggest polypoid endometriosis in the differential diagnosis. To our knowledge, no report exists concerning polypoid endometriosis of the pouch of Douglas in the English literature.

Conclusion Polypoid endometriosis may simulate malignant tumours with imaging findings as well as the clinical features. However, it should be considered in the differential diagnosis of a mixed solid-cystic mass at the pouch of Douglas when other signs of malignancy such as metastases, ascites and enlarged lymph nodes are not present. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Kozawa E, Inoue K, Iwasa N et al. 2012. MR imaging of polypoid endometriosis of the ovary. Magn Reson Med Sci 11:201–204. Lacroix-Triki M, Beyris L, Martel P et al. 2004. Low-grade endometrial stromal sarcoma arising from sciatic nerve endometriosis. Obstetrics and Gynecology 104:1147–1149. Medeiros F, Wang X, Araujo AR et al. 2012. HMGA gene rearrangement is a recurrent somatic alteration in polypoid endometriosis. Human Pathology 43:1243–1248. Ozaki K, Gabata T, Tanaka M et al. 2008. Polypoid endometriosis: An uncommon and distinctive variant of endometriosis. European Journal of Radiology Extra 65:97–100. Parker RL, Dadmanesh F, Young RH et al. 2004. Polypoid endometriosis: a clinicopathologic analysis of 24 cases and a review of the literature. The American Journal of Surgical Pathology 28:285–297. Takeuchi M, Matsuzaki K, Furumoto H et al. 2008. Case report: A case of polypoid endometriosis: MR pathological correlation. The British Journal of Radiology 81:118–119.

Polypoid endometriosis presenting as a mass at the pouch of Douglas.

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