Incidental detection of endometriosis with 18 F-fluorodeoxyglucose positron emission tomography-computed tomography in a patient with cervical intraepithelial neoplasia and adenomyosis

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This case report details the incidental detection of endometriosis using <sup>18</sup>F-FDG PET-CT in a woman with cervical intraepithelial neoplasia and adenomyosis who presented with hematuria.

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This case report describes a 49-year-old woman with a history of treated cervical intraepithelial neoplasia III and hysterectomy for adenomyosis who later presented with abdominal discomfort and frank hematuria. Workup included cystoscopy and MRI showing a bladder wall–involving lesion that could not exclude malignancy, followed by contrast-enhanced CT and 18F-fluorodeoxyglucose PET/CT, which demonstrated FDG-avid disease in the right posterolateral bladder wall but no metabolic activity in small hypodense liver lesions. With differential diagnoses including endometriosis versus worse vaginal carcinoma, she underwent en bloc upper vaginectomy with posterior bladder wall resection and other staging procedures, and histopathology showed multiple endometriotic foci around the vaginal and bladder walls and in the ovaries. The main limitation is that conclusions are based on a single incidental case and the paper notes that PET/CT findings in endometriosis can vary, including with timing relative to menstrual cycle, and that noninvasive imaging cannot replace surgery for definitive diagnosis. This paper is centrally about endometriosis — it uses FDG PET/CT to incidentally detect surgically confirmed endometriotic foci involving the urinary bladder in a patient who also had adenomyosis.

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Abstract

Endometriosis is commonly associated with chronic pelvic pain and its presentation varies between individuals. The only way to confirm the presence of endometriosis is via keyhole or open surgery. In the presence of hematuria, deep endometriotic infiltration needs to be considered. We share an interesting case highlighting the role of 18F-fluorodeoxyglucose positron emission tomography-computed tomography in evaluating a posterior urinary bladder wall lesion and hypodense liver lesions in a middle-aged woman with presenting with frank hematuria in the background of treated cervical intraepithelial neoplasia and adenomyosis.
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Abstract

Endometriosis is commonly associated with chronic pelvic pain and its presentation varies between individuals. The only way to confirm the presence of endometriosis is via keyhole or open surgery. In the presence of hematuria, deep endometriotic infiltration needs to be considered. We share an interesting case highlighting the role of 18F-fluorodeoxyglucose positron emission tomography-computed tomography in evaluating a posterior urinary bladder wall lesion and hypodense liver lesions in a middle-aged woman with presenting with frank hematuria in the background of treated cervical intraepithelial neoplasia and adenomyosis.

Keywords

18F-Fluorodeoxyglucose adenomyosis cervical intraepithelial neoplasia endometriosis positron emission tomography-computed tomography urinary bladder A 49-year-old Chinese female with a history of treatment for cervical intraepithelial neoplasia (CIN) III underwent hysterectomy for dysfunctional uterine bleeding and hematuria in a private medical center in December 2017 which was later histopathologically confirmed to be adenomyosis with no residual CIN on the specimen. She remained well until September 2018 when she presented to our hospital with abdominal discomfort and frank hematuria. Cystoscopy showed a cystic lesion invading to the urinary bladder mucosa which of which histopathological examination reported mullerianosis, but malignancy could not be excluded. The CA-125 was elevated at 102 ng/mL. The magnetic resonance imaging performed to delineate the lesion showed irregular enhancing mass which could represent endometrial cyst [Figure 1a]. In view of history of CIN III and the possibility of Mullerian carcinoma, contrast-enhanced computed tomography (CT) of the abdomen and pelvis performed in October 2018 showed an irregular lobulated soft tissue mass measuring 2.6 cm × 3.6 cm at the right posterolateral aspect of the urinary bladder wall and hypodense liver lesions in segment VII (0.9 cm × 1.0 cm) and VIII (1.1 cm × 0.8 cm). 18F-Fluorodeoxyglucose (FDG) positron emission tomography (PET)/CT was performed to assess the significance of the lesions and to detect other possible metastases. The liver lesions were not metabolically active whereas the lesion at the right posterolateral wall of the urinary bladder was metabolically active [Figure 1b and c]. With the differential diagnosis of either endometriosis or worse-vaginal carcinoma, she underwent an en bloc upper vaginectomy with posterior bladder wall, bilateral salpingo-oophorectomy, omentectomy, and bilateral pelvic lymph node dissection. The histopathological examination of the surgical specimens reported multiple endometriotic foci at the ovaries and around the vaginal and bladder walls causing distortion. The actual prevalence of endometriosis worldwide is unknown as either key-hole or open surgery is required for definitive diagnosis. No noninvasive imaging modality is able to confirm endometriosis with enough accuracy to replace surgery.[1] A study by Eisenberg et al. reported that the point prevalence of endometriosis was 10.8 per 1000 and an average annual incidence rate of 7.2 per 10,000 women in child-bearing age.[2] Endometriosis is a condition where the endometrial tissue is present outside the uterine cavity and causes pelvic inflammation. Although 18F-FDG is sensitive in detecting inflammatory changes, Fastrez et al. did not observe any hypermetabolic activity in relation to endometriosis using 18F-FDG PET/CT in their study of 10 patients.[3] On the contrary, Jeffry et al. reported their case where endometriosis was detected by 18F-FDG PET/CT.[4] In the study by Setubal et al. on nine patients, PET scans were reported to have no value as a major tool to evaluate deep endometriosis or other forms of endometriotic disease.[5] However, it is important to note that endometriosis is affected by the cyclical hormones and 18F-FDG PET/CT studies were performed at variable periods of the affected women's menstrual cycles. The common sites of endometriosis include ovaries, fallopian tubes, pelvic peritoneum, and pelvic ligaments, whereas atypical sites include the gastrointestinal tract, urinary bladder, ureter, abdominal wall, and pleura.[6] Patients with endometriosis at the urinary bladder frequently present with urinary frequency and if longstanding may present with hematuria due to the deep endometriotic infiltration.[78] This is in keeping with our patient whose initial hematuria was overlooked. This case highlights the benefits of 18F-FDG PET/CT in our management planning, by excluding distant metastasis to the liver and identifying the nature of the soft tissue mass at the right posterolateral wall of the urinary bladder. Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.

References

- Imaging modalities for the non-invasive diagnosis of endometriosis. Cochrane Database Syst Rev. 2016;2:CD009591. - [Google Scholar] - Epidemiology of endometriosis: A large population-based database study from a healthcare provider with 2 million members. BJOG. 2018;125:55-62. - [Google Scholar] - Evaluation of 18FDG PET-CT in the diagnosis of endometriosis: A prospective study. Reprod Sci. 2011;18:540-4. - [Google Scholar] - Endometriosis with FDG uptake on PET. Eur J Obstet Gynecol Reprod Biol. 2004;117:236-9. - [Google Scholar] - Beyond the boundaries-endometriosis: Typical and atypical locations. Curr Probl Diagn Radiol. 2011;40:219-32. - [Google Scholar] - Various anatomic locations of surgically proven endometriosis: A single-center experience. Obstet Gynecol Sci. 2015;58:53-8. - [Google Scholar] - Endometrioid adenocarcinoma arising from deep infiltrating endometriosis involving the bladder: A case report and review of the literature. Gynecol Oncol Rep. 2015;13:68-70. - [Google Scholar]

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