British Journal of Radiology (2009) 82, e20-e22
© 2009 British Institute of Radiology
doi: 10.1259/bjr/75304693
British Journal of Radiology 82 (2009),e20-e22 ©2009 The British Institute of Radiology
Clear cell adenocarcinoma of the uterine cervix arising from a background of cervical endometriosis
T HIROMURA, MD,
Y O TANAKA, MD,
T NISHIOKA, MD,
M SATOH, MD and
K TOMITA, MD
Department of Radiology, NTT East Corp. Sapporo Hospital, Sapporo 060-0061, 2Department of Radiology, Graduate School of Comprehensive Human Sciences, University of Tsukuba, Tsukuba, Ibaraki 305-8575, 3Division of Radiological Technology, Department of Health Sciences, University of Hokkaido, Sapporo 060-0812, 4Department of Pathology, NTT East Corp. Sapporo Hospital, Sapporo 060-0061 and 5Department of Internal Medicine, NTT East Corp. Sapporo Hospital, Sapporo 060-0061, Japan
Correspondence: Tadao Hiromura, Department of Radiology, NTT East Corp. Sapporo Hospital, Sapporo 060-0061, Japan. E-mail: thrmr{at}amber.plala.or.jp
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Abstract
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The radiological findings of cervical clear cell adenocarcinoma (CCA) have not been described previously. Here, we present MR findings of this neoplasm that included mixed solid and cystic components with eccentric solid components. These are similar to the MR features of ovarian CCA. Endometriosis was also noted in the uterine cervix. Coexistence of CCA and endometriosis at the cervix suggests that the pathogenesis may be similar to that of ovarian CCA.
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Introduction
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Although most cervical carcinomas are squamous, a significant number are adenocarcinomas. Adenocarcinomas are histologically categorized into mucinous, endometrioid, clear cell, serous and mesonephric subtypes [1]. Clear cell adenocarcinoma (CCA) of the uterine cervix is rare and its radiological features have not been reported to date. Equally unusual is endometriosis of the cervix. We present a case of cervical CCA with coexisting cervical endometriosis. Ovarian CCA can develop from endometriosis. Our case suggests that the CCA arose from a background of cervical endometriosis; this case is the first report of its kind in either the pathology or radiology literatures.
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Case report
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A 57-year-old woman with vaginal bleeding was referred to our hospital. CT (LightSpeed Ultra (an 8-row detector); GE Medical Systems, Milwaukee, WI) and MRI (Signa Excite HD (1.5T system); GE Medical Systems) studies were performed. Post-contrast CT showed a well-enhanced nodular mass measuring 60 x 48 mm at its maximal diameter at the uterine cervix. The mass contained several cystic spaces. MRI revealed multiple nodular masses at the uterine cervix extending to the pouch of Douglas (Figure 1a
). Most of the lesions were solid, but some were cystic (Figure 1b,c). The cystic lesions had eccentrically grown solid components (Figure 1a). The solid lesions exhibited heterogeneous high-signal intensity on T2 weighted images (Figure 1a,c) and were moderately enhanced with meglumine gadopentetate (Figure 1d). The solid components contained several minute cysts, which produced the heterogeneous signal intensity on T2 weighted and enhanced T1 weighted images (Figure 1e). The endocervix appeared intact on T2 weighted and enhanced T1 weighted images. Tumour markers including carbohydrate antigen 19-9, carcinoembryonic antigen and cancer antigen 125 were not elevated. Papanicolaou smear cytology was Class I at the endocervix and endometrium, but Class V at the vagina. Under a clinical diagnosis of cervical cancer, a total abdominal hysterectomy and pelvic lymphadenectomy were performed. The tumour was located in the cervix without extracervical invasion. Pathological examination revealed neither lymph node metastases nor invasion to the cervical canal, endometrium or ovary. Some of the cysts contained haemorrhagic fluid. Histologically, the tumour cells were characterized by clear cytoplasm and a hobnail-like appearance, which led to the diagnosis of CCA. The cyst walls were lined with tumour cells. There were several minute 1.0 mm cavities lined with ectopic endometrial glands, together with haemosiderin deposition close to the tumour. These findings were consistent with endometriosis (Figure 1f). The patient is still being followed without recurrence.
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Figure 1. A 57-year-old woman with clear cell adenocarcinoma (CCA) of the uterine cervix. (a) Sagittal T2 weighted MR image showing multiple round masses extending to the pouch of Douglas. The cystic masses have solid components (black arrow), with an eccentric growth pattern that is often observed in ovarian CCA. (b) Axial fat-suppressed, T1 weighted MR image showing a hyperintense cyst (white arrow), suggesting a haemorrhagic cyst. (c) Axial T2 weighted MR image showing several well-defined round solid masses and a few cystic spaces at the cervix. The solid components are heterogeneously hyperintense. (d) Axial fat-suppressed post-gadolinium T1 weighted image showing moderate enhancement of the solid masses. (e) Near the upper margin of (d), the solid components have minute cystic portions (white arrow). (f) Photomicrograph showing tumour cells clearly margined with connective tissue. There is a cavity measuring 1.0 mm lined by endometrial glands (black arrow), indicating the presence of endometriosis. The scale bar represents 1.0 mm.
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Discussion
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The aetiology and pathogenesis of cervical CCA are unclear. Exposure to diethylstilbestrol in utero is one cause of CCA in younger patients [2]. CCA most commonly arises from the ovary, and is closely associated with ovarian endometriosis [3]. Whether cervical CCA is related to endometriosis has not been addressed. Our case of CCA was associated with cervical endometriosis, suggesting that the pathogenesis of cervical CCA may be similar to that of ovarian CCA.
Cervical endometriosis accounts for only 3% of all cases of endometriosis [4]. Several theories exist for how endometriosis arises, and it is likely that the pathogenesis may vary by location. Superficial cervical endometriosis has a strong predilection for sites of obstetrical and operative trauma caused by dilatation and curettage, biopsy, cautery, abortion and vaginal delivery. Therefore, implantation of ectopic endometrial tissue is a probable explanation for superficial endometriosis [5]. Pelvic endometriosis often involves the cul-de-sac adjacent to the cervix, and may be another origin of cervical endometriosis. Coelomic metaplasia and vascular dissemination are two other theories for the pathogenesis of cervical endometriosis. The cause of cervical endometriosis in our case was not clear, as the patient had no history of cervical trauma, adenomyosis or pelvic endometriosis.
The radiological features of cervical endometriosis have not been reported. A haemorrhagic cyst is one imaging feature of ovarian endometriosis in women of reproductive age. Our case showed signal intensities on MRI consistent with haemorrhagic cysts; however, these were unremarkable compared with those seen in ovarian endometriosis. As our patient was post-menopausal, menstrual haemorrhage into the endometrial cyst did not occur as evidenced by the lack of the characteristic signal intensity of haemorrhage on MRI. Histopathological examination confirmed that these structures were endometrial cysts. The inner surfaces of the cyst walls were lined with tumour cells. The intracystic haemorrhage resulted from tumour bleeding. Other MRI features of cervical endometriosis could not be evaluated because the tumour replaced the cervix entirely.
We compared our MR findings with those of ovarian CCA, which have been described in detail. The MRI features of ovarian CCA are as follows [6]: (i) uni- or multilocular cystic, or nearly solid, masses; (ii) one or a few solid components at the cyst wall, which are round or papillary, and present an eccentric growth pattern; and (iii) several cystic portions within the solid components. Most of these features were seen in our case, with some exceptions. Ovarian CCA is usually cystic, whereas the tumour in our case was predominantly solid. This may be explained by the limitation of endometrial cyst growth in the cervix. Ovarian CCA usually appears within well-developed large endometrial cysts, and has a cystic appearance when detected at an early stage [7]. In the cervix, the fibrous connective tissue surrounding the cyst restricts its growth. Thus, the solid components within the cyst are forced to occupy the lumen. This results in a solid mass encapsulated by the cyst wall.
The differential diagnosis for cystic masses of the cervix encompasses several diseases in addition to endometriosis. Two cases of well-differentiated mucinous adenocarcinoma (endocervical type) with cystic components have been reported [8]. One showed a unilocular cystic lesion with an eccentrically grown papillary protrusion. The other showed a multicystic lesion with enhanced thick septa on MRI. The former resembled our tumour in shape, but differed in that it was unilocular. The latter was consistent with adenoma malignum [9]. Its appearance was so different from our tumour, however, that we excluded it from the differential.
The clinical behaviour of cervical CCA has long been thought to be aggressive and its prognosis generally worse than that of squamous cell carcinomas or non-clear cell adenocarcinomas [10, 11]. However, a recent study revealed that, although CCAs had a slightly worse 5-year survival rate than other cervical carcinomas, the prognosis was equivalent once adjustments had been made for the stage [12]. Therefore, early-stage CCAs, like other early-stage cervical carcinomas, carry a favourable prognosis. A notable diagnostic difference between CCA and other cervical carcinomas is that most cervical CCAs are endophytic and tend to show deep infiltration of the cervix. Furthermore, they extend to the uterine corpus more often than other cervical carcinomas [12]. Given this, a timely and accurate diagnosis is key to its successful management. Additionally, correctly identifying tumour extension is crucial for surgical planning.
In conclusion, we describe a case of uterine cervical CCA associated with cervical endometriosis. The MRI features included well-defined, round, solid and cystic masses, with an eccentric growth pattern of solid components. These findings are similar to those seen in ovarian CCA.
Received for publication September 18, 2007.
Revision received November 26, 2007.
Accepted for publication November 30, 2007.
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Abstract
Introduction
