British Journal of Radiology (2007) 80, 878-883
© 2007 British Institute of Radiology
doi: 10.1259/bjr/16282081
MR findings of pseudoneoplastic lesions in the uterine cervix mimicking adenoma malignum
K Sugiyama, MD
1 and
Y Takehara, MD, PhD
2
1 Department of Radiology, Seirei Numazu Hospital, 902-6 Shichitanda Matsushita Hon-aza, Numazu, Shizuoka, 410-8555 and 2 Department of Radiology, Hamamatsu University School of Medicine, 1-20-1 Handayama, Hamamatsu, Shizuoka, 431-3192, Japan
Correspondence: K Sugiyama, Department of Radiology, Seirei Numazu Hospital, 902-6 Shichitanda Matsushita Hon-aza, Numazu, Shizuoka, 410-8555 Japan. E-mail: koichiss37{at}yahoo.co.jp
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Abstract
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Pseudoneoplastic glandular lesions are benign lesions that are often histologically and radiographically confused with adenoma malignum, which is a rare variant of mucinous adenocarcinoma of the uterine cervix. 15 cases of pseudoneoplastic glandular lesions, characterized by multilocular cystic masses in the uterine cervix, were investigated; these lesions included endocervical glandular hyperplasia, deep nabothian cysts and tunnel cluster. All lesions were proven by conization or hysterectomy. The MR findings correlated closely to the histopathological specimens. As a result, MR findings of pseudoneoplastic glandular lesions were almost identical to those of adenoma malignum; hence, it was almost impossible to differentiate between the two pathologies. Radiologists should be aware that these two conditions are quite similar in morphology.
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Introduction
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Adenoma malignum, which is also called "minimal deviation adenocarcinoma", is known to be a rare variant of well-differentiated mucinous adenocarcinoma of the uterine cervix, which is characterized by multilocular cystic lesions extending from the endocervical glands to the deep cervical stroma. The characteristic MR findings of adenoma malignum have been reported to be very useful in its early diagnosis. However, in recent years, there have been many reports describing pseudoneoplastic glandular lesions, which are benign lesions that are often histologically and radiographically confused with adenoma malignum [1]. We assessed the MR findings of pseudoneoplastic lesions in close correlation with histopathological specimens.
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Methods and patients
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We retrospectively reviewed the MR findings of 15 patients with pseudoneoplastic glandular lesions characterized by a multilocular cystic mass in the uterine cervix revealed on pelvic MR images. Any patients with unilocular uncomplicated cystic lesions and obvious solid lesions were excluded from this study. All patients underwent an MRI study at our institutions between 1998 and 2004. The age of the patients ranged from 34 to 76 years (mean 50.7 years).
All lesions were histopathologically proven either by conization (eight patients) or hysterectomy (seven patients). The lesions included endocervical glandular hyperplasia (10 patients; Figure 1
), deep nabothian cysts (four patients; Figure 2) and tunnel cluster (one patient; Figure 3). Two patients with endocervical glandular hyperplasia were associated with dysplasia. Four patients with endocervical glandular hyperplasia and two patients with nabothian cysts were associated with inflammatory areas either on the cyst walls or in the interstitial compartments among the cysts.
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Figure 1. Endocervical glandular hyperplasia seen in a 54-year-old woman with lower abdominal pain, histopathologically proven by conization. (a) Fat-suppressed T1 weighted spin echo axial image depicts multiple small cysts in the enlarged uterine cervix extending into deep cervical stroma. (b) A photomicrograph shows endocervical glandular hyperplasia without cellular atypia. (original magnification x10; haematoxylin & eosin stain)
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Figure 2. A 76-year-old woman with malignant ovarian tumour. Deep nabothian cysts were found incidentally in the hysterectomy specimen. (a) T2 weighted spin echo sagittal image shows the multilocular cystic lesions in the enlarged uterine cervix. (b) Fat-suppressed gadolinium-enhanced T1 weighted spin echo sagittal image depicts multiple cystic lesions within the cervical stroma. The cyst walls are somewhat thickened and slightly enhanced. (c) A photomicrograph shows multiple mucin-containing cysts composed of a single layer of columnar cells (original magnification x10; haematoxylin & eosin stain).
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Figure 3. Tunnel cluster seen in a 57-year-old woman complaining of a large amount of watery discharge (histopathologically proven by conization). (a) T2 weighted spin echo axial image shows multilocular cystic lesions in the uterine cervix. (b) A photomicrograph shows lobulated aggregates of dilated endocervical glands filled with mucin. Cellular atypia is not observed. The destructive areas on the cyst walls are partially seen where mucinous fluid leakage occurs (original magnification x10; haematoxylin & eosin stain).
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The clinical features included watery discharge (two patients), abnormal genital bleeding (three patients), lower abdominal pain (two patients) and lower abdominal discomfort (two patients); six patients were symptom free. No patient showed an elevated serum level of tumour markers (CA19-9 or CA125). In nine patients, the lesions were incidentally found during MR imaging performed for other symptomatic or asymptomatic disorders, including adenocarcinoma of the uterine corpus, huge ovarian tumours and large uterine myomas.
All MR examinations were obtained on a 1.5 T or 1.0 T MR scanner with a phased array torso coil (Signa Horizon; GE Medical Systems, Waukesha, WI).
The MR examination included sagittal and axial fast spin-echo T2 weighted sequences with or without fat-suppression technique (TR range/TE range: 3200–8000/80–96 in all patients), and T1 weighted images employing spin echo (TR/TE: 600/10 in six patients) or spoiled gradient echo in the steady state (TR range/TE range: 160–220/1.9–4.2 in nine patients). In 14 patients, either gadolinium diethylenetriaminepentaacetic acid (Gd-DTPA) (Magnevist; Nihon Schering, Osaka, Japan) or Gd-DTPA bismethylamide (Gd-DTPA-BMA) (Omniscan; Daiichi Pharma, Tokyo, Japan) was administered intravenously (0.1 mmol kg–1); thereafter, contrast-enhanced T1 weighted images, with or without the use of the fat-suppression technique, were obtained. The slice thickness of axial and sagittal sections was 5–8 mm with an interslice gap of 1.5–2.0 mm. The field of view was 22–26x22–32 cm, and the image matrix was 256–384x128–256.
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Results
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The imaging findings, surgical treatments and our original diagnosis for 15 patients are summarized in Table 1. Multilocular cystic lesions extending from the endocervical gland to the deep cervical stroma were demonstrated. The diameters of the individual cysts ranged from 1–20 mm. On T1 weighted MR images, 14 of the lesions were either hypointense or isointense to the cervical stroma, of which 8 lesions were partially hyperintense. Only one lesion was entirely hyperintense. On T2 weighted images, 14 lesions were entirely hyperintense, whereas 1 lesion was partially isointense with a hyperintense background. On gadolinium-enhanced T1 weighted images performed in 14 patients, the walls of the cysts were thin and smooth, and were slightly enhanced; however, 8 lesions had partially thickened walls and 4 lesions showed a nodular enhancement of the stroma between cysts. Our original MR diagnosis when we encountered such cystic masses in the uterine cervix was almost always a "multilocular cystic mass in the uterine cervix — with adenoma malignum not ruled out" in order to retain the possibility of malignancy, even though we considered that they might be benign entities. Adenoma malignum was also suspected in two patients suffering from watery discharge.
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Table 1. Clinical, radiological, surgical and histopathological findings in patients with pseudoneoplastic lesions in the uterine cervix
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Discussion
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Adenoma malignum is a subtype of mucinous adenocarcinoma of the uterine cervix, which represents about 3% of adenocarcinoma cases and about 0.15%–0.45% of all cervical carcinomas of the uterus in the literature [2, 3]. It is composed of histologically well-differentiated features; therefore, to make a correct diagnosis, it is necessary to find a characteristic pathological feature such as multiple irregular lobulations of distorted glands demonstrating a "hair-pin" shape (Figure 4). Despite the presence of well-differentiated histopathological features, the prognosis of adenoma malignum is known to be poor because of early dissemination into the peritoneal cavity and early distant metastasis. Accordingly, the early differentiation of adenoma malignum from benign conditions is critical for all gynaecological clinicians. However, the histopathological features of adenoma malignum are occasionally similar to pseudoneoplastic glandular lesions, so that the differentiation between the two pathologies is still difficult even for pathologists.
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Figure 4. Adenoma malignum seen in a 49-year-old woman with a large amount of watery discharge (histopathologically proven from a hysterectomy specimen; shown as a reference case). (a) Fat-suppressed T2 weighted spin echo axial image depicts multilocular cystic lesions extending into deep cervical stroma with perifocal oedema. (b) Fat-suppressed gadolinium-enhanced T1 weighted sagittal image depicts multilocular cystic mass. The cyst walls are slightly thickened, and nodularly enhanced areas are shown among the cysts. (c) A photomicrograph shows multiple cystic lesions composed of a single layer of columnar cells that resemble normal endocervical glands. However, most glands have cellular atypia and structural dysplasia with multiple lobulations demonstrating a "hair-pin" shape. In another specimen (not shown), mucinous fluid leakage into the cervical stroma is also seen. Taking all of the above findings into consideration, adenoma malignum is thus considered to be the most likely histopathological diagnosis (original magnification x10; haematoxylin & eosin stain).
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The typical MR findings of adenoma malignum have been reported to consist of a multilocular cystic mass, demonstrating very high-signal intensity on T2 weighted images and either isointensity or slight hyperintensity on T1 weighted images, which extended from the endocervical glands to the deep cervical stroma, with solid components enhanced by contrast media [2, 3]. These findings were thus considered to be an early sign for adenoma malignum. However, in recent years, there have been many reports investigating pseudoneoplastic glandular lesions, which are benign lesions that are often confused with adenoma malignum, either histologically or radiographically [1, 4–6]. Young and Clement [1] reviewed the reports on pseudoneoplastic lesions including papillary endocervicitis, tunnel cluster, deep endocervical glands, deep nabothian cysts, microglandular hyperplasia, mesonephric hyperplasia, diffuse laminar endocervical glandular hyperplasia, glandular hyperplasia not otherwise specified, metaplasias, endometriosis, Atrias-Stella reaction, changes secondary to extravasation of mucin, and infectious and reactive atypias. Our 15 patients with pseudoneoplastic lesions included those with endocervical hyperplasia, deep nabothian cysts and tunnel cluster, appearing as multilocular cystic masses. In nine patients, intracystic fluid, which either partially or entirely showed as hyperintense to adjacent uterine stroma on T1 weighted images, seemed to reflect various degrees of mucin concentration. In six patients, the slightly hyperintense area, which was shown in the cervical stroma surrounding the cystic lesions on T2 weighted images, seemed to be caused by the presence of oedema resulting from either inflammation or a rupture of the expansile cysts. In all patients, the walls of the cysts were mostly thin, smooth and slightly enhanced on gadolinium-enhanced T1 weighted images; however, some portions were partially thickened or accompanied by solid nodular components with slightly stronger enhancement, which seemed to reflect inflammatory processes in the cervical stroma or congestion of the small vessels. Otherwise, the observed small nodular enhancement among the cysts might have been an artefact from the partial volume effect of overlapping thin cyst walls, which occurs because of the limited spatial resolution of MR imaging. Consequently, the MR findings of pseudoneoplastic lesions were almost identical to those of adenoma malignum reported previously in the literature, which suggested that the presence of cellular atypia or structural dysplasia in the pseudoneoplastic lesions did not significantly change MR findings.
A large amount of watery discharge is known to be a chief clinical symptom observed in adenoma malignum [2, 3]. However, the watery discharge was also the main complaint of our patients with tunnel cluster and nabothian cysts with inflammation. Many previous reports have also described how watery discharge is not a unique complaint observed in adenoma malignum [7–9]. Occasionally, adenoma malignum was reported to be found incidentally in the hysterectomy specimen obtained during the investigation for other pathological disorders, such as uterine adenocarcinoma or malignant ovarian tumour [10, 11].
In recent years, case reports concerning the utility of the monoclonal antibody HIK-1083 (Kanto Chemical, Tokyo, Japan) against murine mucin of pyloric glands in the diagnosis of adenoma malignum have been occasionally produced [12]. This unique monoclonal antibody has been indicated to be useful for the correct diagnosis of adenoma malignum because of its specific action against the glands in tissue specimens from this condition. However, some case reports disclosed that endocervical glandular hyperplasia with pyloric gland metaplasia had also been positive for the antigen detected by HIK-1083 examination [4].
In summary, the MR findings of pseudoneoplastic glandular lesions were almost identical to those of adenoma malignum, and differentiation between adenoma malignum and pseudoneoplastic lesions was not simple. Radiologists should be aware of these two conditions, particularly their similarities in morphology but differences in required management. Adenoma malignum is generally rare, whereas benign cystic lesions in the uterine cervix are much more common; therefore, most radiologists tend to diagnose a benign abnormality when faced with a multilocular cystic mass in the uterine cervix. We must be cautious not to be too confident when making a diagnosis, even if characteristic findings of adenoma malignum are observed on MR imaging. However, in contrast, we must carefully recommend either conization or a hysterectomy to clinicians when the patient is still suspected to have a malignant abnormality owing to symptoms of a continuous watery discharge.
Received for publication August 4, 2006.
Revision received December 6, 2006.
Accepted for publication January 30, 2007.
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References
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Abstract
Introduction
