British Journal of Radiology (2005) 78, 558-561
© 2005 British Institute of Radiology
doi: 10.1259/bjr/82283833
Cystic adenomyosis with florid glandular differentiation mimicking ovarian malignancy
I Imaoka, MD
1
Y Kaji, MD
1,2
Y Kobashi, MD
3
A Wada, MD
1
G Honjo, MD
3
M Hayashi, MD
4
M Yoshida, MD
4 and
M Matsuo, MD
1
1 Department of Radiology, MR Division, Tenri Hospital, 200 Mishima, Tenri, 632-8552, 2 Department of Radiology, Kobe University School of Medicine, 7-5-2 Kusunoki, Chuo-ku, Kobe, 650-0017, 3 Department of Pathology, Tenri Hospital, 200 Mishima, Tenri, 632-8552 and 4 Department of Obstetrics and Gynaecology, Tenri Hospital, 200 Mishima, Tenri, 632-8552, Japan
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Abstract
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We report a case of cystic adenomyosis, presenting as a huge exophytic cystic mass with florid glandular differentiation. MR findings of the mass mimicked ovarian carcinoma associated with endometriosis. The presence of signal voids bridging the uterus and tumour should suggest a mass of uterine origin. Hyperintense protuberance in a hypointense loculus on T2 weighted images may suggest benign disease. However, surgical exploration and resection is still required to exclude an ovarian malignancy.
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Introduction
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Adenomyosis is a common gynaecological disorder with a reported incidence ranging from 5% to 70%, with a mean of 20–30% [1]. It is characterized as ectopic endometrial glands and stroma extending into the myometrium. MRI shows diffuse or focal thickening of the junctional zone as an area of low signal intensity compared with outer myometrium on T2 weighted images. Ectopic glands and haemorrhage in adenomyosis are usually small and punctate. Cystic adenomyosis (adenomyotic cyst) is a rare form of adenomyosis with extensive glandular cystic changes and haemorrhage.
The purpose of this report is to present MRI characteristics in a case of cystic adenomyosis, which presented as a huge exophytic cystic mass with florid glandular differentiation. MR findings of the mass mimicked ovarian carcinoma associated with endometriosis.
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Case report
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A 41-year-old woman (gravida 2) presented with dysmenorrhoea and hypermenorrhoea lasting for 16 years. Pelvic endometriosis had been previously diagnosed and she received gonadotropin releasing hormone (GnRH) analogue therapy from the age of 39 years. She presented with elevated serum levels of CA125 and CA19-9 of 673 U ml–1 and 846 U ml–1, respectively. Transvaginal ultrasound showed adenomyosis and a left ovarian tumour of cystic and solid appearance. Left ovarian carcinoma was suspected and MRI was performed with a 1.5 T magnet (Magnetom Vision; Siemens Medical Systems, Erlangen, Germany) to confirm the diagnosis and for a staging. A multiloculate cystic mass of 8 cm x 8 cm x 6 cm was demonstrated abutting the left side of the uterus. The cystic loculi showed high signal intensity on T1 weighted images and variable signal intensity on T2 weighted images, which suggested chocolate-like haemorrhage (Figure 1a–c
). The solid component within the mass contained foci of high and low T2 signal intensity (Figure 1b,c). On T1 weighted images, the solid structure demonstrated isosignal intensity compared with the myometrium, and markedly enhanced after intravenous administration of Gadoteridol (0.1 mmol kg–1) (Figure 1d). In the uterus, adenomyosis was demonstrated on T2 weighted images, and the interface between the solid component of the mass and myometrium with adenomyosis was irregular and unclear. Signal voids bridged the mass and the uterus, suggesting a hypervascular ovarian tumour invading the uterus. Our pre-operative diagnosis was adenomyosis and a left malignant ovarian tumour associated with endometriosis (i.e. endometrioid carcinoma or clear cell carcinoma) invading the uterus (stage IIA). Total abdominal total hysterectomy and bilateral salpingo-oophorectomy were performed.
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Figure 1. A 41-year-old woman with cystic adenomyosis. (a) Axial T1 weighted MR image shows hyperintense cystic mass (arrows) abutting left side of the uterus (U). Solid structure (arrowheads) with small hyperintense foci was seen within the mass. (b) Axial and (c) sagittal T2 weighted images with fat saturation shows hyperintense solid structure (arrowheads) in the hypointense loculus. The interface between the mass and uterus was irregular and unclear. Note that signal voids bridge between the uterus and the mass (short arrows). Adenomyosis is recognized in the uterus (long arrows). (d) Axial contrast-enhanced T1 weighted image with fat saturation shows marked enhancement in the solid structure (arrowheads). (e) Photomicrograph of the cystic adenomyosis (haematoxylin and eosin, x10). The cystic adenomyosis (arrows) includes the protuberance with florid glandular differentiation (arrowheads). Adenomyosis noted in the uterine myometrium (long arrows). (f) Photomicrograph of the glandular protuberance within the cystic adenomyosis (haematoxylin and eosin, x40). Endometrioid glands vary in size lie in haemorrhagic cellular stroma with no malignant transformation. Therefore they were considered as simple endometrial hyperplasia.
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Macroscopic histology showed a fist-sized tumour adhering to the uterine myometrium with adenomyosis (Figure 1e
). Microscopically, the tumour was encapsulated by the hypertrophic myometrium and filled with haemorrhage. The solid protuberance within the tumour consisted of florid endometrioid glands that varied in size and haemorrhagic cellular stroma (Figure 1f). This was consistent with simple endometrial hyperplasia without malignant transformation. Microscopic examination with Berlin blue staining revealed the wall of the cyst contained haemosiderin-laden macrophages. The final pathological diagnosis was cystic adenomyosis. The patient has remained well for 2 years after surgery.
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Discussion
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Adenomyosis usually presents as diffuse or focal thickening of the junctional zone on MRI. Myometrial hyperplasia, a feature of adenomyosis, demonstrates low signal intensity compared with the outer myometrium on T2 weighted images. It manifests as increased junctional zone thickness with unclear margins [2, 3]. Ectopic endometrial glands correspond to high signal intensity foci on T2 weighted images, some of which are haemorrhagic [3, 4]. Ectopic endometrial glands in adenomyosis are characterized by a predominance of zona basalis and it does not respond to cyclic hormonal stimulation, whereas the zona functionalis does in endometriosis [5, 6]. Therefore haemorrhagic lesions in adenomyosis are usually small and punctate. The cystic form of adenomyosis with extensive glandular cystic changes and haemorrhage is rare [7, 8]. Haemosiderin deposits in the wall of adenomyotic cyst may explain that haemorrhage has occurred several times in the cyst as does in endometriotic cyst. A few MRI reports presented the cases of myometrial and subserosal cystic adenomyosis as prominent high intensity cysts on both T1 and T2 weighted images [7, 8]. The cysts were surrounded by low intensity tissue [7, 8]. Our MR findings differed from those of other reports. In our case, abundant solid structure within the mass led to misdiagnoses of ovarian carcinoma associated with endometriotic cyst. The interface between the solid structure and myometrium was diffuse. Signal voids bridging the uterus and tumour were demonstrated and should have suggested a mass of uterine origin. However, we interpreted these findings as invasion of the uterus by an ovarian carcinoma.
The spectrum of epithelial abnormalities including epithelial metaplasia, hyperplasia, atypia, and adenocarcinoma (i.e. endometrioid carcinoma and clear cell carcinoma) have been described in ovarian endometriotic cysts [9]. A report showed that endometriotic cyst with malignant transformation seldom shows low signal intensity on T2 weighted images and usually has enhancing mural nodules [10]. The authors speculated that adenocarcinomas in endometriosis may produce some fluid that can dilute dense haemorrhagic material. In contrast, in our case of cystic adenomyosis, the loculi showed variable intensity and the enhancing solid structures were in the hypointense loculus on T2 weighted images.
There have been few reports of adenocarcinomas arising from adenomyosis. The adenocarcinomas were present in the myometrium without involvement of the eutopic endometrium. Koshimaya et al [11] reported four cases of adenocarcinoma arising form adenomyosis, including a transition from benign adenomyotic to carcinomatous glands. In these cases, MRI and ultrasound did not detect the disease correctly and pre-operative diagnoses were ovarian carcinoma in two cases, adenomyosis in one case, and leiomyoma or leiomyosarcoma in one case. In two cases mimicking ovarian malignancy, tumours showed 7–7.5 cm solid and cystic appearance.
The process of the florid endometrioid glands (endometrial hyperplasia, simple) induced within the cystic adenomyosis remains unclear. The previous use of GnRH analogue might modify the glandular differentiation, since it is known that adenomyosis recurs when hormonal therapy is discontinued. MRI demonstrated this glandular protuberance high signal intensity on T2 weighted images and marked enhancement. It is interesting that the same has been experienced for simple endometrial hyperplasia of the uterine endometrium [12].
In conclusion, in our patient with huge subserosal cystic adenomyosis, a confident differentiation of uterine versus ovarian mass, and of benign versus malignant tumour could not be made. Signal voids bridging the uterus and tumour should suggest a diagnosis of a mass of uterine origin. Hyperintense protuberance in the hypointense loculus on T2 weighted images may be an atypical appearance of malignant ovarian tumour associated with endometriosis. However, surgical exploration and resection is still required to exclude an ovarian malignancy.
Received for publication May 13, 2004.
Revision received January 4, 2005.
Accepted for publication January 26, 2005.
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Abstract
Introduction
