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British Journal of Radiology (2003) 76, 66-68
© 2003 British Institute of Radiology
doi: 10.1259/bjr/19147576
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Case report

Adenomyoma mimicking an aggressive uterine neoplasm on MRI

A M Connors 1 N M deSouza 1 and G A McIndoe, MRCOG 2

Departments of 1 Radiology and 2 Gynaecology, Hammersmith Hospital, Du Cane Road, London W12 0HS, UK


   Abstract
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 Abstract
Case report
 Discussion
 References
 
This case report concerns a nulliparous female with prolonged vaginal bleeding, where MRI demonstrated a mass with an aggressive, tumour like appearance involving the posterior aspect of the uterus. Histological examination confirmed that this was an adenomyoma. The unusual imaging appearance of this lesion and its differential diagnosis are discussed. Adenomyoma should be considered in the differential diagnosis of aggressive-appearing uterine masses.


   Case report
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 Abstract
 Case report
Discussion
 References
 
A 30-year-old nulliparous female presented with an 8-week history of continuous vaginal bleeding. Prior to this her menstrual cycle was regular. She had previously taken the combined oral contraceptive pill but had discontinued doing so several months before the excessive bleeding commenced. On pelvic examination there was a muco-purulent discharge from the cervix. This discharge was initially considered to be infective in origin, but failed to respond to antibiotic therapy.

Alpha fetoprotein, beta-human chorionic gonadotrophin and carcinoembryonic antigen (CEA) were within the normal range. The CA-125 was elevated at 239 U ml-1 (normal range 0–23 U  ml-1).

Ultrasound demonstrated a bulky uterus containing several hypoechoic, thin-walled discrete masses, believed to be abscesses. The ovaries were not visualized. There was no ascites. An examination under anaesthesia revealed pus and mucus in the vagina. Despite further antibiotic therapy, the complex uterine masses persisted.

MRI of the pelvis was performed on a 0.5 T Apollo scanner (Marconi Medical Systems, Highland Heights, OH). Sequence details included sagittal and axial T1 (638 ms repetition time (TR) and 20 ms echo time (TE)) and T2 (2500 ms TR and 80 ms TE) weighted imaging fast spin echo 4000 ms TR and 88 ms (effective TE) and short Tau inversion recovery (STIR) (2000 ms TR, 107 ms time to inversion (TI) and 30 ms TE) coronal imaging. The cervix was distorted and elongated. There was a 9 x 10 x 11 cm mass lying to the left of the midline on the posterior aspect of the uterus (Figure 1Go). It was heterogeneous in signal intensity, solid peripherally and cystic centrally, within which was a loculated air collection. The T1 weighted appearance was predominantly low signal, although there were several small, high signal foci within. The T2 weighted appearance was more heterogeneous with areas of high and low signal throughout, the high signal foci on T1 weighted images showing low T2 signal. The endocervical canal was continuous with a cavity within the mass, which contained air. The central portion of the mass communicated directly with the endometrial cavity. All layers of the uterus were involved, but there was no invasion of adjacent structures. No pelvic or inguinal lymphadenopathy was seen. The ovaries were normal in appearance. Due to the aggressive tumour like appearance, surgery was arranged. Frozen section was planned with possible uterine conservation, although the patient was also consented for hysterectomy.



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  		Figure 1. Adenomyoma. (a) Sagittal T2 weighted (fast spin echo; 4000 ms repetition time and 88 ms echo time) and (b) transverse T2 weighted (spin echo; 4000 ms repetition time and 80 ms echo time) images show a large posterior myometrial mass. The mass contains heterogeneous solid components (arrowhead) and is partly cystic (black arrow) with loculated air (curved arrow). The cystic component communicates directly with the endometrial cavity (open white arrow) and endocervical cavity. No fistula to bowel or bladder is seen.

 
Intra-operatively, a 10 cm mass was identified arising from the posterior aspect of the uterus and cervix, containing solid and cystic elements. On incising the serosa, a natural plane of dissection developed and the lesion "shelled out", leaving a defect in the posterior wall of the uterus. Frozen section histology was performed showing the gross specimen to be a lobulated mass with areas of cystic degeneration, measuring 9 x 10 x 5 cm. The histology of the excised lobulated mass was composed of smooth muscle fascicles containing glands lined by mucinous and ciliated epithelium. No malignant features were identified. The features were in keeping with an adenomyoma. The defect in the posterior aspect of the uterus was repaired and the patient made an uneventful post-operative recovery.

Complete histological staining demonstrated a uterine mass composed of abundant smooth muscle and irregularly distributed benign glands. The periglandular stroma was pauci-cellular hyalinized but, occasionally, specialized endometrial type stroma was also seen. No atypical features were identified, confirming an adenomyoma.


   Discussion
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 Abstract
 Case report
 Discussion
References
 
True adenomyomas are unusual benign encapsulated tumours of the uterus. The most common location for adenomyomas is the uterine corpus and endocervical region [1]. Macroscopic gross appearance is usually that of a well circumscribed polypoid mass, cystic areas can be seen on sectioning. Focal haemorrhage has been described [2]. The histological appearance of adenomyomas is that of a proliferation of endometrial glands, endometrioid stroma and smooth muscle without atypical features [1]. Leiomyomas, by comparison, consist of smooth muscle with varying quantities of connective tissue, without a glandular component [3]. Although of uncertain aetiology, adenomyomas are frequently associated with multiparity in pre-menopausal women [4]. Atypical polypoid adenomyoma is another entity, consisting of endometrial glands, with varying degrees of atypia and hyperplasia, within smooth muscle.

On MRI, adenomyomas are typically ill-defined in outline, have an irregular interface with the myometrium and are of low signal intensity, relative to myometrium on T2 weighted imaging and contrast enhanced T1 weighted imaging. High signal foci on either T2 weighting alone or on both T1 weighting and T2 weighting are seen in the majority of cases of focal adenomyosis, although not as frequently in diffuse adenomyosis [4]. These have been shown to correspond to islands of heterotopic endometrium and haemorrhagic foci [5]. Leiomyomas have similar imaging characteristics but are well circumscribed round lesions and are usually homogeneous in appearance, but may be heterogeneous when associated with degeneration [3]. Atypical polypoid adenomyomas are usually hypointense polypoid masses with areas of high signal intensity on T2 weighted images [6].

The differential diagnosis of a large uterine lesion includes leiomyoma (this entity may be indistinguishable from adenomyoma [2]), endometrial adenocarcinoma and uterine sarcoma. Uterine sarcomas are most commonly leiomyosarcomas, mixed müllerian tumours or endometrial stromal sarcomas. Sahdev et al [7] describe two major imaging patterns in their series of 22 patients with uterine sarcomas. One is that of a large heterogeneous pelvic mass and the second is that of an endometrial mass indistinguishable from endometrial carcinoma. Endometrial adenocarcinomas are usually heterogeneous masses extending into the uterine cavity; spread beyond the endometrium is demonstrated on MRI as intermediate signal intensity on T2 weighting disrupting the endometrial/myometrial interface [8]. Endometrial stromal sarcoma and endometrial adenocarcinoma are commonly associated with deep invasion of the myometrium, reflecting their aggressive nature.

In the patient described here, the uterine lesion was large in size, occupying most of the posterior aspect of the uterus, and extended through all layers of uterus. The endometrial surface was ulcerated and had a heterogeneous appearance on MRI. These features pointed towards an aggressive lesion.

The CA-125 was elevated at 239 U ml-1 (normal range 0–23 U ml-1). There are several possible causes of elevated CA-125, both benign and malignant. Benign gynaecological conditions that lead to CA-125 elevation include adenomyosis, endometriosis, leiomyoma, pelvic inflammatory disease and pregnancy. Malignant conditions include epithelial ovarian carcinoma, endometrial carcinoma and certain non-gynaecological cancers e.g. pancreas, breast, colon and lung. Eltabbakh et al [9] found that with a CA-125 level above 65 U ml-1, approximately 87% of patients will have malignant disease and 13% benign, and that the association with malignancy increases as the CA-125 level increases. Our patient's CA-125 level was three times greater than this threshold and was taken as further pre-operative evidence of malignancy.

Fortunately for this young patient, who was hoping to start a family, frozen section histology showed that this mass was benign, in spite of the CA-125 level and its aggressive appearance, and the lesion was resected, sparing the uterus. The CA-125 level returned to normal after surgery and remained normal on subsequent follow-up. We suggest that in cases of uterine masses that appear aggressive in nature, adenomyoma should be considered in the differential diagnosis.

Received for publication March 20, 2002. Revision received June 24, 2002. Accepted for publication July 12, 2002.


   References
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 Abstract
 Case report
 Discussion
 References
 

  1. Gilks CB, Clement PB, Hart WR, Young RH. Uterine adenomyomas excluding atypical adenomyomas and adenomyomas of the endocervical type: a clinicopathologic study of 30 cases of an underemphasized lesion that may cause diagnostic problems with brief consideration of adenomyomas of other female genital tract sites. Int J Gynecol Pathol 2000;19:195–205.[Medline]
  2. Reinhold C, Tafazoli F, Mehio A, Wang L, Atri M, Siegelman ES, et al. Uterine adenomyosis: endovaginal US and MR imaging features with histopathologic correlation. Radiographics 1999;19(Suppl.):S147–60.
  3. Murase E, Siegelman ES, Outwater EK, Perez-Jaffe LA, Tureck RW. Uterine leiomyomas: histopathologic features, MR imaging findings, differential diagnosis, and treatment. Radiographics 1999;19:1179–97.[Abstract/Free Full Text]
  4. Byun JY, Kim SE, Choi BG, Ko GY, Jung SE, Choi KH. Diffuse and focal adenomyosis: MR imaging findings. Radiographics 1999;19(Suppl.):S161–70.
  5. Togashi K, Ozasa H, Konishi I, Itoh H, Nishimura K, Noma I, et al. Enlarged uterus: differentiation between adenomyosis and leiomyoma with MR imaging. Radiology 1989;171:531–4.[Abstract/Free Full Text]
  6. Yamashita Y, Torashima M, Hatanaka Y, Takahashi M, Fukumatsu K, Tanaka N, et al. MR imaging of atypical polypoid adenomyoma. Comput Med Imaging Graph 1995;19:351–5.[CrossRef][Medline]
  7. Sahdev A, Sohaib SA, Jacobs I, Shepherd JH, Oram DH, Reznek RH. MR imaging of uterine sarcomas. AJR 2001;177:1307–11.[Abstract/Free Full Text]
  8. Scoutt LM, McCarthy SM. Female pelvis. In: Stark DD, Bradley WG, editors. Magnetic Resonance Imaging, (3rd edn). St Louis, MO: Mosby,1999:571–4.
  9. Eltabbakh GH, Belinson JL, Kennedy AW, Gupta M, Webster K, Blumenson LE. Serum CA-125 measurements >65 U/ml. Clinical value. J Reprod Med 1997;42:617–24.[Medline]



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