Adenosarcoma is a rare tumor which consists of benign glandular epithelium and malignant mesenchymal component. Here we report a case of adenosarcoma of the uterine corpus.
Adenosarcoma is a rare tumor which consists of benign glandular epithelium and malignant mesenchymal component. This entity was originally described by Clement and Scully [
A 59-year-old postmenopausal woman, gravida 2, para 2, presented with vaginal bleeding and visited a local clinic. Cytological tests of uterine cervix and endometrium were both negative. She had a uterine tumor pointed out and was referred to our hospital. Vaginal examination revealed enlarged uterus and ultrasound scans revealed a large heterogeneous mass occupying the whole uterine cavity (Figure
Ultrasound scans revealed a large heterogeneous mass occupying the whole uterine cavity.
Cytological test of endometrium was performed again but the result was negative. A fractional endometrial curettage revealed only fibrous tissue with epithelial-like cells. Magnetic resonance imaging (MRI) revealed a heterogeneous solid tumor of 77 × 76 mm (Figure
MRI (T2-weighted) revealed a heterogeneous solid tumor of 77 × 76 mm.
Degenerated myoma, leiomyosarcoma, or endometrial stromal sarcoma was suspected. Serum levels of CA125 were slightly elevated to 41.4 U/mL, whereas CA19-9 and CEA were within normal limits. The patient was admitted and total abdominal hysterectomy with bilateral salpingo-oophorectomy and pelvic lymphadenectomy was carried out (Figure
The surgical specimen.
At laparotomy, the uterus was fist size and no serosal invasion was observed. Both ovaries were intact. The tumor was arising from the uterine body and occupied the whole uterine cavity. Glandular epithelium with little atypia and proliferation of atypical mesenchymal cells were seen. Mitosis exceeded 2 per 10 high power fields. No myometrial invasion or lymph node metastasis was seen. The lesion was confined to the uterus. Peritoneal cytology revealed no malignant cells. Histopathological final diagnosis was adenosarcoma (Figure
(a) Phyllodes-like architecture on low magnification (H.E. ×100). (b) Periglandular cuffing of tumor cells (H.E. ×400).
Her postoperative course was uneventful. She was discharged without postoperative treatment and remains alive without disease 6 months after the surgery.
Mixed epithelial-mesenchymal tumors of the uterus include adenofibroma, adenosarcoma, and carcinosarcoma. Adenofibroma has benign glandular epithelial element and benign mesenchymal stroma, whereas carcinosarcoma has both malignant epithelial and mesenchymal stroma. Adenosarcoma is one of the rare diseases consisting of benign glandular epithelial element and malignant mesenchymal component. It may be classified as an intermediate state between the two formerly stated entities. It accounts for 8% of all uterine sarcomas. This entity was originally described by Clement and Scully [
Adenosarcoma is a typically low grade tumor and behave like low grade sarcoma. Adenosarcoma with sarcomatous overgrowth was first used by Clement in 1989 for those tumors that contain more than 25% of sarcomatous component [
Common symptom is genital bleeding. As for MRI findings, Yoshizako
Unfavourable prognostic factors are sarcomatous overgrowth, deep myometrial invasion, presence of heterologous elements and extrauterine spread [
As for lymphadenectomy, Kaku et al. reported a lymph node metastasis rate of 6.5% and para-aortic lymph node metastasis rate of 0% in 31 patients with adenosarcoma. Two patients with lymph node metastasis had myometrial invasion, heterologous elements, and sarcomatous overgrowth [
There is no optimal adjuvant or systemic treatment strategy but standard sarcoma chemotherapy regimens appear to have efficacy in both adenosarcoma and adenosarcoma with sarcomatous overgrowth [
Adenosarcoma of the uterus should be a differential diagnosis when a large polypoid mass is occupying the endometrial cavity and protruding into the vaginal cavity. A biopsy specimen often fails to diagnose this entity, and pathological diagnosis should be made on surgical specimen.
The authors declare that there is no conflict of interests regarding the publication of this paper.