Feasibility of uterine preservation in the management of early-stage uterine adenosarcomas: a single institute experience
© The Author(s). 2017
Received: 4 November 2016
Accepted: 22 March 2017
Published: 19 April 2017
We aimed to evaluate the efficacy and the safety of uterine preservation in patients with early-stage uterine adenosarcoma who want to preserve future fertility.
We performed a retrospective review of patients with stage I uterine adenosarcoma diagnosed and treated at a single institute from 1998 through 2014.
Among the total of 31 patients, uterine preservation surgery was performed in 7 of the nulliparas. Of the 7 patients receiving uterine preservation surgery, 3 showed no evidence of disease (NED), 2 had persistent disease confined to the uterus, and 2 were alive with disease (AWD) after recurrence. One patient with an NED status had a vaginal delivery at term. In the uterine preservation group, 1 patient had sarcomatous overgrowth at the time of diagnosis and experienced disease recurrence. In the hysterectomy group, 3 of 24 patients had tumor recurrence. Of the five patients with tumor recurrence, four (80%) had sarcomatous overgrowth at diagnosis and it was significantly associated with recurrence by univariate analysis (OR 13.3, p = 0.027).
Uterine preservation represents a possible treatment option for young female patients who want to maintain fertility. However, a detailed explanation of the risk of recurrence is necessary, especially in patients with sarcomatous overgrowth, which seems to be associated with a higher risk of recurrence.
KeywordsFertility preservation Mullerian adenosarcoma of the uterus Uterine adenosarcoma
Mullerian adenosarcoma is a rare malignancy composed of benign epithelial and malignant stromal components and usually arises from the uterus . Uterine adenosarcoma is considered to be a less aggressive disease than carcinosarcoma because its malignant component is usually low grade, and often a hysterectomy is curative . The presence of sarcomatous overgrowth seems to be the factor most strongly associated with an aggressive clinical course, postoperative recurrence, metastasis, and a fatal outcome [3, 4]. The presence of heterologous elements, tumor grade, and myometrial invasion depth are other risk factors for metastasis .
Due to its rarity, there are limited data on the optimal therapy (i.e., primary surgery or adjuvant therapy) for uterine adenosarcoma. Total hysterectomy is generally considered the primary intervention for this disease, but uterine preservation is often desired in reproductive-age women. We aimed to analyze clinical outcomes according to which therapeutic methods were used in patients with early-stage uterine adenosarcoma to evaluate the efficacy of uterine preservation in patients wanting to preserve their future fertility.
Characteristics of the 31 study patients with early-stage uterine adenosarcoma
No. of patients (%)
Age at diagnosis, median (range)
44.5 (21–81 years)
Largest tumor diameter, mean (range)
4.66 (0.8–22.0 cm)
FIGO 2009 stage
Clinical outcomes of the study patients with early-stage uterine adenosarcoma who received uterine preservation therapy
Age at diagnosis
Time to recurrence, months
MPA 3 months
Cx mass excision
TAH c BSO d/t seeding
FIGO criteria (2009) for Mullerian adenosarcomas
Tumor limited to uterus
Tumor limited to endometrium and endocervix with no myometrial invasion
Less than or equal to half myometrial invasion
More than half myometrial invasion
Tumor extends beyond the uterus, within the pelvis
Tumor extends to extrauterine pelvic tissue
Tumor invades abdominal tissues (not just protruding into the abdomen)
More than one site
Metastasis to pelvic and or para-aortic lymph nodes
Tumor invades bladder and or rectum
Univariate and multivariate analysis for factors associated with uterine adenosarcoma recurrence
Odds ratio (95% CI)
Odds ratio (95% CI)
Removal or destruction of sexual organs is used as a primary therapy in most cases of gynecological malignancies originating from reproductive organs, such as the uterus, ovaries, fallopian tube, vulva, and vagina. However, destructive surgery or radiation can result in abrupt onset of menopause, fertility loss, sexual function decline, pelvic pain, depression, and anxiety. Because of these factors, careful counseling is needed when making a decision about these therapeutic methods. For uterine adenosarcoma, total hysterectomy with bilateral salpingo-oophorectomy is considered the standard primary therapy. However, in young patients with uterine adenosarcoma, surgery that spares fertility should be considered, as the malignant component is usually low grade, leading to a good prognosis. In the largest published study to date, only 2 out of 100 patients had adnexal involvement, and both of these had grossly abnormal adnexa . Given the low reported rates of ovarian involvement, ovarian preservation in patients of reproductive age with uterine adenosarcoma seems to be a reasonable option in the absence of gross metastasis [2, 8]. However, there are limited data on the safety and effectiveness of uterine-preserving surgery in patients with uterine adenosarcoma.
In our report, 7 of 9 nullipara received uterine preservation therapy, such as hysteroscopic mass excision, cervical mass excision, or dilatation and curettage. Of these 7 patients, 3 showed NED, 2 had persistent disease confined to the uterus, and 2 were AWD after recurrence at a median follow-up of 32 months. One patient with NED status was married after her diagnosis and went on to have a vaginal delivery 17 months after primary therapy. Therefore, 1 of 1 patient (100%) in the uterine preservation group who tried to get pregnant was successful. Two patients with persistent disease and 1 patient with disease recurrence had tumor lesions confined to the uterus during the follow-up period. Therefore, at a median follow-up of 32 months, 6 of 7 patients (85.7%) in the uterine preservation group still have the possibility of a future pregnancy.
Characteristics and clinical outcomes of uterine adenosarcoma (literature review and present study)
No. of patients
Stage I of FIGO classification
SO + recurrence
Time to recurrencea
Caroll, et al. 
Tanner et al. 
Bernard et al. 
Kaku et al. 
To our knowledge, our current report is the first study to focus on uterine preservation in young patients with uterine adenosarcoma. Uterine preservation represents a possible treatment option for carefully screened young female patients who want to maintain fertility. However, a detailed explanation of the risks and benefits of the treatment alternatives and a strict oncological follow-up are necessary. Uterine preservation therapy might be risky, especially in patients with sarcomatous overgrowth, and definitive treatment such as hysterectomy seems to be a better choice. More data concerning the long-term outcomes of uterine preservation and adjuvant therapy are required in the future.
No funds were received in support of this work.
Availability of data and materials
The datasets during and/or analyzed during the current study are available from the corresponding author on reasonable requests.
KDY conceived the study, participated in its design, and critically revised the manuscript. LYJ assisted in the data analysis and drafting of the manuscript. SDS, KJH, KYM, KYT, NJH’s contributions were data acquisition and interpretation and drafting of the manuscript. All authors above made significant contributions to the content, design, and revision of this manuscript. All authors read and approved the final manuscript.
The authors declare that they have no competing interests.
Consent for publication
Ethics approval and consent to participate
This study obtained approval from the institutional review board at the Asan Medical Center (IRB No. 2015-0722).
There are neither commercial interests nor financial and/or commercial support.
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