Conservative management of uterine adenosarcoma: lessons learned from 20\xa0years of follow-up

Ariadne L’Heveder,Robert Richardson,J Richard Smith,Srdjan Saso,Baljeet Kaur,Sadaf Ghaem-Maghami,Benjamin P Jones,Jen Barcroft,Joseph Yazbek

doi:10.1007/s00404-019-05306-6

Abstract

PurposeUterine adenosarcomas (UAs) account for 5–8% of cases of uterine sarcomas. Treatment includes total abdominal hysterectomy (TAH) and bilateral salpingo-oophorectomy (BSO).Fertility preservation is an emerging concept in gynaecology oncology and is particularly relevant in UA, where cases are diagnosed as young as 15-year-old. This manuscript demonstrates a case of UA which was treated conservatively, achieved successful livebirths and underwent completion hysterectomy after two decades of follow-up.MethodThis was a retrospective case note review.ResultsAn 18-year-old nulliparous woman presented with abnormal vaginal bleeding. Ultrasound identified an endometrial polyp, which was histologically diagnosed as low-grade adenosarcoma. She was advised to undergo TAH and BSO, but instead decided to preserve her fertility and opted for conservative management. She was monitored with pelvic ultrasound, hysteroscopy and endometrial biopsy bi-annually, with annual pelvic magnetic resonance imaging for 10 years which was uneventful. 11 years post-operatively she conceived following in-vitro fertilization (IVF) but suffered a miscarriage at 16 weeks likely due to cervical incompetence. She subsequently conceived with twins. She delivered spontaneously preterm at 28 weeks. Both children are alive and well. After 20 years of follow-up, she underwent a laparoscopic hysterectomy with no evidence of recurrence. She remains disease free.ConclusionWhilst radical completion surgery should be advised in UA, this case, in addition to all published conservatively managed cases of UA, demonstrates that conservative management is possible in appropriately selected women. Intensive monitoring post-operatively is essential owing to the risk of recurrence; however, this may pose deleterious side effects which require consideration.

Highlights

We present a case of a young woman with Uterine adenosarcomas (UAs) who was treated conservatively, subsequently achieved livebirth and underwent completion hysterectomy after two decades of follow-up
There was no histological evidence of disease recurrence. This case demonstrates the feasibility of Fertility sparing surgery (FSS) in appropriately selected women with UA
Two (20%) women received adjuvant chemotherapy: one had nine cycles of vincristine, dactinomycin and cyclophosphamide following polypectomy with no residual disease at a subsequent dilatation and curettage (D&C) [11], whilst the other was treated with ifosfamide and cisplatin [9]

Summary

Introduction

Uterine adenosarcoma (UA) accounts for 5–8% of cases of uterine sarcomas, a rare form of neoplasm comprising less than 10% of uterine malignancies [1]. Eleven years post-operatively, she underwent in vitro fertilization (IVF) and successfully conceived following the first cycle She suffered a second trimester miscarriage at 16 weeks’ gestation, with a history suggestive of cervical incompetence after admission with a dilated cervix and bulging membranes. She was offered hysterectomy after delivery, and at each subsequent follow-up appointment, but declined as she was uncertain as to whether or not her family was complete Following pregnancy, her follow-up intensity was reduced to annually with continued ultrasound scans along with hysteroscopy and endometrial biopsy. Her follow-up intensity was reduced to annually with continued ultrasound scans along with hysteroscopy and endometrial biopsy She continued to defer definitive treatment until, after a total of 20 years of follow-up, she underwent an uncomplicated laparoscopic hysterectomy with ovarian conservation.

Discussion

Findings

Compliance with ethical standards