Ooi Katsuhiro, Wakimoto Yu, Oki Noriyoshi, Sakata Mina, Mabuchi Seiji
Gynecologic Oncology, Hyogo College of Medicine, Nishinomiya, JPN.
Obstetrics and Gynecology, Chibune General Hospital, Osaka, JPN.
Cureus. 2024 Nov 26;16(11):e74539. doi: 10.7759/cureus.74539. eCollection 2024 Nov.
Low-grade endometrial stromal sarcoma (LGESS) is a rare disease, accounting for less than 1% of all uterine malignancies. Standard treatment is total hysterectomy and bilateral tubal oophorectomy, although fertility preservation may be desirable because of the young age of onset. We document a case of fertility preservation in a 27-year-old nulligravida diagnosed with LGESS, which not only enabled the successful birth of two live infants but also underscores the efficacy of a multidisciplinary approach to patient treatment through the Hyogo Oncofertility Network (HOF-net). The patient underwent laparoscopic removal of a lesion initially suspected to be either a uterine myoma or an adnexal tumor. Through the HOF-net, a pioneering collaboration among patients, oncologists, and fertility specialists, she was connected to our department with the aim of achieving pregnancy in the future, even amidst her cancer diagnosis. Following the surgery, and during a medically advised period of contraception, assisted reproductive technology with the double stimulation (DuoStim) method was utilized to cryopreserve six embryos, followed by high-dose progestin therapy to mitigate the risk of cancer recurrence. Once the contraceptive mandate had been concluded, the implantation of the first thawed embryo during a hormone replacement cycle led to a viable pregnancy and the subsequent birth of a healthy child by cesarean section. Similarly, during the contraceptive period, after undergoing high-dose progestin therapy, a second thawed embryo transfer was performed, resulting in a successful pregnancy and the birth of a second child. Subsequently, she underwent a total hysterectomy and bilateral salpingectomy with preservation of both ovaries at another hospital 39 months after the initial surgery. No recurrence or residual disease was observed. The necessity for comprehensive informed consent was underscored by the potential for LGESS recurrence. Furthermore, the efficient coordination facilitated by the HOF-net enabled swift access to assisted reproductive services, aligning with the patient's primary healthcare plan. This case highlights the critical role of early engagement with assisted reproductive technologies and a multidisciplinary treatment strategy in facilitating successful outcomes for patients with LGESS, demonstrating the feasibility of fertility preservation in managing this condition.
低级别子宫内膜间质肉瘤(LGESS)是一种罕见疾病,占所有子宫恶性肿瘤的比例不到1%。标准治疗方法是全子宫切除术和双侧输卵管卵巢切除术,不过由于发病年龄较轻,保留生育功能可能是患者所期望的。我们记录了一例27岁未孕女性被诊断为LGESS后保留生育功能的病例,这不仅使她成功诞下两名活婴,还凸显了通过兵库肿瘤生育网络(HOF-net)采用多学科方法治疗患者的有效性。该患者最初接受了腹腔镜手术,切除了一个最初怀疑是子宫肌瘤或附件肿瘤的病变。通过HOF-net,患者、肿瘤学家和生育专家之间进行了开创性合作,即便在她被诊断患有癌症的情况下,仍以未来怀孕为目标将她转诊至我们科室。手术后,在医学建议的避孕期内,采用双刺激(DuoStim)法辅助生殖技术冷冻保存了6枚胚胎,随后进行大剂量孕激素治疗以降低癌症复发风险。一旦避孕期结束,在激素替代周期植入第一枚解冻胚胎后成功怀孕,随后通过剖宫产诞下一名健康婴儿。同样,在避孕期内,患者在接受大剂量孕激素治疗后,进行了第二次解冻胚胎移植,再次成功怀孕并诞下第二名婴儿。随后,在初次手术后39个月,她在另一家医院接受了全子宫切除术和双侧输卵管切除术,同时保留了双侧卵巢。未观察到复发或残留疾病。LGESS复发的可能性凸显了全面知情同意的必要性。此外,HOF-net促成的高效协调使患者能够迅速获得辅助生殖服务,与患者的基本医疗计划相契合。该病例突出了早期采用辅助生殖技术和多学科治疗策略在促进LGESS患者获得成功治疗结果方面的关键作用,证明了在这种情况下保留生育功能的可行性。