Unexpected uterine leiomyosarcoma in a woman with multiple myomas treated with ulipristal acetate: case report and literature review

Abstract

Ulipristal acetate (UPA) has been recognized as an alternative strategy to surgery in the management of symptomatic women with uterine fibroids. We present a case report on a woman with hereditary fibrinogen deficiency exclusively treated with UPA for myoma-related menorrhagia and abdominal pain, who subsequently underwent a hysterectomy because of clinical worsening. A FIGO IB uterine leiomyosarcoma was found among multiple myomas. A review of the literature found two other cases of uterine leiomyosarcoma in patients treated with UPA: clinical data are reported. The aim of this case report is to increase clinicians' awareness that, although rare, leiomyosarcoma can develop in a uterus with multiple myomas and no reliable diagnostic tools exist yet. Thus, a clinical and instrumental careful reevaluation and patient counseling should be a priority when planning to repeat UPA treatment cycles.

Chinese abstract

在有症状的子宫肌瘤患者的管理中, 醋酸乌利司他（Ulipristal acetate, UPA）已被认为是一种可代替手术的策略。我们作一个病例报告, 一名患有遗传性纤维蛋白原缺乏症的女性只用UPA治疗肌瘤相关的月经过多和腹痛, 该患者因为临床症状恶化随后行子宫切除术。FIGO分期IB期子宫平滑肌肉瘤在多发性子宫肌瘤中发现。文献综述中找到在UPA治疗的患者中其他两例子宫平滑肌肉瘤：临床数据被报告。这份病例报告的目的是提高临床医生的意识：平滑肌肉瘤虽然少见, 但其可在多发肌瘤的子宫中形成且目前尚无可靠的诊断工具。因此, 当计划重复UPA治疗周期时, 临床和仪器的认真重新评估以及患者的咨询应优先考虑。

Keywords:

• ulipristal acetate
• uterine myoma
• uterine leiomyosarcoma

Disclosure statement

Paola Modaffari, Marta D’Alonzo, Marta Garbagnati, Silvia Pecchio, Prof Guido Menato have no conflict of interest, nor financial ties that may be deemed to influence the objectivity of the paper. Prof Nicoletta Biglia has served as a consultant, lecturer and/or member of Advisory Board for: Shinogi, Gedeon Richter, Italfarmaco and MSD.