Tatara Mio, Tanaka Yuji, Takebayashi Akie, Tsuji Shunichiro, Murakami Takashi
Department of Obstetrics and Gynecology, Shiga University of Medical Science, Otsu, JPN.
Department of Obstetrics and Gynecology, Shiga University of Medical Science, Ostu, JPN.
Cureus. 2024 Dec 21;16(12):e76168. doi: 10.7759/cureus.76168. eCollection 2024 Dec.
Familial Mediterranean fever (FMF) is an autoinflammatory disease characterized by periodic fever, serositis, and arthritis. In women, FMF attacks can sometimes be triggered by the menstrual cycle. Once diagnosed, prophylactic treatment with colchicine is generally recommended. Here, we report the case of a 34-year-old nulligravid Japanese woman who met the Tel-Hashomer criteria for FMF with menstruation-associated attacks and experienced a severe FMF episode following controlled ovarian hyperstimulation (COH) for in vitro fertilization (IVF) without prophylactic colchicine. We also discuss management strategies for menstruation-associated FMF in patients who do not receive prophylactic therapy. Although prophylactic colchicine treatment was advised, the patient declined, and a plan for symptomatic management was established. She was referred to our clinic for infertility and subsequently underwent IVF due to endometriosis. At the initial examination, an increase in follicle-stimulating hormone (FSH) levels was observed, and endogenous endocrine function was regulated by administering estrogen and progestin. She experienced two withdrawal bleeding episodes without FMF attacks. Subsequently, COH using a gonadotropin-releasing hormone (GnRH) antagonist protocol was initiated, and oocyte retrieval was performed when her estradiol (E2) level reached 953 pg/mL. Immediately after the withdrawal bleeding following oocyte retrieval, she experienced her most severe FMF attack to date, presenting with fever above 38°C, diffuse abdominal pain, vomiting, and joint pain. To prevent further FMF attacks during the waiting period before embryo transfer, colchicine or dienogest was administered, effectively suppressing additional episodes. This case suggests that in patients with menstruation-associated FMF, withdrawal bleeding after COH may strongly provoke FMF attacks due to the abrupt decline in estrogen levels. It underscores the importance of prophylactic therapy with colchicine or other agents during assisted reproductive technology (ART). Furthermore, when prophylactic treatment is not feasible, alternative strategies such as fresh embryo transfer should be considered.
家族性地中海热（FMF）是一种自身炎症性疾病，其特征为周期性发热、浆膜炎和关节炎。在女性中，FMF发作有时可由月经周期触发。一旦确诊，通常建议使用秋水仙碱进行预防性治疗。在此，我们报告一例34岁未孕日本女性病例，该患者符合FMF的Tel-Hashomer标准，有与月经相关的发作，在未进行预防性秋水仙碱治疗的情况下，因体外受精（IVF）进行控制性卵巢刺激（COH）后经历了一次严重的FMF发作。我们还讨论了未接受预防性治疗的与月经相关的FMF患者的管理策略。尽管建议进行预防性秋水仙碱治疗，但患者拒绝了，于是制定了对症管理计划。她因不孕症转诊至我们的诊所，随后因子宫内膜异位症接受了IVF。在初次检查时，观察到促卵泡生成素（FSH）水平升高，并通过给予雌激素和孕激素来调节内源性内分泌功能。她经历了两次撤退性出血发作，未出现FMF发作。随后，启动了使用促性腺激素释放激素（GnRH）拮抗剂方案的COH，当她的雌二醇（E2）水平达到953 pg/mL时进行了取卵。取卵后的撤退性出血后，她立即经历了迄今为止最严重的FMF发作，表现为体温高于38°C、弥漫性腹痛、呕吐和关节疼痛。为防止胚胎移植前等待期进一步发生FMF发作，给予了秋水仙碱或地诺孕素，有效抑制了额外发作。该病例表明，在与月经相关的FMF患者中，COH后的撤退性出血可能因雌激素水平突然下降而强烈诱发FMF发作。这凸显了在辅助生殖技术（ART）期间使用秋水仙碱或其他药物进行预防性治疗的重要性。此外，当预防性治疗不可行时，应考虑替代策略，如新鲜胚胎移植。
