Ameku Koken, Higa Mariko
Department of Respiratory Medicine, Okinawa Prefectural Nanbu Medical Center & Children's Medical Center, Okinawa, Japan.
Case Rep Hematol. 2018 Jan 23;2018:5217301. doi: 10.1155/2018/5217301. eCollection 2018.
Protein S (PS) deficiency, an autosomal dominant hereditary thrombophilia, is more prevalent in East Asian populations than in Caucasians. PS-deficient patients have historically been administered a heparin product followed by warfarin for the treatment and secondary prevention of venous thromboembolism (VTE). However, warfarin can be ineffective or causes detrimental effects in rare cases. While direct oral anticoagulants (DOACs) are being increasingly used for the treatment and prevention of VTE, their efficacy in PS-deficient patients has not been established. We describe a 91-year-old woman who presented with chronic bilateral lower leg swelling with VTE that was refractory to warfarin anticoagulation therapy for over 1 year. Her recurrent VTE was diagnosed as quantitative hereditary PS deficiency. Rivaroxaban was administered as maintenance therapy instead of warfarin; after 8 weeks, the severities of the patient's leg swelling and venous ulcerations were significantly reduced with rivaroxaban compared to warfarin, thus demonstrating the efficacy of rivaroxaban for warfarin-refractory chronic VTE associated with hereditary PS deficiency. This case illustrates that rivaroxaban can potentially serve as therapeutic agents to treat warfarin-refractory VTE in PS-deficient patients. Further investigations are required to confirm the efficacy of rivaroxaban on the long term in this regard.
蛋白S(PS)缺乏症是一种常染色体显性遗传性血栓形成倾向疾病,在东亚人群中比在白种人中更为普遍。历史上,PS缺乏症患者一直先接受肝素类产品治疗,随后使用华法林来治疗和二级预防静脉血栓栓塞(VTE)。然而,华法林在罕见情况下可能无效或产生有害影响。虽然直接口服抗凝剂(DOACs)越来越多地用于治疗和预防VTE,但其在PS缺乏症患者中的疗效尚未得到证实。我们描述了一名91岁女性,她因慢性双侧小腿肿胀伴VTE就诊,华法林抗凝治疗1年多效果不佳。她的复发性VTE被诊断为遗传性定量PS缺乏症。使用利伐沙班替代华法林进行维持治疗;8周后,与华法林相比,利伐沙班显著减轻了患者的腿部肿胀和静脉溃疡程度,从而证明了利伐沙班对与遗传性PS缺乏症相关的华法林难治性慢性VTE的疗效。该病例表明,利伐沙班有可能作为治疗药物用于治疗PS缺乏症患者的华法林难治性VTE。在这方面,需要进一步研究以证实利伐沙班的长期疗效。