Akyol Lütfi, Toz Bahtiyar, Bayındır Özün, Zengin Orhan, Cansu DöndüÜsküdar, Yiğit Murat, Çetin Gözde Yıldırım, Omma Ahmet, Erden Abdulsamet, Küçükşahin Orhan, Altuner Mehmet Şakir, Çorba BurçinŞeyda, Ünal Ali Uğur, Küçük Hamit, Küçük Adem, Balkarli Ayşe, Gönüllü Emel, Tufan Ayşe Nur, Bakırcı Sibel, Öner Sibel Yılmaz, Balcı Mehmet Ali, Kobak Şenol, Yazıcı Ayten, Özgen Metin, Şahin Ali, Koca Süleyman Serdar, Erer Burak, Gül Ahmet, Aksu Kenan, Keser Gökhan, Onat Ahmet Mesut, Kısacık Bünyamin, Kaşifoğlu Timuçin, Çefle Ayşe, Kalyoncu Umut, Sayarlıoğlu Mehmet
Department of Internal Medicine, Division of Rheumatology, Health Sciences University, Gazi Yaşargil Training and Research Hospital, Diyarbakır, Turkey.
Department of Medicine, Icahn School of Medicine At Mount Sinai, Queens Hospital Center, New York, NY, USA.
Clin Rheumatol. 2022 Jan;41(1):177-186. doi: 10.1007/s10067-021-05878-2. Epub 2021 Aug 9.
To compare the clinical features, laboratory findings, and prognosis of Behçet's disease (BD) patients with and without Budd-Chiari syndrome (BCS).
This multicenter retrospective study investigated 61 (M/F: 41/20) patients with BD, having coexistent BCS, and 169 (M/F:100/69) BD patients as the control group without BCS from 22 different centers of Turkey diagnosed between 1990 and 2017.
Of the total 61 BD patients with BCS, the onset of the first symptom and the median age of diagnosis were earlier in contrast to BD patients without BCS (p = 0.005 and p = 0.007). Lower extremity deep vein and inferior vena cava (IVC) thrombosis were more common in patients with BCS (all; p < 0.01) compared to the control group. Mortality was significantly higher in BD-BCS patients with IVC thrombosis than in the controls (p = 0.004). Since most of the cases in our cohort had chronic and silent form of BCS, mortality rate was 14.8%, which was on the lower range of mortality rate reported in literature (14-47%). While all BD-BCS patients received immunosuppressive (IS) agents, only half of them received additional anticoagulant treatments. Among IS agents, interferon treatment was more frequently used in this cohort (19%), compared to other series reported in literature (2.3%).
To our knowledge, this is the largest series of BD patients with BCS. Our patients had earlier disease onset and diagnosis, higher frequency of IVC thrombosis, and higher mortality rate, compared to BD patients without BCS. Mortality was significantly higher in BD-BCS patients with IVC thrombosis compared to controls. Key Points • Mortality rate is higher in BD-associated BCS patients with IVC involvement. • Chronic and silent form of BD-associated BCS has a better prognosis. • The main treatment options are corticosteroids and immunosuppressive agents, whereas anticoagulant treatment remains controversial.
比较伴有和不伴有布加综合征(BCS)的白塞病(BD)患者的临床特征、实验室检查结果及预后。
这项多中心回顾性研究调查了来自土耳其22个不同中心的61例(男/女:41/20)患有BD且合并BCS的患者,以及169例(男/女:100/69)不伴有BCS的BD患者作为对照组,这些患者均在1990年至2017年期间被诊断。
在总共61例伴有BCS的BD患者中,与不伴有BCS的BD患者相比,首发症状出现时间及诊断时的中位年龄更早(p = 0.005和p = 0.007)。与对照组相比,BCS患者下肢深静脉和下腔静脉(IVC)血栓形成更为常见(均p < 0.01)。伴有IVC血栓形成的BD - BCS患者死亡率显著高于对照组(p = 0.004)。由于我们队列中的大多数病例为慢性和无症状形式的BCS,死亡率为14.8%,处于文献报道的死亡率范围下限(14 - 47%)。虽然所有BD - BCS患者均接受免疫抑制(IS)药物治疗,但其中只有一半接受了额外的抗凝治疗。在IS药物中,与文献报道的其他系列(2.3%)相比,本队列中干扰素治疗的使用更为频繁(19%)。
据我们所知,这是最大规模的伴有BCS的BD患者系列研究。与不伴有BCS的BD患者相比,我们的患者疾病起病和诊断更早,IVC血栓形成频率更高,死亡率更高。伴有IVC血栓形成的BD - BCS患者死亡率显著高于对照组。要点 • 伴有IVC受累的BD相关BCS患者死亡率更高。 • BD相关BCS的慢性和无症状形式预后较好。 • 主要治疗选择是皮质类固醇和免疫抑制药物,而抗凝治疗仍存在争议。