Department of Internal Medicine, Cheikh Khalifa International University Hospital, Mohammed VI University of Health Sciences, Casablanca, Morocco.
Laboratory of Clinical Immunology, Inflammation and Allergy, Faculty of Medicine and Pharmacy of Casablanca, Hassan II University of Casablanca, Casablanca, Morocco.
Medicine (Baltimore). 2022 Nov 4;101(44):e31308. doi: 10.1097/MD.0000000000031308.
Budd-Chiari syndrome (BCS) is considered a rare but serious complication of Behçet's disease (BD). This study was performed to define the prevalence, clinical and biological features, treatment, and clinical course of BSC associated with BD in a Moroccan population. We retrospectively analyzed the medical records of 1578 patients fulfilling the international diagnostic criteria for BD, including those with BSC. Eighteen male and 3 female patients, with a mean age of 36 ± 8.6 years. The inferior vena cava was involved in 81% (n = 17) of cases. All forms of BCS were noted: the chronic form in 52.4% (n = 11), the subacute form in 38% (n = 8), and the fulminant form (2 cases). Ascites was the main clinical sign and was present in 62% of patients (n = 13). Other venous thromboses (superior vena cava and lower limbs) were associated with BSC in 52.4% of patients (n = 11). Arterial involvement was noted in 28.6% (n = 6). Cardiac manifestations were present in 19% (n = 4) of the patients. All the patients received anticoagulants associated with corticosteroids. Immunosuppressants were used in 95% (n = 20). One patient received infliximab. Severe complications were noted in 38% (n = 8) of patients (digestive bleeding, confusion, infections and liver failure). Four patients have died during the study period. BCS in patients with BD is not uncommon and can be life threatening. It is frequently associated with other vascular manifestations that can be difficult to treat, particularly in the presence of pulmonary artery aneurysms. Prognosis improved with the use of immunosuppressants. Biologics can be promising in the early stages.
布加综合征(BCS)被认为是白塞病(BD)的一种罕见但严重的并发症。本研究旨在确定摩洛哥人群中与 BD 相关的 BCS 的患病率、临床和生物学特征、治疗和临床过程。我们回顾性分析了符合 BD 国际诊断标准的 1578 例患者的病历,包括 BCS 患者。18 名男性和 3 名女性,平均年龄 36±8.6 岁。81%(n=17)的病例涉及下腔静脉。所有形式的 BCS 均有报道:慢性形式占 52.4%(n=11),亚急性形式占 38%(n=8),暴发性形式(2 例)。腹水是主要的临床体征,62%的患者(n=13)存在腹水。52.4%的患者(n=11)合并其他静脉血栓形成(上腔静脉和下肢)。28.6%(n=6)的患者存在动脉受累。19%(n=4)的患者存在心脏表现。所有患者均接受抗凝治疗联合皮质类固醇治疗。95%的患者(n=20)使用免疫抑制剂。1 例患者接受英夫利昔单抗治疗。38%的患者(n=8)出现严重并发症(消化道出血、意识障碍、感染和肝功能衰竭)。研究期间,4 例患者死亡。BD 患者的 BCS 并不罕见,可能危及生命。它经常与其他血管表现相关,这些表现可能难以治疗,尤其是在存在肺动脉瘤的情况下。使用免疫抑制剂可改善预后。生物制剂在早期可能有前途。