Ponce Ana, Rodríguez-Pintó Ignasi, Espinosa Gerard, Quintas Helena, Erkan Doruk, Shoenfeld Yehuda, Cervera Ricard
Autoimmune Diseases Unit, Department of Internal Medicine, Hospital de Viladecans, Viladecans, Catalonia, Spain.
Autoimmune Diseases Unit, Department of Internal Medicine, Hospital Universitari Mútua de Terrassa, Terrassa, Catalonia, Spain.
Semin Arthritis Rheum. 2023 Dec;63:152265. doi: 10.1016/j.semarthrit.2023.152265. Epub 2023 Oct 7.
To describe the pulmonary involvement in patients with catastrophic antiphospholipid syndrome (CAPS), focusing on its relationship with extrapulmonary involvement, laboratory, radiological, and pathological findings.
This retrospective cross-sectional study includes all patients grouped in the "CAPS Registry". All cases were reviewed, and those with pulmonary thromboembolism (PE) and/or diffuse alveolar hemorrhage (DAH) were selected. Data on pulmonary and extrapulmonary clinical presentation, radiologic patterns, laboratory findings, associated autoimmune diseases, treatments, and outcomes were analyzed. Frequency distribution and measures of central tendency were used to describe the cohort. Comparison between groups regarding qualitative variables was undertaken by chi-square or Fisher exact test, while T-test for independent variables was used to compare groups regarding continuous variables. IBM-SPSS v.22 was used for data analysis.
PE was reported in 129 (48.6 %) episodes, DAH in 75 (28.3 %) episodes, and overlap (DAH plus PE) in 7 (2.6 %) episodes. Bronchoalveolar lavage (BAL) was performed in 35 (4.9 %) CAPS episodes, and lung pathology samples were obtained in 84 (10.5 %) episodes (including autopsies). A significant relationship was observed between DAH and laboratory features of thrombotic microangiopathy (TMA). A meaningful relationship was also found between triple antiphospholipid antibody positivity and pathological TMA (26.5 %) as well as hypocomplementemia and DAH (24 %).
Pulmonary involvement may include both TMA and non-thrombotic inflammation, which can be differentiated into three patterns: PE, DAH with systemic TMA with hypocomplementemia or DAH without systemic TMA with/without hypocomplementemia.
描述灾难性抗磷脂综合征(CAPS)患者的肺部受累情况,重点关注其与肺外受累、实验室检查、影像学及病理学表现的关系。
这项回顾性横断面研究纳入了“CAPS注册登记处”的所有患者。对所有病例进行了回顾,并选取了发生肺血栓栓塞(PE)和/或弥漫性肺泡出血(DAH)的患者。分析了肺部和肺外临床表现、放射学模式、实验室检查结果、相关自身免疫性疾病、治疗方法及预后的数据。采用频率分布和集中趋势测量来描述该队列。对于定性变量,采用卡方检验或Fisher精确检验进行组间比较;对于连续变量,则采用独立样本t检验进行组间比较。使用IBM-SPSS v.22进行数据分析。
报告有129例(48.6%)发生PE,75例(28.3%)发生DAH,7例(2.6%)为二者重叠(DAH加PE)。35例(4.9%)CAPS病例进行了支气管肺泡灌洗(BAL),84例(10.5%)病例获取了肺病理样本(包括尸检)。观察到DAH与血栓性微血管病(TMA)的实验室特征之间存在显著关系。还发现抗磷脂抗体三联阳性与病理性TMA(26.5%)以及低补体血症与DAH(24%)之间存在有意义的关系。
肺部受累可能包括TMA和非血栓性炎症,可分为三种模式:PE、伴有低补体血症的系统性TMA的DAH或伴有/不伴有低补体血症的无系统性TMA的DAH。