Department of Immunology, IML and IdSSC, Hospital Clínico San Carlos, Madrid, Spain.
Department of Immunology, Ophthalmology and ENT, School of Medicine, Complutense University, Madrid, Spain.
Sci Rep. 2021 Jun 9;11(1):12211. doi: 10.1038/s41598-021-91791-2.
The broad and heterogeneous clinical spectrum that characterizes common variable immunodeficiency (CVID) is associated with quite different disease course and prognosis, highlighting the need to develop tools that predict complications. We developed a multianalyte VISUAL score (variable immunodeficiency score upfront analytical link) aimed to predict severity using individual CVID patient data at baseline of a cohort of 50 CVID patients from two different centers in Portugal and Spain. We retrospectively applied VISUAL to the CVID clinical severity scores proposed by Ameratunga and Grimbacher after 15 years follow-up of our cohort. VISUAL score at CVID diagnosis showed adequate performance for predicting infectious and non-infectious severe complications (Cluster B). Compared to switched memory B lymphocyte phenotype alone, VISUAL provided a more accurate identification of clinically meaningful outcome, with significantly higher sensitivity (85% vs 55%, p = 0.01), and negative predictive value (77% vs 58%) and AUC of the ROC curves (0.72 vs 0.64), with optimal cut-off level of 10. For every increase of 1 point in the VISUAL scale, the odds of being in the higher risk category (Cluster B) increased in 1.3 (p = 0.005) for Ameratunga's severity score and 1.26 (p = 0.004) for Grimbacher's severity score. At diagnosis of CVID, VISUAL score ≥ 10 showed 8.94-fold higher odds of severe prognosis than below this threshold. Kaplan-Meier estimates for the VISUAL ≥ 10 points showed significantly earlier progression to Cluster B than those with VISUAL < 10 (p = 0.0002). This prognostic laboratory score might allow close monitoring and more aggressive treatment in patients with scores ≥ 10 on a personalized basis approach. Further studies are needed to prospectively validate VISUAL score.
常见变异性免疫缺陷(CVID)的广泛而多样的临床谱与截然不同的疾病过程和预后相关,突出了开发预测并发症的工具的必要性。我们开发了一种多分析物 VISUAL 评分(可变免疫缺陷评分初步分析链接),旨在使用来自葡萄牙和西班牙两个不同中心的 50 名 CVID 患者队列的基线患者个体数据来预测严重程度。我们对队列 15 年随访后 Ameratunga 和 Grimbacher 提出的 CVID 临床严重程度评分进行了回顾性 VISUAL 应用。在 CVID 诊断时,VISUAL 评分在预测感染和非感染性严重并发症(Cluster B)方面表现出良好的性能。与单独的转换记忆 B 淋巴细胞表型相比,VISUAL 更准确地识别了有临床意义的结果,其灵敏度显著提高(85%比 55%,p=0.01),阴性预测值(77%比 58%)和 ROC 曲线下面积(0.72 比 0.64),最佳截断值为 10。VISUAL 量表每增加 1 分,在 Ameratunga 严重程度评分中,较高风险类别(Cluster B)的几率增加 1.3 倍(p=0.005),在 Grimbacher 严重程度评分中,几率增加 1.26 倍(p=0.004)。在 CVID 诊断时,VISUAL 评分≥10 与低于此阈值相比,严重预后的几率高 8.94 倍。VISUAL≥10 点的 Kaplan-Meier 估计表明,与 VISUAL<10 相比,向 Cluster B 的进展明显更早(p=0.0002)。这种预后实验室评分可能允许根据患者的分数(≥10)进行密切监测和更积极的治疗。需要进一步的前瞻性研究来验证 VISUAL 评分。