The Diagnostic Performance of Early Sjögren's Syndrome Autoantibodies in Juvenile Sjögren's Syndrome: The University of Florida Pediatric Cohort Study.
The Diagnostic Performance of Early Sjögren's Syndrome Autoantibodies in Juvenile Sjögren's Syndrome: The University of Florida Pediatric Cohort Study.
复制标题
DOI：
10.3389/fimmu.2021.704193
复制
发表时间：
2021
影响因子：
7.3
通讯作者：
Cha S
中科院分区：
文献类型：
--
作者：
Thatayatikom A;Jun I;Bhattacharyya I;Berg K;Lee YJ;Kim Y;Adewumi A;Zhang W;Thatayatikom S;Shah A;Beal C;Modica R;Elder ME;Cha S
本研究的目的是评估早期干燥综合征（SS）自身抗体（eSjA）的临床有效性，该抗体最初用于早期诊断SS，用于最近确定的儿童队列中的青少年SS（JSS）。在佛罗里达大学孤儿自身免疫性疾病中心评价了总共105例具有eSjA结果的症状性受试者，并入组本研究。JSS诊断基于2016年ACR/EULAR SS标准。比较了JSS（n = 27）和非JSS（n = 78）之间的人口统计学/临床/实验室参数，包括eSjA（SP 1，抗唾液蛋白; CA 6，抗碳酸酐酶VI; PSP，抗腮腺分泌蛋白）和经典SS自身抗体（cSjA; ANA、SSA/SSB、RF和其他）单独或联合的阳性率、灵敏度和特异性。eSjA和诊断/腺体参数之间的关联也通过Fisher精确检验确定。与非JSS相比，JSS患者表现出干燥症状，表现为非刺激唾液流率（USFR）降低和唾液腺超声（SGUS）显示的异常腺体特征。在cSjA中，ANA的敏感性最高，为69.2%，而SSA、SSB和RF的特异性约为95%。JSS中的阳性SSA %显著高于非JSS（56% vs. 5%）。在eSjA中，抗CA 6 IgG是最普遍的，而不区分JSS（37%）和非JSS（32%）。eSjA的敏感性和特异性分别为55.6%和26.9%。仅在cSjA中观察到对JSS具有潜在适用特异性/灵敏度的自身抗体，未纳入单个eSjA。在整个队列、JSS和非JSS分析中，未检测到eSjA与病灶评分（FS）、USFR、SSA、SGUS和腮腺炎/腺体肿胀之间的相关性。然而，在一小组阳性SSA中发现抗PSP和腮腺炎/腺体肿胀之间存在负相关性（n = 19，p = 0.02），而在抗PSP阳性与抗PSP阴性之间没有发现这种相关性。JSS和非JSS组在FS、USFR和EULAR SS患者报告的CA 6/PSP/ANA、SP1和SSA阳性组的干燥指数/平均值方面分别存在差异。此外，RF阳性个体的FS高于RF阴性个体。eSjA在区分JSS与非JSS方面表现低于cSjS。发现eSjA对JSS早期诊断的临床影响需要进行纵向研究。
The aim of this study was to evaluate the clinical validity of early Sjögren’s syndrome (SS) autoantibodies (eSjA), which were originally marketed for early diagnosis of SS, for juvenile SS (JSS) in a recently identified pediatric cohort. A total of 105 symptomatic subjects with eSjA results available were evaluated at the Center for Orphaned Autoimmune Disorders at the University of Florida and enrolled for this study. JSS diagnosis was based on the 2016 ACR/EULAR SS criteria. Demographic/clinical/laboratory parameters were compared between JSS (n = 27) and non-JSS (n = 78) for % positivity, sensitivity, and specificity of eSjA (SP1, anti-salivary protein; CA6, anti-carbonic anhydrase VI; PSP, anti-parotid secretory protein) and classic SS-autoantibodies (cSjA; ANA, SSA/SSB, RF, and others) either alone or in combination. Associations between eSjA and diagnostic/glandular parameters were also determined by Fisher’s exact test. Compared to non-JSS, JSS patients exhibited sicca symptoms demonstrating reduced unstimulated salivary flow rate (USFR) and abnormal glandular features revealed by salivary gland ultrasound (SGUS). Among cSjA, ANA demonstrated the highest sensitivity of 69.2%, while SSA, SSB, and RF showed around 95% specificities for JSS diagnosis. The % positive-SSA was notably higher in JSS than non-JSS (56% vs. 5%). Of eSjA, anti-CA6 IgG was the most prevalent without differentiating JSS (37%) from non-JSS (32%). Sensitivity and specificity of eSjA were 55.6 and 26.9%, respectively. Autoantibodies with potentially applicable specificity/sensitivity for JSS were seen only in cSjA without a single eSjA included. There were no associations detected between eSjA and focus score (FS), USFR, SSA, SGUS, and parotitis/glandular swelling analyzed in the entire cohort, JSS, and non-JSS. However, a negative association between anti-PSP and parotitis/glandular swelling was found in a small group of positive-SSA (n = 19, p = 0.02) whereas no such association was found between anti-PSP-positive compared to anti-PSP-negative. JSS and non-JSS groups differed in FS, USFR, and EULAR SS Patient Reported Index Dryness/Mean in CA6/PSP/ANA, SP1, and SSA-positive groups, respectively. Additionally, a higher FS was found in RF-positive than RF-negative individuals. eSjA underperformed cSjS in differentiating JSS from non-JSS. The discovery of clinical impact of eSjA on early diagnosis of JSS necessitates a longitudinal study.
登录
查看更多内容
影响因子：
2.8
作者：
Bunya VY;Ying GS;Maguire MG;Kuklinski E;Lin MC;Peskin E;Asbell PA;DREAM Study Research Group
通讯作者：
DREAM Study Research Group
影响因子：
0.9
作者：
Li, Xinjun;Sjostedt, Cecilia;Sundquist, Kristina
通讯作者：
Sundquist, Kristina
影响因子：
2.8
作者：
Bunya, Vatinee Y.;Massaro-Giordano, Mina;Ying, Gui-shuang
通讯作者：
Ying, Gui-shuang
影响因子：
29.4
作者：
GORDON, SC;QUATTROCIOCCHILONGE, TM;KIECHLE, FL
通讯作者：
KIECHLE, FL
影响因子：
2.9
作者：
Frulloni, L;Bovo, P;Cavallini, G
通讯作者：
Cavallini, G