Department of Rheumatology and Clinical Immunology, Chinese Academy of Medical Sciences & Peking Union Medical College, National Clinical Research Center for Dermatologic and Immunologic Diseases (NCRC-DID), Ministry of Science & Technology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital (PUMCH), Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing, China.
Department of Epidemiology and Biostatistics, Institute of Basic Medical Sciences Chinese Academy of Medical Sciences, School of Basic Medicine Peking Union Medical College, Beijing, PR China.
Autoimmun Rev. 2022 May;21(5):103084. doi: 10.1016/j.autrev.2022.103084. Epub 2022 Mar 24.
Patients with primary Sjögren's syndrome(pSS) have increased risk of non-Hodgkin lymphoma (NHL). However, whether pSS patients have increased risk of other malignancies is unclear. The aim of this study is to investigate the association between pSS and the risk of malignancy, with a focus on hematological malignancies besides lymphoma and solid tumors through a systematic review and meta-analysis.
We searched PubMed and EMBASE by March 21st 2021. Inclusion criteria were as follows: (1) pSS was the exposure of interest; (2) newly developed malignancies were the outcome of interest; (3) standardized incidence ratio or relative risk with 95% confidence interval or essential data to calculate them were reported. (4) Study design was cohort study. Patient with other connective diseases were excluded. Quality assessment was conducted according to Newcastle-Ottawa Scale for cohort study. Random or fixed effect models were used to calculate the pooled SIR according to heterogeneity measured by I.
A total of 1003 articles were found by a comprehensive search in PubMed and EMBASE. Twenty-eight articles were eligible. Four of them were from the same database, and the one with longest observational span was chosen. Therefore, twenty-five articles were included for final analysis, which involved more than 47,607 pSS patients with the follow-up of more than 452,468 person-year. We found that pSS was significantly associated with increased risks of overall malignancy(pooled SIR 2.17, 95%1.57-3.00), hematological malignancy(pooled SIR 11.55, 95%CI 4.32-30.90) including NHL(pooled SIR 13.71, 95%CI 8.83-21.29), Hodgkin lymphoma(pooled SIR 8.84, 95%CI 5.00-15.61), multiple myeloma(pooled SIR 8.27, 95%CI 3.08-22.24), leukemia(pooled SIR 2.56, 95%CI 1.78-3.69) and solid tumors(pooled SIR 1.39, 95%CI 0.90-2.13) including lung cancer(pooled SIR 1.55, 95%CI 1.29-1.85), thyroid cancer(pooled SIR 2.05, 95%CI 1.20-3.48), non-melanoma skin cancer(pooled SIR 1.71, 95%CI 1.08-2.72), kidney/urinary tract cancer(pooled SIR 1.36, 95%CI 1.02; 1.81), liver cancer(pooled SIR 1.70, 95%CI 1.13-2.57) and prostate cancer(pooled SIR 1.50, 95%CI 1.02-2.22).
This meta-analysis showed that pSS patients had increased risk of overall cancer, which not only contributed by NHL, but also by other hematological malignancies and solid tumors.
原发性干燥综合征（pSS）患者发生非霍奇金淋巴瘤（NHL）的风险增加。然而，pSS 患者发生其他恶性肿瘤的风险是否增加尚不清楚。本研究旨在通过系统评价和荟萃分析，探讨 pSS 与恶性肿瘤风险之间的关系，重点关注除淋巴瘤和实体瘤以外的血液恶性肿瘤。
我们于 2021 年 3 月 21 日在 PubMed 和 EMBASE 中进行了检索。纳入标准如下：（1）pSS 为暴露因素；（2）新发恶性肿瘤为结局；（3）报道了标准化发病比或相对风险及 95%置信区间或计算所需的基本数据；（4）研究设计为队列研究。排除其他结缔组织疾病患者。根据纽卡斯尔-渥太华量表对队列研究进行质量评估。根据 I²测量的异质性，采用随机或固定效应模型计算汇总 SIR。
通过对 PubMed 和 EMBASE 的全面检索，共发现 1003 篇文章。28 篇文章符合纳入标准。其中 4 篇来自同一数据库，选择了观察跨度最长的一篇。因此，最终纳入 25 篇文章进行分析，共纳入 47607 例以上 pSS 患者，随访时间超过 452468 人年。我们发现 pSS 与总体恶性肿瘤风险增加显著相关（汇总 SIR 2.17，95%CI 1.57-3.00），血液恶性肿瘤（汇总 SIR 11.55，95%CI 4.32-30.90）包括 NHL（汇总 SIR 13.71，95%CI 8.83-21.29），霍奇金淋巴瘤（汇总 SIR 8.84，95%CI 5.00-15.61），多发性骨髓瘤（汇总 SIR 8.27，95%CI 3.08-22.24），白血病（汇总 SIR 2.56，95%CI 1.78-3.69）和实体瘤（汇总 SIR 1.39，95%CI 0.90-2.13）包括肺癌（汇总 SIR 1.55，95%CI 1.29-1.85），甲状腺癌（汇总 SIR 2.05，95%CI 1.20-3.48），非黑色素瘤皮肤癌（汇总 SIR 1.71，95%CI 1.08-2.72），肾/泌尿道癌（汇总 SIR 1.36，95%CI 1.02-1.81），肝癌（汇总 SIR 1.70，95%CI 1.13-2.57）和前列腺癌（汇总 SIR 1.50，95%CI 1.02-2.22）。
本荟萃分析表明，pSS 患者发生总体癌症的风险增加，这不仅与 NHL 有关，还与其他血液恶性肿瘤和实体瘤有关。
