Department of Gastroenterology, AP-HP, Hôpital Saint-Antoine and UPMC University of Paris, Paris, France.
Inflamm Bowel Dis. 2012 Nov;18(11):2063-71. doi: 10.1002/ibd.22889. Epub 2012 Jan 23.
It remains to be shown whether inflammatory bowel disease (IBD) is associated with an increased risk of primary intestinal lymphoproliferative disorders (PILD). We assessed this risk in the CESAME French nationwide prospective observational cohort.
In all, 680 gastroenterologists enrolled 19,486 patients with IBD (Crohn's disease in 60.3%) from May 2004 to June 2005. Follow-up ended on 31 December 2007. Available biopsy samples and surgical specimens from patients with PILD (n = 14) were centralized for review. The reference incidence of PILD in the general population was obtained from the Côte d'Or registry and was used as a comparator to assess the standardized incidence ratio (SIR). The influence of thiopurine exposure was explored in a nested case-control study.
In the CESAME population the crude incidence of PILD was 0.12/1000 patient-years, with a corresponding SIR of 17.51 (95% confidence interval [CI], 6.43-38.11; P < 0.0001). The risk was highest in patients exposed to thiopurines (SIR 49.52, 95% CI 13.49-126.8; P < 0.0001), while it did not reach statistical significance in patients naïve to thiopurines (SIR 4.83, 95% CI, 0.12-26.91; P = 0.37). The odds ratio associated with ongoing thiopurine exposure (vs. naïve) was 2.97 (95% CI, 0.30-infinity; P = 0.38). All 14 cases of PILD were non-Hodgkin's B-cell LD, 78.6% occurred in males, 85.7% arose in IBD lesions, and 45.5% were Epstein-Barr virus-positive. Eleven cases occurred in patients with Crohn's disease. Mean (SD) age at PILD diagnosis was 55.1 (5.6) years and the median time since IBD onset was 8.0 years (interquartile range, 3.0-15.8).
Patients with IBD have an increased risk of developing PILD.
炎症性肠病（IBD）是否会增加原发性肠道淋巴组织增生性疾病（PILD）的风险仍有待证实。我们在法国全国性前瞻性观察队列 CESAME 中评估了这种风险。
2004 年 5 月至 2005 年 6 月，680 名胃肠病学家共招募了 19486 名 IBD 患者（克罗恩病占 60.3%）。随访于 2007 年 12 月 31 日结束。对 PILD 患者（n=14）的可用活检样本和手术标本进行集中审查。一般人群中 PILD 的参考发病率来自科多尔登记处，并作为比较评估标准化发病率比（SIR）。在巢式病例对照研究中探讨了硫嘌呤暴露的影响。
在 CESAME 人群中，PILD 的粗发病率为 0.12/1000 患者年，相应的 SIR 为 17.51（95%置信区间 [CI]，6.43-38.11；P<0.0001）。暴露于硫嘌呤的患者风险最高（SIR 49.52，95%CI 13.49-126.8；P<0.0001），而未使用硫嘌呤的患者风险未达到统计学意义（SIR 4.83，95%CI，0.12-26.91；P=0.37）。与持续使用硫嘌呤（与未使用）相关的比值比为 2.97（95%CI，0.30-无穷大；P=0.38）。所有 14 例 PILD 均为非霍奇金 B 细胞 LD，78.6%发生于男性，85.7%发生于 IBD 病变，45.5%为 EBV 阳性。11 例发生于克罗恩病患者。PILD 诊断时的平均（SD）年龄为 55.1（5.6）岁，从 IBD 发病到 PILD 的中位时间为 8.0 年（四分位间距，3.0-15.8）。
IBD 患者发生 PILD 的风险增加。
