Center for Genetic Epidemiology, Norris Comprehensive Cancer Center, University of Southern California, Los Angeles, California.
Department of Chronic Disease Epidemiology, Yale School of Public Health, New Haven, Connecticut.
Cancer Epidemiol Biomarkers Prev. 2020 Jun;29(6):1162-1167. doi: 10.1158/1055-9965.EPI-20-0093. Epub 2020 Apr 3.
Synovial sarcoma is a rare cancer with peak incidence in the young adult period. Despite poor outcomes of this aggressive cancer, there is little epidemiologic research addressing its etiology.
We collected birth characteristic data on synovial sarcoma cases born during 1978-2015 and diagnosed during 1988-2015 in California ( = 244), and 12,200 controls frequency-matched on year of birth. We also constructed a dataset of cancer cases in siblings of sarcoma subjects to assess familial risk.
In multivariable logistic regression analyses, synovial sarcoma was more frequent in Hispanics compared with non-Hispanic whites [OR, 1.48; 95% confidence interval (CI), 1.06-2.08]. Higher birth weight was a risk factor in Hispanics; each 500 g increase in birth weight was associated with a 22% increase in disease risk (OR, 1.22; 95% CI, 1.00-1.48). Also, a strong role for birth order was suggested, with highest risk for the first born (second child compared with first: OR, 0.61; 95% CI, 0.44-0.84; third or later compared with first: OR, 0.53; 95% CI, 0.36-0.77). Siblings of patients with synovial sarcoma did not display elevated cancer incidence, suggesting the low likelihood that strong familial predisposition alleles play a significant role in this disease.
The associations with birth weight and birth order suggest that nutritional, developmental, and environmental factors may play a role in the etiology of synovial sarcoma.
Further epidemiologic research on synovial sarcoma should evaluate epigenetic and developmental mechanisms and the formation of the archetypical t(X;18) translocation that defines this disease.
滑膜肉瘤是一种罕见的癌症,发病率高峰在青年期。尽管这种侵袭性癌症的预后较差,但针对其病因的流行病学研究却很少。
我们收集了 1978 年至 1988 年期间在加利福尼亚出生并于 1988 年至 2015 年期间诊断为滑膜肉瘤的病例(=244 例)和按出生年份匹配的 12200 名对照者的出生特征数据。我们还构建了一个包含肉瘤患者兄弟姐妹的癌症病例数据集,以评估家族风险。
在多变量逻辑回归分析中,与非西班牙裔白人相比,西班牙裔滑膜肉瘤的发病率更高[比值比(OR),1.48;95%置信区间(CI),1.06-2.08]。较高的出生体重是西班牙裔的一个危险因素;出生体重每增加 500 克,疾病风险增加 22%(OR,1.22;95%CI,1.00-1.48)。此外,出生顺序的作用也很强,第一个出生的孩子风险最高(第二个孩子与第一个孩子相比:OR,0.61;95%CI,0.44-0.84;第三个或更多的孩子与第一个孩子相比:OR,0.53;95%CI,0.36-0.77)。滑膜肉瘤患者的兄弟姐妹没有表现出癌症发病率升高,这表明强烈的家族易感性等位基因不太可能在这种疾病中发挥重要作用。
出生体重和出生顺序的相关性表明,营养、发育和环境因素可能在滑膜肉瘤的病因中发挥作用。
针对滑膜肉瘤的进一步流行病学研究应评估表观遗传和发育机制,以及定义这种疾病的典型 X;18 易位的形成。