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神经嵴细胞起源的出生缺陷与儿科胚胎性肿瘤之间的关联。

Associations between birth defects with neural crest cell origins and pediatric embryonal tumors.

机构信息

Department of Epidemiology, Gillings School of Global Public Health, The University of North Carolina at Chapel Hill, Chapel Hill, North Carolina, USA.

Section of Hematology-Oncology, Department of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.

出版信息

Cancer. 2023 Nov 15;129(22):3595-3602. doi: 10.1002/cncr.34952. Epub 2023 Jul 11.

Abstract

BACKGROUND

There are few assessments evaluating associations between birth defects with neural crest cell developmental origins (BDNCOs) and embryonal tumors, which are characterized by undifferentiated cells having a molecular profile similar to neural crest cells. The effect of BDNCOs on embryonal tumors was estimated to explore potential shared etiologic pathways and genetic origins.

METHODS

With the use of a multistate, registry-linkage cohort study, BDNCO-embryonal tumor associations were evaluated by generating hazard ratios (HRs) and 95% confidence intervals (CIs) with Cox regression models. BDNCOs consisted of ear, face, and neck defects, Hirschsprung disease, and a selection of congenital heart defects. Embryonal tumors included neuroblastoma, nephroblastoma, and hepatoblastoma. Potential HR modification (HRM) was investigated by infant sex, maternal race/ethnicity, maternal age, and maternal education.

RESULTS

The risk of embryonal tumors among those with BDNCOs was 0.09% (co-occurring n = 105) compared to 0.03% (95% CI, 0.03%-0.04%) among those without a birth defect. Children with BDNCOs were 4.2 times (95% CI, 3.5-5.1 times) as likely to be diagnosed with an embryonal tumor compared to children born without a birth defect. BDNCOs were strongly associated with hepatoblastoma (HR, 16.1; 95% CI, 11.3-22.9), and the HRs for neuroblastoma (3.1; 95% CI, 2.3-4.2) and nephroblastoma (2.9; 95% CI, 1.9-4.4) were elevated. There was no notable HRM by the aforementioned factors.

CONCLUSIONS

Children with BDNCOs are more likely to develop embryonal tumors compared to children without a birth defect. Disruptions of shared developmental pathways may contribute to both phenotypes, which could inform future genomic assessments and cancer surveillance strategies of these conditions.

摘要

背景

目前很少有评估方法可以评估神经嵴细胞发育起源相关先天畸形(BDNCO)与胚胎性肿瘤之间的关联,胚胎性肿瘤的特征是未分化的细胞具有类似于神经嵴细胞的分子特征。通过评估 BDNCO 对胚胎性肿瘤的影响,来探索潜在的共同发病机制和遗传起源。

方法

利用多状态、登记-链接队列研究,通过 Cox 回归模型生成风险比(HR)和 95%置信区间(CI),评估 BDNCO-胚胎性肿瘤的关联。BDNCO 包括耳部、面部和颈部畸形、先天性巨结肠病和部分先天性心脏病。胚胎性肿瘤包括神经母细胞瘤、肾母细胞瘤和肝母细胞瘤。通过婴儿性别、母亲种族/民族、母亲年龄和母亲教育情况,对潜在的 HR 修饰(HRM)进行了研究。

结果

与无出生缺陷的儿童相比,有 BDNCO 的儿童发生胚胎性肿瘤的风险为 0.09%(共病 n=105),而无出生缺陷的儿童为 0.03%(95%CI,0.03%-0.04%)。与未患出生缺陷的儿童相比,患有 BDNCO 的儿童患胚胎性肿瘤的可能性高出 4.2 倍(95%CI,3.5-5.1 倍)。BDNCO 与肝母细胞瘤密切相关(HR,16.1;95%CI,11.3-22.9),神经母细胞瘤(HR,3.1;95%CI,2.3-4.2)和肾母细胞瘤(HR,2.9;95%CI,1.9-4.4)的 HR 也有所升高。上述因素没有明显的 HRM。

结论

与无出生缺陷的儿童相比,患有 BDNCO 的儿童更有可能患上胚胎性肿瘤。共享发育途径的中断可能会导致这两种表型,这可以为这些疾病的未来基因组评估和癌症监测策略提供信息。

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