Dobkin Shoshana Leftin, Chang Xiao, Mentch Frank, Hakonarson Hakon
Division of Pulmonary Medicine, The Hub for Clinical Collaboration, Children's Hospital of Philadelphia, 3501 Civic Center Boulevard, Philadelphia, PA, 19104-4318, USA.
Center for Applied Genomics, Children's Hospital of Philadelphia, 3615 Civic Center Boulevard, Suite 1216, Philadelphia, PA, 19104-4318, USA.
Sci Rep. 2025 May 2;15(1):15462. doi: 10.1038/s41598-025-98305-4.
Childhood cancer survivors (CCS) often face late-onset adverse effects, with pulmonary complications being particularly common. In this study, we analyzed 104 CCS with pulmonary issues post-treatment using a genome-wide association study (GWAS) to identify genetic susceptibility markers. Among the cohort, 20% were diagnosed with interstitial lung disease (ILD), while other complications included pulmonary fibrosis (3%) and bronchiolitis obliterans (3%). Pulmonary function test (PFT) impairments were present in 74% of patients, with symptoms such as exertional dyspnea (14%) and chronic cough (9%). Imaging revealed bronchiectasis in 7% of patients. Although no genome-wide significant loci were identified, several loci of suggestive significance were detected, including rs6804526 (P = 6.21 × 10-5) and rs117985268 (P = 2.98 × 10-5), which are previously associated with breast cancer and respiratory failure, respectively. Additionally, copy number variation (CNV) analysis identified a 21q22.3 deletion in one patient, a region linked to lung cancer. While no genome-wide significant loci were found, these suggestive loci and CNV findings highlight potential genetic modifiers of pulmonary risk in CCS, warranting further investigation in larger cohorts to confirm these associations.
儿童癌症幸存者(CCS)常常面临迟发性不良反应,肺部并发症尤为常见。在本研究中,我们对104名治疗后出现肺部问题的CCS进行了全基因组关联研究(GWAS),以确定遗传易感性标记。在该队列中,20%被诊断为间质性肺疾病(ILD),其他并发症包括肺纤维化(3%)和闭塞性细支气管炎(3%)。74%的患者存在肺功能测试(PFT)损伤,症状包括运动性呼吸困难(14%)和慢性咳嗽(9%)。影像学检查显示7%的患者有支气管扩张。虽然未发现全基因组显著位点,但检测到了几个具有提示意义的位点,包括rs6804526(P = 6.21×10-5)和rs117985268(P = 2.98×10-5),它们之前分别与乳腺癌和呼吸衰竭有关。此外,拷贝数变异(CNV)分析在一名患者中发现了21q22.3缺失,该区域与肺癌有关。虽然未发现全基因组显著位点,但这些具有提示意义的位点和CNV结果突出了CCS肺部风险的潜在遗传修饰因素,需要在更大的队列中进一步研究以证实这些关联。