Annual and cumulative detection rate and three-year outcome of congenital heart disease diagnosed within one year after birth among children born during 2012 – 2018 in Ningbo city
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摘要:
目的 分析自然出生队列中婴幼儿在生后1年内先天性心脏病（以下简称先心病）的累计检出、年度变化趋势和生后3年内的结局转归情况，探索合适的监测时间和影响结局的因素。 方法 从浙江省宁波市自然出生队列中获取2012 — 2018年在宁波江北出生并完成生后3年随访的28 208例活产儿，收集生后1年内报告的全部先心病个案，描述性分析婴儿期内先心病的累计检出率；以年度变化百分比为主要指标，采用Join Point回归描述先心病检出的变化趋势和类型分布。随访调查1 304例先心病在出生后3年内发生死亡、自然愈合、手术等转归情况；使用多因素logistics回归模型分析转归结局中先心病不能自然愈合的影响因素和Kaplan-Meier生存分析计算自愈时间。 结果 宁波市2012 — 2018年自然出生队列监测28 208例活产儿，婴儿期内共发现1 304例先心病，每100名活产儿生后1年内的先心病累计检出率为4.62%。2012 — 2018年度累计检出率分别为1.87%、2.58%、3.13%、4.25%、6.20%、6.37%、8.26%，并呈逐年上升趋势，2016年为变化转折点。从先心病分类来看，构成比列前3位的分别是房间隔缺损431例（33.05%）、动脉导管未闭合并卵圆孔未闭232例（17.79%）和房间隔缺损合并卵圆孔未闭196例（15.03%）。出生后3、6、12个月内的先心病累计检出率分别为4.29%、 4.51%和4.62%，约97.62%的先心病在出生后6个月内被发现。全部1 304例先心病病例在出生后3年时间内，发生自然愈合934例（71.63%），M（P25，P75）自愈时间为8.6个月（7.8，9.4个）；接从受先心病手术60例（4.60%）；因先心病死亡22例（1.69%）；未愈149例（11.43%）；另外失访或拒绝随访139例（10.66%）。母亲年龄的增加会降低未愈风险（OR = 0.9342，95%CI = 0.893～0.974）；相对本省户籍，外省户籍增加儿童未愈风险（OR = 1.893，95%CI = 1.459～2.456）。先心病类型会显著影响自愈时长，复合型先心病自愈时长显著长于单纯型先心病，女婴愈合时间长于男婴，差异有统计学意义。 结论 可以延长先心病监测期至生后1年；超70%在出生3年内发生自然愈合，需谨慎选择手术时机。应重点关注外地户籍年轻母亲分娩的患复合型先心病女婴的先心病随访。 Abstract:Objective To analyze annual and cumulative detection rate and three-year outcome of congenital heart disease (CHD) diagnosed within one year after birth among children born during 2012 – 2018 in Ningbo city for developing appropriate measures on diagnosis and treatment of the disease. Methods The data on 28 208 live births during 2012 – 2018 in a district of Ningbo city, Zhejiang province were extracted from a natural birth cohort study conducted in the city. Totally 1 304 cases of CHD diagnosed within one year after birth among the live births were identified and followed up for 3 years after birth to collect the information on their outcomes of CHD. The annual and cumulative detection rate of CHD in the live births were analyzed and the change trend in annual detection rate of CHD were described with annual percentage change using join point regression. Kaplan-Meier survival analysis was used to calculate the duration of spontaneous-healing of CHD and multivariate logistic regression model was adopted to explore factors associated with CHD spontaneous healing of the cases. Results For all the live births, the cumulative detection rate of CHD diagnosed within one year after birth was 4.62 per 100 live births and the annual detection rate for years from 2012 to 2018 were 1.87, 2.58, 3.13, 4.25, 6.20, 6.37, and 8.26 per 100 live births, respectively, indicating an increasing trend. The number (proportion) of top three types of CHD detected were 431 (33.05%) for atrial septal defect, 232 (17.79%) for patent ductus arteriosus and patent foramen ovale, and 196 (15.03%) for atrial septal defect and patent foramen ovale. The cumulative detection rate of CHD diagnosed within 3, 6 , and 12 months after birth were 4.29, 4.51 and 4.62 per 100 live births. Of all the CHD cases, 97.62% were detected within 6 months after birth. The three-year after birth follow-ups revealed that among all the CHD cases, 934 (71.63%) had spontaneous-healing at a median time of 8.6 months (25th percentile: 7.8 months, 75th percentile 9.4 months); 60 (4.60%) underwent surgeries for CHD; 22 (1.69%) died of the disease; 149 (11.43%) were not healed or cured; and 139 (10.66%) were lost or refused to the follow-up. The results of multivariate logistic regression analysis showed that the CHD cases with elder maternal age at the birth were at decreased risk of without spontaneous healing three-year after birth (odd ratio [OR] = 0.9342, 95% confidence interval [95%CI]: 0.893 – 0.974) but the cases with the household registry of other provinces were at an increased risk of the spontaneous-healing compared to the cases with local province household registry (OR = 1.893, 95%CI: 1.459 – 2.456). The time of spontaneous healing of the female cases and the cases with complex CHD were significantly longer than that of male cases and the cases with simple CHD. Conclusion The results of the survey suggest that the CHD surveillance period could be extended to one year after birth among infants, especially among those being female, with younger maternal age at birth or those with household registry other than local province, and more than 70% of the CHD cases diagnosed within one year after birth could have spontaneous healing within three years after birth and this should be concerned in determining the time of surgery for CHD. -
Key words:
- congenital heart disease /
- cumulative detection rate /
- change trend /
- spontaneous healing
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表 1 宁波市2012 — 2018年分娩活产儿在生后1年的单纯性先心病检出率
Table 1. Annual and total detection rate of simple CHD within one year after birth among children born during 2012 – 2018 in Ningbo city
年份 活产数 失访或拒绝 诊断前死亡 合并其他畸形 单纯性先心病数 检出率（%） 2012 4302 114 11 5 78 1.87 2013 3789 86 10 8 95 2.58 2014 4438 82 5 9 136 3.13 2015 4021 80 10 3 167 4.25 2016 4433 90 8 11 268 6.20 2017 4372 89 4 7 272 6.37 2018 3572 72 6 9 288 8.26 合计 28927 613 54 52 1304 4.62 
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表 2 先心病病例数顺位前5病种诊断时间分布
Table 2. Total detection rate by months of age at diagnosis for top five cyanotic and non-cyanotic CHD most frequently diagnosed within one year after birth among children born during 2012 – 2018 in Ningbo city
先心病类型a 例数 构成比（%） 检出率（%） 诊断时间（月龄） 0～ 3～ 6～ 例数 构成比（%） 例数 构成比（%） 例数 构成比（%） 非紫绀型先心病 1274 97.70 4.52 1186 93.09 59 4.63 29 2.28 房间隔缺损 431 33.05 1.53 376 87.24 37 8.58 18 4.18 动脉导管未闭 + 卵圆孔未闭 232 17.79 0.82 226 97.41 4 1.72 2 0.86 房间隔缺损 + 卵圆孔未闭 196 15.03 0.69 188 95.92 6 3.06 2 1.02 房间隔缺损 + 动脉导管未闭 122 9.36 0.43 120 98.36 2 1.64 0 0.00 房间隔缺损 + 动脉导管未闭 + 卵圆孔未闭 74 5.67 0.26 74 100.00 0 0.00 0 0.00 紫绀型先心病 30 2.30 0.11 24 80.00 4 13.33 2 6.67 肺动脉狭窄 6 0.46 0.02 5 83.33 1 16.67 0 0.00 肺动脉狭窄 + 房间隔缺损 5 0.38 0.02 3 60.00 1 20.00 1 20.00 法洛四联症 4 0.31 0.01 4 100.00 0 0.00 0 0.00 肺动脉狭窄 + 卵圆孔未闭 3 0.23 0.01 3 100.00 0 0.00 0 0.00 主动脉狭窄 2 0.15 0.01 1 50.00 0 0.00 1 50.00 合计 1304 100.00 4.62 1210 92.79 63 4.83 31 2.38 注：a 仅列举发病率前5位的先心病类型。
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表 3 不同随访时间先心病转归情况
Table 3. Follow-up year-specific outcome for 1 304 CHD diagnosed within one year after birth among children born during 2012 – 2018 in Ningbo city
生后随访时间（年） 例数 自然愈合 手术 未愈 死亡 失访或拒绝 例数 构成比（%） 例数 构成比（%） 例数 构成比（%） 例数 构成比（%） 例数 构成比（%） 0～ 1304 333 25.54 15 1.15 889 68.17 17 1.30 50 3.83 0.5～ 889 312 35.10 19 2.14 515 57.93 3 0.34 40 4.50 1～ 515 197 38.25 20 3.88 263 51.07 2 0.39 33 6.41 2～ 263 92 34.98 6 2.28 149 56.65 0 0.00 16 6.08 0～3 1304 934 71.63 60 4.60 149 11.43 22 1.69 139 10.66
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表 4 多因素二分类logistic回归模型分析结果
Table 4. Associations of parental age and household registry with 3-year spontaneous healing rate of CHD diagnosed within one year after birth – multivariate dichotomous logistic regression analysis
变量 β $S_{\bar x}$ Wald χ2 值 P 值 HR 值 95%CI 父亲年龄 0.027 0.020 1.953 0.162 1.028 0.989～1.068 母亲年龄 – 0.700 0.022 9.937 0.002 0.933 0.893～0.974 户籍 外省 0.638 0.133 23.087 < 0.001 1.893 1.459～2.456 常量 – 0.074 0.441 0.028 0.866 0.828 注：父亲、母亲年龄为连续变量，外省参照组为本省。
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表 5 影响自愈时长因素分析
Table 5. Association of CHD type and gender with mean duration of spontaneous healing of CHD for 934 spontaneously cured CHD diagnosed within one year after birth among children born during 2012 – 2018 in Ningbo city
基本特征 总人数 自愈中位数时长（d） 自愈时长95%CI（d） H 值 P 值 先心病类型 – 3.708 < 0.001 单纯 580 204.5 123.25～376.50 复合 354 247.5 152.75～497.75 患儿性别 – 2.841 0.005 男童 499 199.0 127.00～376.00 女童 435 237.0 143.00～488.00
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[1] Dolk H, Loane M, Garne E, et al. Congenital heart defects in Europe: prevalence and perinatal mortality, 2000 to 2005[J]. Circulation, 2011, 123(8): 841 – 849. doi: 10.1161/CIRCULATIONAHA.110.958405 [2] Liu YJ, Chen S, Zühlke L, et al. Global birth prevalence of congenital heart defects 1970 – 2017: updated systematic review and meta - analysis of 260 studies[J]. International Journal of Epidemiology, 2019, 48(2): 455 – 463. doi: 10.1093/ije/dyz009 [3] 金肖. 青岛地区先天性心脏病发病率及影响因素的前瞻性队列研究[D]. 青岛: 青岛大学, 2021. [4] 李烁琳, 顾若漪, 黄国英. 儿童先天性心脏病流行病学特征[J]. 中国实用儿科杂志, 2017, 32(11): 871 – 875. doi: 10.19538/j.ek2017110617 [5] 李文先, 杜莉, 李旻明, 等. 2016 — 2018年上海市不同监测时段出生缺陷发病率比较[J]. 中华疾病控制杂志, 2021, 25(10): 1226 – 1230. [6] 赵趣鸣. 中国新生儿先天性心脏病筛查体系的建立与应用研究[D]. 上海: 复旦大学, 2013. [7] 胡盛寿. 《先天性心脏病外科治疗中国专家共识》述评[J]. 中国胸心血管外科临床杂志, 2021, 28(1): 1 – 3. [8] 刘小清, 麦劲壮, 庄建. 先天性心脏病流行病学研究方法新认识[J]. 中国循环杂志, 2011, 26(1): 74 – 76. doi: 10.3969/j.issn.1000-3614.2011.01.023 [9] Li MD, Collins G, Disenhouse R, et al. Spontaneous closure of ventricular septal defect[J]. The Cananian Medical Association Journal, 1969, 100(16): 737 – 743. [10] 张蕾, 安美玉, 朱冰, 等. 重庆乡镇0~3岁儿童先天性心脏病筛查与随访[J]. 中国当代儿科杂志, 2017, 19(7): 748 – 753. [11] 王金秀, 张琳, 李晓英, 等. 常州市103例新生儿先天性心脏病随访分析[J]. 临床儿科杂志, 2014(9): 839 – 842. doi: 10.3969/j.issn.1000-3606.2014.09.010 [12] 中华人民共和国卫生部. 中国妇幼卫生监测工作手册(2013版)[EB/OL]. (2013 – 08 – 30)[2023 – 04 – 07]. http://www.mchscn.cn/National-22/450.html. [13] 中国出生缺陷监测中心, 全国妇幼卫生监测办公室. 中国出生缺陷监测系统疑难和微小畸形报告指南 (试用版2012)[EB/OL]. (2012 – 11 – 01)[2023 – 04 – 07]. http://www.mchscn.cn/BirthDefectMonitoring-25/417.html. [14] 中华人民共和国卫生部. 卫生部关于印发《国家基本公共卫生服务规范(2011年版)》的通知[EB/OL]. (2011 – 05 – 24)[2023 – 04 – 07]. http://wjw.ningbo.gov.cn/art/2011/5/24/art_1229129177_49048520.html. [15] 宁波卫生计生委. 宁波市江北区《母子健康手册》试点工作全面启动[EB/OL]. (2016 – 05 – 17)[2023 – 04 – 07]. http://wjw.ningbo.gov.cn/art/2016/5/17/art_1229128343_48372139.html [16] Fernandes PS, Magalhães LR, Pezzini TR, et al. Congenital heart diseases trends in São Paulo State, Brazil: a national live birth data bank analysis[J]. World Journal of Pediatrics, 2022, 18(7): 472 – 481. doi: 10.1007/s12519-022-00543-3 [17] Zhang YC, Zhang W, Xu HY, et al. Epidemiological aspects, prenatal screening and diagnosis of congenital heart defects in Beijing[J]. Frontiers in Cardiovascular Medicine, 2021, 8: 777899. doi: 10.3389/fcvm.2021.777899 [18] 王瑞娟, 张文玲, 杨言军, 等. 生育政策改变对兰州市孕产妇及出生缺陷影响[J]. 中国公共卫生, 2022, 38(10): 1300 – 1304. doi: 10.11847/zgggws1136338 [19] Liu J, Song L, Qiu J, et al. Reducing maternal mortality in China in the era of the two - child policy[J]. BMJ Global Health, 2020, 5(2): e002157. doi: 10.1136/bmjgh-2019-002157 [20] Zhang XH, Sun Y, Zhu JJ, et al. Epidemiology, prenatal diagnosis, and neonatal outcomes of congenital heart defects in eastern China: a hospital - based multicenter study[J]. BMC Pediatrics, 2020, 20(1): 416. doi: 10.1186/s12887-020-02313-4 [21] Qu YJ, Liu XQ, Zhuang J, et al. Incidence of congenital heart disease: the 9 - year experience of the Guangdong registry of congenital heart disease, China[J]. PLoS One, 2016, 11(7): e0159257. doi: 10.1371/journal.pone.0159257 [22] 潘飞霞, 徐玮泽, 李嘉斌, 等. 中国先天性心脏病疾病负担三十年变化分析[J]. 浙江大学学报(医学版), 2022, 51(3): 267 – 277. [23] 赵一鸣, 史慧静. 出生缺陷监测进展[J]. 实用预防医学, 2019, 26(1): 125 – 127. [24] 赵一鸣, 史慧静, 袁芳. 宁波市江北区2010 — 2015年基于孕产人群队列的子代生后2年内出生缺陷发病率研究[J]. 中国儿童保健杂志, 2019, 27(4): 381 – 384. [25] 庞林鸿, 卢江, 宋宏琳, 等. 中国西部8省 ≤ 18岁儿童先天性心脏病不同延迟诊断时长与术后康复结果对比分析[J]. 中国公共卫生, 2022, 38(6): 671 – 675. [26] 陈瑜, 陈梅. 南京地区学龄前儿童先天性心脏病的患病情况调查与发病因素分析[J]. 实用预防医学, 2020, 27(11): 1384 – 1387. [27] Sun PF, Ding GC, Zhang MY, et al. Prevalence of congenital heart disease among infants from 2012 to 2014 in Langfang, China[J]. Chinese Medical Journal, 2017, 130(9): 1069 – 1073. doi: 10.4103/0366-6999.204923 [28] 江玮, 徐昊立. 广东省围产儿先天性心脏病的类型及转归分析[J]. 中国优生与遗传杂志, 2020, 28(6): 755 – 756. doi: 10.13404/j.cnki.cjbhh.2020.06.033 [29] 许文婧, 魏莉莉, 王莹, 等. 我国围生儿先天性心脏病发病率的Meta分析[J]. 国际生殖健康/计划生育杂志, 2020, 39(4): 269 – 275. [30] Hoffman JIE, Kaplan S. The incidence of congenital heart disease[J]. Journal of the American College of Cardiology, 2002, 39(12): 1890 – 1900. doi: 10.1016/S0735-1097(02)01886-7 [31] Pfitzer C, Helm PC, Ferentzi H, et al. Changing prevalence of severe congenital heart disease: results from the National Register for Congenital Heart Defects in Germany[J]. Congenital Heart Disease, 2017, 12(6): 787 – 793. doi: 10.1111/chd.12515 [32] Egbe A, Uppu S, Stroustrup A, et al. Incidences and sociodemographics of specific congenital heart diseases in the United States of America: an evaluation of hospital discharge diagnoses[J]. Pediatric Cardiology, 2014, 35(6): 975 – 982. doi: 10.1007/s00246-014-0884-8 [33] Yuan Z, Zhang LZ, Li B, et al. Investigation of echocardiographic characteristics and predictors for persistent defects of patent foramen ovale or patent ductus arteriosus in Chinese newborns[J]. Biomedical Journal, 2021, 44(2): 209 – 216. doi: 10.1016/j.bj.2019.12.007 [34] 李杏. 单纯性室间隔缺损胎儿羊水多肽组学分析及其自然闭合影响因素的研究[D]. 南京: 南京医科大学, 2016. [35] Zhao QM, Niu C, Liu F, et al. Spontaneous closure rates of ventricular septal defects (6, 750 consecutive neonates)[J]. The American Journal of Cardiology, 2019, 124(4): 613 – 617. doi: 10.1016/j.amjcard.2019.05.022 [36] Chang JK, Jien WY, Chen HL, et al. Color Doppler echocardiographic study on the incidence and natural history of early - infancy muscular ventricular septal defect[J]. Pediatrics amd Neonatology, 2011, 52(5): 256 – 260. doi: 10.1016/j.pedneo.2011.06.003 [37] Barwad P, Prasad K, Dinakar B, et al. Percutaneous closure of small subpulmonic ventricular septal defect with an ADO I PDA occluder in a child[J]. Annals of Pediatric Cardiology, 2020, 13(4): 349. doi: 10.4103/apc.APC_159_19 [38] Bassareo PP, Calcaterra G, Deidda M, et al. Does oxygen content play a role in spontaneous closure of perimembranous ventricular Septal defects?[J]. Children, 2021, 8(10): 881. doi: 10.3390/children8100881 [39] 谢军. 室间隔缺损自然愈合相关因素研究[D]. 重庆: 重庆医科大学, 2013. [40] 徐睿宏, 李香伟, 周一凡, 等. 广西地区先天性心脏病防治工作十年回顾与展望[J]. 中国临床新医学, 2022, 15(9): 777 – 781. [41] 黄奕言, 姜柏生. 生育文化与医学风险: 高龄三胎的孕产经验分析[J]. 南京医科大学学报(社会科学版), 2021, 21(6): 567 – 571. [42] 钟文津, 杨炳昂, 景香香, 等. 超声随访小儿房间隔缺损及室间隔缺损的自然愈合[J]. 中国超声医学杂志, 2008, 24(11): 996 – 998. [43] Baeyens N, Bandyopadhyay C, Coon BG, et al. Endothelial fluid shear stress sensing in vascular health and disease[J]. The Journal of Clinical Investigation, 2016, 126(3): 821 – 828. doi: 10.1172/JCI83083 [44] Uygur A, Lee RT. Mechanisms of cardiac regeneration[J]. Developmental Cell, 2016, 36(4): 362 – 374. doi: 10.1016/j.devcel.2016.01.018 [45] Ali SR, Hippenmeyer S, Saadat LV, et al. Existing cardiomyocytes generate cardiomyocytes at a low rate after birth in mice[J]. Proceedings of the National Academy of Sciences of the United States of America, 2014, 111(24): 8850 – 8855. [46] Han PD, Bloomekatz J, Ren J, et al. Coordinating cardiomyocyte interactions to direct ventricular chamber morphogenesis[J]. Nature, 2016, 534(7609): 700 – 704. doi: 10.1038/nature18310 -
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