High Prevalence of Unrecognized Congenital Heart Disease in School-Age Children in Rural China: A Population-Based Echocardiographic Screening Study

HomeCirculationVol. 144, No. 23High Prevalence of Unrecognized Congenital Heart Disease in School-Age Children in Rural China: A Population-Based Echocardiographic Screening Study Free AccessLetterPDF/EPUBAboutView PDFView EPUBSections ToolsAdd to favoritesDownload citationsTrack citationsPermissions ShareShare onFacebookTwitterLinked InMendeleyRedditDiggEmail Jump toFree AccessLetterPDF/EPUBHigh Prevalence of Unrecognized Congenital Heart Disease in School-Age Children in Rural China: A Population-Based Echocardiographic Screening Study Quming Zhao, MD, Hongyan Chen, PhD, Guowen Zhang, BM, Weicheng Chen, MD, Bing Jia, MD, Fang Liu, MD, Xiaojing Ma, MD, Weili Yan, PhD, Conway Niu, MD and Guoying Huang, MD Quming ZhaoQuming Zhao Pediatric Heart Center (Q.Z., W.C., B.J., F.L., X.M., G.H.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. Shanghai Key Laboratory of Birth Defects, China (Q.Z., W.C., B.J., F.L., X.M., G.H.). *Q. Zhao and H. Chen contributed equally. Search for more papers by this author , Hongyan ChenHongyan Chen https://orcid.org/0000-0003-2264-3034 Department of Clinical Epidemiology and Clinical Trial Unit (H.C., W.Y.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. *Q. Zhao and H. Chen contributed equally. Search for more papers by this author , Guowen ZhangGuowen Zhang Pediatric Department, Luchun County People’s Hospital, Yunnan Province, China (G.Z.). Search for more papers by this author , Weicheng ChenWeicheng Chen Pediatric Heart Center (Q.Z., W.C., B.J., F.L., X.M., G.H.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. Shanghai Key Laboratory of Birth Defects, China (Q.Z., W.C., B.J., F.L., X.M., G.H.). Search for more papers by this author , Bing JiaBing Jia https://orcid.org/0000-0002-6560-3991 Pediatric Heart Center (Q.Z., W.C., B.J., F.L., X.M., G.H.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. Shanghai Key Laboratory of Birth Defects, China (Q.Z., W.C., B.J., F.L., X.M., G.H.). Search for more papers by this author , Fang LiuFang Liu Pediatric Heart Center (Q.Z., W.C., B.J., F.L., X.M., G.H.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. Shanghai Key Laboratory of Birth Defects, China (Q.Z., W.C., B.J., F.L., X.M., G.H.). Search for more papers by this author , Xiaojing MaXiaojing Ma Pediatric Heart Center (Q.Z., W.C., B.J., F.L., X.M., G.H.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. Shanghai Key Laboratory of Birth Defects, China (Q.Z., W.C., B.J., F.L., X.M., G.H.). Search for more papers by this author , Weili YanWeili Yan https://orcid.org/0000-0002-7633-7449 Department of Clinical Epidemiology and Clinical Trial Unit (H.C., W.Y.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. Search for more papers by this author , Conway NiuConway Niu Department of General Paediatrics, Perth Children’s Hospital, Australia (C.N.). Search for more papers by this author and Guoying HuangGuoying Huang Correspondence to: Guoying Huang, MD, Pediatric Heart Center, Children’s Hospital of Fudan University, National Children’s Medical Center, 399 Wan Yuan Road, Shanghai 201102, China. Email E-mail Address: [email protected] https://orcid.org/0000-0001-6002-2226 Pediatric Heart Center (Q.Z., W.C., B.J., F.L., X.M., G.H.), Children’s Hospital of Fudan University, National Children’s Medical Center, Shanghai, China. Shanghai Key Laboratory of Birth Defects, China (Q.Z., W.C., B.J., F.L., X.M., G.H.). Search for more papers by this author Originally published6 Dec 2021https://doi.org/10.1161/CIRCULATIONAHA.121.056455Circulation. 2021;144:1896–1898Timely diagnosis of congenital heart disease (CHD) is pivotal for early treatment and reduction of complications but is still challenging in rural settings because of limited access to health care.1 Accurate estimates of unrecognized CHD in school-age children could contribute to quantifying unmet health needs for diagnosis and treatment. Ideally, this could be done using population-based screening by echocardiography. Completed as part of the Health Poverty Alleviation Project in China, we provided such an estimate in a selected rural area and identified factors associated with this phenomenon.We did a population-based echocardiographic screening study of all school children aged 5 to 18 years in 1 town and 4 townships in Luchun County, one of the most deprived counties located in southern Yunnan province, China, between December 1, 2019, and November 31, 2020. All echocardiographic scans were performed by the same pediatric cardiologist at each school using a Philips CX50 ultrasound machine. A second pediatric cardiologist was on site to double-check the findings of each echocardiogram. Images and loops of echocardiograms without concordant judgment and those confirmed as CHD were ascertained by 3 other pediatric cardiologists via video conferencing. Unrecognized CHD was defined as patients with CHD diagnosed for the first time during the echocardiographic screening and was divided into severe (defects that cause heart failure symptoms), moderate (defects that are not yet causing heart failure symptoms but are physically appreciable and require treatment), and mild (defects that may not be physically appreciable and often do not require treatment). This study was approved by the Ethics Committee of the Children’s Hospital of Fudan University (Institutional Review Board No. 2020132). Oral informed consent was obtained from the caregivers of the participating children. The data that support the findings of this study are available from the corresponding author on reasonable request.A total of 21 861 children were screened, accounting for 99.2% of all school-age children in the study areas. The mean (SD) age was 11.9 (3.6) years, 52.3% were boys, and 88.8% were of Hani ethnicity. CHD was identified in 285 children, with an overall prevalence of 13.0 per 1000 (95% CI, 11.6–14.6). Among them, 252 were unrecognized CHD (mild, 27.4%; moderate, 60.7%; severe, 11.9%), yielding a prevalence of 11.5 per 1000 (95% CI, 10.2–13.0) (Figure). Atrial septal defect accounted for 63.1% of the unrecognized CHD, followed by patent ductus arteriosus (10.3%), abnormalities of tricuspid valve (7.5%), and ventricular septal defect (7.1%). In multivariable logistic regression analyses, high-poverty area and poor household were significantly associated with unrecognized CHD (odds ratio, 2.34 [95% CI, 1.01–5.45] and 4.16 [95% CI, 1.64–10.40]), whereas ethnicity was not (1.81 [95% CI, 0.51–6.43]). With the addition of 3 recognized but unrepaired severe CHD, the prevalence of unrepaired CHD was 11.7 per 1000 (10.3–13.2). Among 186 children in need of intervention, 80 (43.0%) of their caregivers refused to receive treatments because of a lack of disease awareness.Download figureDownload PowerPointFigure. Diagnosis, severity, and repaired conditions of the 285 patients with CHD; spectrum of unrecognized CHD and its associated risk factors. The sunburst chart (top left) shows the hierarchical relationships between recognized status (innermost), repaired status (middle), and disease severity (outer) of the overall 285 patients with CHD. The innermost circle for recognized status serves as the top of the hierarchy. Segments of this ring serve as the parent ring segments for the sequential ring (ie, repaired status), and so repaired status for disease severity. Top right, The spectrum of the 252 patients with unrecognized CHD. Bottom right, Risk factors associated with the likelihood of being unrecognized. Proportions were omitted for continuous variables. We performed binary logistic regression analyses within all children identified with CHD (N=285), with unrecognized CHD as the primary outcome (N=252). All the listed risk factors were included in the model simultaneously to explore their independent associations with unrecognized CHD. Body mass index (BMI) was calculated as weight in kilograms divided by the square of height in meters (kg/m2). Because the age of the children varied, BMI was converted to age and sex SD scores (z scores) based on the Growth Curve of Children and Adolescents in China. Poor household was defined as a household that had CNY <3000 or USD <464 net income per year, and high-poverty area as an area with ≥20% populations being poor households. Statistical analyses were performed by R version 4.0.2 (R Development Core Team, Vienna, Austria). A 2-sided P value <0.05 was considered statistically significant. BAV indicates bicuspid aortic valve; CHD, congenital heart disease; and CNY, Chinese yuan.This is the first echocardiographic screening with a total population study design to investigate the prevalence and associated factors for unrecognized CHD in children. Our accurate estimate was considerably higher than a global estimate (1.4 per 1000) primarily on the basis of traditional screening strategy (ie, clinical assessment followed by echocardiographic confirmation in suspected cases).2 We provide direct evidence for the relationships of living in poverty areas and poor households with unrecognized CHD, supporting that limited access to health care and lack of resources and referral systems probably lead to the missed diagnosis of CHD.3 Of note, there was no association between ethnicity and unrecognized CHD in our study population. Therefore, although our data from a selected rural area warrant further validation in different populations or ethnicities, we consider that socioeconomic deprivation underlies ethnic differences in CHD prevalence. Viewed in this light, our findings should be generalizable to other rural areas across populations.Although accurate for the detection of CHD in our contexts, population screening by standard echocardiography is less likely to be practical or affordable under most current healthcare systems. Given this, the traditional survey strategy would remain the mainstay of the screening modality. Nevertheless, the high prevalence of unrecognized CHD makes it imperative to improve the overall efficiency and accuracy of this modality in rural settings. This will hopefully be addressed by strengthening cardiovascular examination training in primary hospitals and increasing awareness of CHD among community doctors.4 In the near future, handheld echocardiography with its portability, ease of use, and accurate diagnosis of cardiac structure and function with relatively low cost may represent a promising cost-effective screening tool in resource-constrained areas.5In conclusion, unrecognized CHD was prevalently high in school-age children in a rural region in Yunnan, China, and was mainly composed of moderate and severe cases. This phenomenon is likely related to socioeconomic deprivation, as reflected by poor regional and family household status. Optimization of health care resource allocation in rural areas to facilitate screening and to improve access to care, and promotion of public health awareness of the disease burden, are urgent policy priorities in reducing the missed diagnosis of CHD.Article InformationAcknowledgmentsThe authors sincerely thank the Luchun County Government, Luchun Education Bureau, and teachers from the participating schools for their help in the screening work. We also acknowledge the staff of the Social Work Department at Children’s Hospital of Fudan University for arranging the transfer of patients, and the Nu Skin Children’s Heart Fund for their help in the treatment of patients.Sources of FundingThe study was funded by the National Key Research and Development Program of China (2016YFC1000506) and the Chinese Academy of Medical Sciences Research Unit (2018RU002).Disclosures None.Footnotes*Q. Zhao and H. Chen contributed equally.For Sources of Funding and Disclosures, see page 1898.Circulation is available at www.ahajournals.org/journal/circCorrespondence to: Guoying Huang, MD, Pediatric Heart Center, Children’s Hospital of Fudan University, National Children’s Medical Center, 399 Wan Yuan Road, Shanghai 201102, China. Email [email protected]edu.cnReferences1. GBD 2017 Congenital Heart Disease Collaborators. Global, regional, and national burden of congenital heart disease, 1990-2017: a systematic analysis for the global burden of disease study 2017.Lancet Child Adolesc Health. 2020; 4:185–200. doi:10.1016/S2352-4642(19)30402-XCrossrefMedlineGoogle Scholar2. Liu Y, Chen S, Zühlke L, Babu-Narayan SV, Black GC, Choy MK, Li N, Keavney BD. Global prevalence of congenital heart disease in school-age children: a meta-analysis and systematic review.BMC Cardiovasc Disord. 2020; 20:488. doi: 10.1186/s12872-020-01781-xCrossrefMedlineGoogle Scholar3. Knowles RL, Ridout D, Crowe S, Bull C, Wray J, Tregay J, Franklin RC, Barron DJ, Cunningham D, Parslow RC, et al.. Ethnic and socioeconomic variation in incidence of congenital heart defects.Arch Dis Child. 2017; 102:496–502. doi: 10.1136/archdischild-2016-311143CrossrefMedlineGoogle Scholar4. Zhao QM, Liu F, Wu L, Ma XJ, Niu C, Huang GY. Prevalence of congenital heart disease at live birth in China.J Pediatr. 2019; 204:53–58. doi: 10.1016/j.jpeds.2018.08.040CrossrefMedlineGoogle Scholar5. Chamsi-Pasha MA, Sengupta PP, Zoghbi WA. Handheld echocardiography: current state and future perspectives.Circulation. 2017; 136:2178–2188. doi: 10.1161/CIRCULATIONAHA.117.026622LinkGoogle Scholar Previous Back to top Next FiguresReferencesRelatedDetails December 7, 2021Vol 144, Issue 23Article InformationMetrics © 2021 American Heart Association, Inc.https://doi.org/10.1161/CIRCULATIONAHA.121.056455PMID: 34871107 Originally publishedDecember 6, 2021 Keywordscongenital heart diseaseechocardiographyPDF download Advertisement SubjectsEchocardiographyEpidemiologyPediatricsRisk Factors