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      Prevalence of prenatally diagnosed congenital cystic adenomatoid malformation among fetuses in China

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          Abstract

          The prevalence of congenital cystic adenomatoid malformation among fetuses still varies in different studies in China. The present meta-analysis was intended to evaluate the pooled prevalence of fetuses in China. Four English (Pubmed, Elsevier Science Direct, Web of Science and the Cochrane Library) and four Chinese (the Chinese Biological Medical Literature database, the Chinese National Knowledge Infrastructure database, VIP database and Wanfang Data) databases were searched from inception to July 2016. Meta-analyses were performed using Stata (version 12.0), with prevalence and corresponding 95% confidence intervals using the random effect model. Five studies with 393496 fetuses were chosen for this meta-analysis. The overall pooled prevalence was 4.01/10000 (2.03/10000 - 6.00/10000) fetuses. Sensitivity analysis revealed that the results were stable, and Begg’s test and Egg’s test showed no potential risk of publication bias. This is, to our knowledge, the first study to systematically evaluate the literature of the prevalence of congenital cystic adenomatoid malformation among fetuses in China. Results showed that the prevalence among fetuses should be considerable. A large-scale multicenter study on the epidemiology across different areas in China is required.

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          Congenital cystic adenomatoid malformation of the lung. Classification and morphologic spectrum.

          Thirty-eight cases of congenital cystic adenomatoid malformation of the lung are described, and a classification based on clinical, gross, and microscopic criteria is proposed. The type I lesion is composed of single or multiple large cysts (more than 2 cm. in diameter), frequently producing mediastinal herniation. The cysts are lined by ciliated psuedostratified columnar epithelium. The walls of the cysts contain prominent smooth muscle and elastic tissue. Mucus producing cells are present in approximatley one-third of the cases, and cartilage in the wall is rarely seen. Relatively normal alveoli may be seen between the cysts. The prognosis is good. Radiographic analysis of the type I lesion can preoperatively suggest the diagnosis, especially with the typical multicystic pattern. The gross appearance of the lesion corresponds closely to the radiographic image and adds another dimension to the pathologist's evaluation of the disease. The type II lesion is composed of multiple small cysts (less than 1 cm. in diameter) lined by ciliated cuboidal to columnar epithelium. Structures resembling respiratory bronchioles and distended alveoli are present between the epithelium lined cysts. Mucous cells and cartilage are not present. Striated muscle fibers may be seen rarely. The type II lesion is associated with a high frequency of other congenital anomalies, and the prognosis is poor. The type III lesion is a large, bulky noncystic lesion producing mediastinal shift. Bronchiole-like structures are lined by ciliated cuboidal epithelium and separated by masses of alveolus-sized structures lined by nonciliated cuboidal epithelium. The prognosis is poor.
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            Birth defects surveillance in China.

            Birth defects are a global public health problem because of their large contribution to infant mortalities and disabilities. It is estimated that 4%-6% of Chinese newborns are affected by birth defects every year. Surveillance is a basic approach to understanding the occurrence and associated factors of birth defects. The Ministry of Health of China initiated a national hospital-based birth defects monitoring system 20 years ago. Nearly every province in this country has established its own surveillance system in the past. The authors reviewed the result of the monitoring system at different administrative levels in China. Available publications on the surveillance of birth defects and data from national and provincial birth defects surveillance systems were reviewed to evaluate the effectiveness of the surveillance systems. According to the 2009 data, the national hospital-based birth defects surveillance system monitored over 1.3 million births, which accounted for more than 8% of births in China. In addition, 30 provincial hospital-based surveillance programs covered a birth population of more than 3.6 million (22% of births in China). Great achievements have been made in terms of case ascertainment, data quality control, and online reporting. But the surveillance systems in China still have some limitations. A short ascertainment period may miss some internal anomalies, inherited metabolic diseases, and malformed fetus aborted before the 28th gestational week. Discrepancies in antenatal or postnatal diagnosis of birth defects between surveillance institutes may affect the detection rate and introduce biases. Absence of baseline data and lack of integrated database systems limit the application of surveillance data to etiological studies and affect the process of decision-making. The surveillance system for birth defects is prerequisite to propose, conduct and assess any interventions for the disease. To meet the need of study and prevention of birth defects, measures should be taken to improve the national and provincial birth defects surveillance systems in China.
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              Epidemiology of Birth Defects Based on a Birth Defect Surveillance System from 2005 to 2014 in Hunan Province, China

              Objective To describe the epidemiology of birth defects (BDs) in perinatal infants in Hunan Province, China, between 2005 and 2014. Methods The BD surveillance data of perinatal infants (for stillbirth, dead fetus or live birth between 28 weeks of gestation and 7 days after birth) were collected from 52 registered hospitals of Hunan between 2005 and 2014. The prevalence rates of BDs with 95% confidence interval (CI) and crude odds ratio (ORs) were calculated to examine the associations of infant gender, maternal age, and region (urban vs rural) with BDs. Results From 2005 to 2014, there were a total of 925413 perinatal infants of which 17753 had BDs, with the average prevalence of 191.84 per 10000 PIs (perinatal infants), showing a significant uptrend. The risks of BDs are higher in urban areas versus rural areas (OR = 1.20), in male infants versus female infants (OR = 1.19), and in mothers above age 35 versus those below age 35 (OR = 1.24). The main five types of BDs are Congenital heart defects (CHD), Other malformation of external ear (OMEE), Polydactyly, Congenital malformation of kidney (CMK), and Congenital talipes equinovarus (CTE). From 2005 to 2014, the prevalence rates (per 10000 PIs) of CHD and CMK increased significantly from 22.56 to 74 (OR = 3.29, 95%CI: 2.65–4.11) and from 7.61 to 14.62 (OR = 1.92, 95%CI:1.30–2.84), respectively; the prevalence rates of congenital hydrocephalus and neural tube defects (NTDs) decreased significantly from 11.8 to 5.29 (OR = 0.45, 95%CI: 0.31–0.65) and from 7.87 to 1.74 (OR = 0.22, 95%CI: 0.13–0.38), respectively. Conclusions The prevalence rates of specific BDs in perinatal infants in Hunan have changed in the last decade. Urban pregnant women, male perinatal infants, and mothers above age 35 present different prevalence rates of BDs. Wider use of new diagnosis technology, improving the ability of monitoring, strengthening the publicity and education are important to reduce the prevalence of BDs.
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                Author and article information

                Journal
                Oncotarget
                Oncotarget
                Oncotarget
                ImpactJ
                Oncotarget
                Impact Journals LLC
                1949-2553
                3 October 2017
                20 June 2017
                : 8
                : 45
                : 79587-79593
                Affiliations
                1 Foshan Institute of Fetal Medicine, Southern Medical University Affiliated Maternal & Child Health Hospital of Foshan, Foshan, Guangdong, 528000, China
                2 Department of Obstetrics, Southern Medical University Affiliated Maternal & Child Health Hospital of Foshan, Foshan, Guangdong, 528000, China
                3 Department of Epidemiology and Biostatistics, School of Public Health, Anhui Medical University, Hefei, Anhui, 230032, China
                4 Department of Library, The First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, Zhejiang, 310003, China
                5 Department of Maternal, Child and Adolescent Health, School of Public Health, Anhui Medical University, Hefei, Anhui, 230032, China
                6 School of Integrated Traditional and Western Medicine, Anhui University of Chinese Medicine, Hefei, Anhui, 230038, China
                Author notes
                Correspondence to: Zhengping Liu, liuzphlk81@ 123456outlook.com
                [*]

                These authors have contributed equally to this work and should be considered co-first author

                Article
                18579
                10.18632/oncotarget.18579
                5668071
                e95f6fc0-de69-423d-846c-6f488197db12
                Copyright: © 2017 Fan et al.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License 3.0 (CC BY 3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                History
                : 12 August 2016
                : 21 May 2017
                Categories
                Clinical Research Paper

                Oncology & Radiotherapy
                congenital cystic adenomatoid malformation,fetuses,prevalence,meta-analysis

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