An international collaborative study of the epidemiology of esophageal atresia or stenosis

Robert, Elisabeth; Mutchinick, Osvaldo M.; Mastroiacovo, Pierpaolo; Knudsen, Lisbeth B.; Daltveit, Anne Kjersti; Castilla, Eduardo E.; Lancaster, Paul; Källén, Bëngt; Cocchi, Guido

doi:10.1016/0890-6238(93)90085-l

Cited by 79 publications

(88 citation statements)

References 27 publications

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“…However, sensitivity analysis limiting data to member programs with optimal case ascertainment revealed an almost identical total prevalence (2.47). The findings are also similar to an earlier study of EA among nine ICBDSR programs reporting a prevalence of 2.56 per 10,000 births from 1965 to 1989 (Robert et al, 1993).…”

Section: Discussionsupporting

confidence: 90%

“…In addition to maternal ethnicity and geographic location, previous studies have reported EA to be associated with maternal age (Leck et al, 1968;Harris et al, 1995), multiple gestation (Harris et al, 1995;Riley et al, 1998), infant sex (Robert et al, 1993) and use of assisted reproductive technology (Reefhuis et al, 2009). In a recent study of EA in the United States, women using assisted reproductive technology had a 4.5-fold increased risk of having an infant with EA (Reefhuis et al, 2009).…”

Section: Discussionmentioning

confidence: 99%

“…The underlying etiology of EA has been described as multifactorial and is likely to differ across settings (Robert et al, 1993;Felix et al, 2009;de Jong et al, 2010b). In addition to maternal ethnicity and geographic location, previous studies have reported EA to be associated with maternal age (Leck et al, 1968;Harris et al, 1995), multiple gestation (Harris et al, 1995;Riley et al, 1998), infant sex (Robert et al, 1993) and use of assisted reproductive technology (Reefhuis et al, 2009).…”

Section: Discussionmentioning

confidence: 99%

“…Differences in lifestyle factors, such as smoking or dietary habits within countries or regional variation, may also affect estimates. However, lack of temporal trends among the included programs suggests environmental factors, which change over time, are less likely to play a role in the development of EA (Robert et al, 1993;Canfield et al, 2006;Garne et al, 2007). Underlying and still unknown (Genevieve et al, 2011) genetic susceptibility, biologic or physical differences may also modify risk of exposures.…”

Section: Discussionmentioning

confidence: 99%

See 3 more Smart Citations

Prevalence of esophageal atresia among 18 international birth defects surveillance programs

Nassar

Leoncini²,

Amar³

et al. 2012

Birth Defects Research

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130

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Section: Discussionsupporting

confidence: 90%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

Prevalence of esophageal atresia among 18 international birth defects surveillance programs

Nassar

Leoncini²,

Amar³

et al. 2012

Birth Defects Research

Self Cite

130

View full text Add to dashboard Cite

show abstract

“…Quality control of the collected information is routinely performed, and the accumulated data are highly reliable. Several joint investigations were performed in association with American and European registries [4, 5, 6, 7]. The quality of any clinical-epidemiological program is necessarily limited by the accuracy of the clinical diagnoses.…”

Section: From Clinical Epidemiology To State-of-the-art Technologymentioning

confidence: 99%

ECLAMC: The Latin-American Collaborative Study of Congenital Malformations

Castilla

Orioli

2004

Public Health Genomics

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226

212

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Definition: ECLAMC (‘Estudio Colaborativo Latino Americano de Malformaciones Congénitas’) is a program for the clinical and epidemiological investigation of risk factors in the etiology of congenital anomalies in Latin-American hospitals, using a case-control methodological approach. It is a voluntary agreement among professionals lacking institutional base as well as designated budgets. ECLAMC has been usually funded by research-funding agencies rather than public health ministries. The National Research Councils of Argentina and Brazil have been the main sources of support during its 36 years of existence. Since vital and health statistics are unreliable in South America, ECLAMC collects all the information required for the denominators in a hospital-based sample of births. ECLAMC can be defined as a continental network of persons interested in research and prevention of birth defects. History and Evolution: From the institutional point of view, ECLAMC has had headquarters in diverse centers of Argentina and Brazil, but always as an independent research project, without a defined administrative link. ECLAMC began operating in 1967, as an investigation limited to the city of Buenos Aires, Argentina, and it gradually expanded until covering all the 10 countries of South America as well as Costa Rica and the Dominican Republic. Even though ECLAMC has maintained essentially the same original experimental design since 1967, due to the data accumulated by the program, the increasing experience as well as the development in science, technical modifications occurred including a DNA bank and a fully informatized data handling system. Since 1974 ECLAMC has been a founder member of the International Clearinghouse for Birth Defects Monitoring Systems; since 1994 a WHO Collaborating Center for the Prevention of Congenital Malformations, and since 2000 a collaborating member of the NIH Global Netwok for Women’s and Children’s Health Research. Methodology: The maternity hospital network of ECLAMC examines around 200,000 births per year. All major and minor anomalies diagnosed at birth in infants weighing 500 g or more are registered according to a manual of procedures. The next non-malformed baby of the same sex born in the same hospital is selected as a control subject for each case. Thus, a one-to-one healthy control group matched by sex, time and place of birth is obtained. As a system of epidemic surveillance, ECLAMC systematically observes the fluctuations in the frequencies of different malformations and, in the case of an alarm for a probable epidemic of a given malformation, at a given moment, and given area, it acts to identify its cause. As termination of pregnancy has severe legal restrictions in South America, prevention of birth defects should concentrate on primary, preconceptional and tertiary measures. Tertiary measures aim to avoid complications of the affected patients from the medical, psychological, and social standpoints.

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VATERL: An epidemiologic analysis of risk factors

Rittler¹,

Paz

Castilla

1997

Am. J. Med. Genet.

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This work analyzed the incidence of risk factors in 138 cases presenting two or more of five components defining VATERL, with no other recognized unrelated anomalies: vertebral anomalies, anal atresia, esophageal atresia with or without tracheoesophageal fistula, renal anomalies, and preaxial defects of the upper limbs, including polydactyly of the thumb. The 138 infants were ascertained among 1,811,461 births examined in the 1967-1994 period by the Latin-American Collaborative Study of Congenital Malformations: ECLAMC. One healthy and one malformed control newborn infant were matched to each VATERL case. The birth prevalence rates (per 100,000 births) for VATERL were significantly lower in Venezuela (3.1) than in the other eight countries (8.8) (P < 0.001). Venezuela also had lower rates for all five VATERL defects, even after excluding the 138 VATERL cases. VATERL cases were preferentially males (male proportion 0.6261) (P < 0.02), and, when compared with healthy controls, they had a higher perinatal mortality rate (63.7%) (P < 0.005), a higher frequency of fetal losses in previous pregnancies (12.6%) (P < 0.05), and lower mean birthweights (2,361.79 +/- 809.63 g) (P < 0.005). VATERL cases showed a higher rate than matched malformed controls for prenatal exposures to drugs and physical agents (P < 0.02 and P < 0.05, respectively), although no specific pharmacological or physical group was involved. The lower birth prevalence rates found in Venezuela, for VATERL as well as for each of the five congenital anomalies involved in this association, seem to be biologically meaningful. Since we could not identify a potential risk factor, nor a common cause of underascertainment unique to the Venezuelan subsample and common to all six hospitals, no hypothesis can be advanced here for this phenomenon. Nevertheless, this unequal geographic distribution strongly suggests a common etiopathogenicity for the five congenital anomalies involved in the VATERL association.

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An international collaborative study of the epidemiology of esophageal atresia or stenosis

Cited by 79 publications

References 27 publications

Prevalence of esophageal atresia among 18 international birth defects surveillance programs

Prevalence of esophageal atresia among 18 international birth defects surveillance programs

ECLAMC: The Latin-American Collaborative Study of Congenital Malformations

VATERL: An epidemiologic analysis of risk factors

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