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Investigation of a temporal cluster of left sided congenital heart disease
Insights
A cluster of critical congenital heart disease cases in newborns was observed in rural New Jersey. Despite extensive investigation, no specific cause was identified for these congenital heart anomalies.
Area of Science:
- Epidemiology
- Neonatal Health
- Congenital Anomalies
Background:
- Observed a cluster of eight newborns with critical congenital heart disease (CHD) in a rural New Jersey unit during Oct-Nov 1977.
- Six of these cases involved left-sided lesions, representing 30% of all neonatal left-sided lesions seen over a three-year period.
- Temporal clustering of these congenital heart anomalies was statistically significant.
Purpose of the Study:
- To investigate a temporal cluster of critical congenital heart disease in newborns.
- To identify potential etiological factors associated with this cluster of congenital heart anomalies.
Main Methods:
- Conducted a case-control study matching an average of four controls to each case.
- Administered a detailed questionnaire to mothers of cases and controls covering various exposures (occupation, nutrition, environmental, lifestyle, medical history).
- Utilized scan statistics to assess temporal clustering of congenital heart disease cases.
Main Results:
- No statistically significant differences were found between cases and controls regarding maternal occupation, nutrition, radiation, chemical exposure, alcohol/tobacco use, medications, immunizations, or infections.
- The temporal scan statistic indicated a significant clustering of cases (p < 0.05).
Conclusions:
- The etiology of this cluster of critical congenital heart disease remains undetermined.
- Speculatively, the cluster might be linked to a common teratogenic agent, possibly influenza B, affecting conceptions in the winter of 1976/77 in the northeastern US, given contemporaneous trisomy clusters reported elsewhere.
- Further research is needed to explore potential environmental teratogenic exposures and their role in congenital heart anomalies.
Abstract:
During October and November 1977, eight newborns with critical congenital heart disease, six of these with left sided lesions, were admitted to a New Jersey newborn unit serving two predominantly rural counties with 9700 annual live births. The six left heart lesions (three cases of hypoplastic left heart, two of interrupted aortic arch, and one of aortic coarctation) represented 30% of all neonatal left sided lesions seen at the unit in the three year period 1976-8. The scan statistic for temporal clustering was significant (p less than 0.05). A case-control study was performed in which an average of four controls were matched to each case. A questionnaire was administered to the mothers of cases and controls concerning occupation, periconceptional and prenatal nutrition, radiation and chemical exposure, use of alcohol and tobacco, medications, immunisations, infections, and other exposures. No statistically significant differences between cases and controls were found on any of these items. Although no aetiology for this cluster of congenital heart anomalies could be found, it is of interest that three temporal clusters of fetuses or newborns with chromosomal trisomies have been reported in the medical literature whose conceptions were roughly contemporaneous with those of the infants in our series. These occurred in Rhode Island/Massachusetts, Maryland, and New York City. A speculative possibility is that these four point epidemics represented exposure to a common teratogenic agent, perhaps influenza B, in the winter of 1976/77 in the northeastern United States.