Hirschsprung's disease prevalence in Europe: a register based study.
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Best Kate E, Addor Marie-Claude, Arriola Larraitz, Balku Eszter, Barisic Ingeborg, Bianchi Fabrizio, Calzolari Elisa, Curran Rhonda, Doray Berenice, Draper Elizabeth, Garne Ester, Gatt Miriam, Haeusler Martin, Bergman Jorieke, Khoshnood Babak, Klungsoyr Kari, Martos Carmen, Materna-Kiryluk Anna, Matias Dias Carlos, McDonnell Bob, Mullaney Carmel, Nelen Vera, O'Mahony Mary, Queisser-Luft Annette, Randrianaivo Hanitra, Rissmann Anke, Rounding Catherine, Sipek Antonin, Thompson Rosie, Tucker David, Wellesley Diana, Zymak-Zakutnia Natalya, Rankin Judith
Аннотация
METHODS: Cases of Hirschsprung's disease delivered during 1980 to 2009 notified to 31 European Surveillance of Congenital Anomaly registers formed the population-based case-series. Prevalence rates and 95% confidence intervals were calculated as the number of cases per 10,000 births. Multilevel Poisson regression was performed to investigate trends in prevalence, geographical variation and the association with maternal age.
RESULTS: There were 1,322 cases of Hirschsprung's disease among 12,146,210 births. The total prevalence was 1.09 (95% confidence interval, 1.03-1.15) per 10,000 births and there was a small but significant increase in prevalence over time (relative risk = 1.01; 95% credible interval, 1.00-1.02; p = 0.004). There was evidence of geographical heterogeneity in prevalence (p < 0.001). Excluding 146 (11.0%) cases with chromosomal anomalies or genetic syndromes, there were 1,176 cases (prevalence = 0.97; 95% confidence interval, 0.91-1.03 per 10,000 births), of which 137 (11.6%) had major structural anomalies. There was no evidence of a significant increased risk of Hirschsprung's disease in cases born to women aged ≥35 years compared with those aged 25 to 29 (relative risk = 1.09; 95% credible interval, 0.91-1.31; p = 0.355).
CONCLUSION: This large population-based study found evidence of a small increasing trend in Hirschsprung's disease and differences in prevalence by geographic location. There was also no evidence of an association with maternal age.