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Prevalence of Hirschsprung's disease in premature infants: a systematic review

Johannes W Duess1, Alejandro D Hofmann, Prem Puri

  • 1National Children's Research Centre, Our Lady's Children's Hospital, Crumlin, Dublin 12, Ireland.

Pediatric Surgery International
|July 3, 2014
PubMed

Insights

Hirschsprung's disease (HD) is more prevalent in premature infants than previously thought, with recent data showing a 14% occurrence. Consider HD in preterm infants with intestinal obstruction symptoms.

Area of Science:

  • Neonatalogy
  • Pediatric Surgery
  • Gastroenterology

Background:

  • Intestinal dysmotility in preterm infants is often linked to an immature enteric nervous system.
  • Hirschsprung's disease (HD) is considered rare in premature infants, with limited data on its exact prevalence.
  • Classical HD signs can be obscured by prematurity symptoms, complicating diagnosis.

Purpose of the Study:

  • To systematically review and determine the prevalence and presentation of Hirschsprung's disease (HD) in premature infants.
  • To document the changing prevalence of HD in preterm infants over time.
  • To identify key clinical features of HD in this population.

Main Methods:

  • A systematic literature review was conducted using keywords related to Hirschsprung's disease and prematurity.
  • Publications reporting on infants born before 37 weeks gestation or described as preterm were included.
  • Data on epidemiology and morbidity were extracted and analyzed.

Main Results:

  • Twenty-six studies from 1964-2013 reported on premature infants with HD, totaling 257 cases out of 4,147 infants (6% prevalence).
  • Prevalence increased from 5% (1964-1999) to 14% (2000-2013).
  • Total colonic aganglionosis occurred in 13% of preterm infants; common symptoms included abdominal distension (80%), delayed meconium passage (57%), and bile-stained vomiting (37%).

Conclusions:

  • The prevalence of Hirschsprung's disease (HD) in premature infants has notably increased in recent years.
  • Hirschsprung's disease should be a strong consideration for preterm infants exhibiting signs of intestinal obstruction.
  • Early recognition and diagnosis are crucial for managing HD in this vulnerable population.
Abstract

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