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Exstrophy variants: should they be considered malformation complexes separate from classic exstrophy?

D K Gupta1, A R Charles, M Srinivas

  • 1Department of Pediatric Surgery, All India Institute of Medical Sciences New Delhi, India. devendra6@hotmail.com

European Journal of Pediatric Surgery : Official Journal of Austrian Association of Pediatric Surgery ... [Et Al] = Zeitschrift Fur Kinderchirurgie
|January 27, 2004
PubMed
Summary

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Exstrophy variants, rare congenital anomalies, frequently present with other malformations, suggesting distinct causes from classical bladder exstrophy. Further research is needed to understand these unique developmental origins.

Area of Science:

  • Pediatric Surgery
  • Congenital Malformations
  • Urology

Background:

  • Exstrophy variants are rare congenital anomalies with a generally better prognosis than classical bladder exstrophy.
  • This study focuses on superior vesical fissure (SVF), a type of exstrophy variant.

Observation:

  • The authors encountered two unique cases: SVF with esophageal atresia and tracheoesophageal fistula (EATEF), and SVF with severe limb anomalies.
  • A retrospective review of 9 exstrophy variant cases diagnosed between 1989 and 2000 was conducted.

Findings:

  • Seven out of nine exstrophy variant cases (78%) exhibited significant associated malformations.
  • These included EATEF, urethral atresia, absent radius, large umbilical hernia, low or high anorectal malformation, and true diphallus with bifid scrotum.

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Implications:

  • The high frequency of associated anomalies in exstrophy variants challenges their classification alongside classical bladder exstrophy.
  • Further investigation into these cases may reveal distinct dysembryogenic mechanisms and etiologies for bladder exstrophy variants.