Currarino's syndrome in twins presenting as neonatal intestinal obstruction--identical presentation in non-identical twins

BMJ Case Rep. 2014 Sep 8:2014:bcr2014204276. doi: 10.1136/bcr-2014-204276.

Abstract

We report a case of non-identical twins who presented with identical neonatal intestinal obstruction with features of anorectal stenosis, presacral mass and sacral anomaly consistent with Currarino's syndrome or triad. Plain sacral radiograph, contrast enema and MRI were diagnostic. Initial management involved a defunctioning colostomy followed by a posterior sagittal anorectoplasty with excision of the teratoma ± anterior sacral meningocele and finally closure of colostomy in a staged multidisciplinary approach. The twins' father is also affected with features of Currarino's syndrome but was diagnosed during family screening. Currarino's syndrome presenting with identical neonatal low intestinal obstruction in a non-identical set of twins is rare and interesting. Antenatal diagnosis of Currarino's syndrome is difficult and may prove to be a challenge even in the postnatal period. Sacral spine radiograph, contrast enema and MRI are diagnostic. Management requires high index of suspicion, low threshold for MRI and multidisciplinary staged approach.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple*
  • Anal Canal / abnormalities*
  • Anal Canal / diagnostic imaging
  • Anal Canal / surgery
  • Anorectal Malformations
  • Anus, Imperforate
  • Colostomy
  • Digestive System Abnormalities / complications
  • Digestive System Abnormalities / diagnosis*
  • Digestive System Abnormalities / diagnostic imaging
  • Digestive System Abnormalities / surgery
  • Diseases in Twins*
  • Female
  • Humans
  • Infant, Newborn
  • Infant, Newborn, Diseases*
  • Intestinal Obstruction / diagnosis*
  • Intestinal Obstruction / diagnostic imaging
  • Intestinal Obstruction / etiology
  • Intestinal Obstruction / surgery
  • Magnetic Resonance Imaging
  • Male
  • Meningocele
  • Radiography
  • Rectum / abnormalities*
  • Rectum / diagnostic imaging
  • Rectum / surgery
  • Sacrum / abnormalities*
  • Sacrum / diagnostic imaging
  • Sacrum / surgery
  • Spine / diagnostic imaging
  • Syndrome
  • Syringomyelia / complications
  • Syringomyelia / diagnosis*
  • Syringomyelia / diagnostic imaging
  • Syringomyelia / surgery
  • Teratoma / etiology
  • Teratoma / surgery
  • Twins, Dizygotic*

Supplementary concepts

  • Currarino triad