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A newborn fails to pass meconium in the first 48 hours and develops abdominal distension. Contrast enema shows a transition zone in the rectosigmoid. Rectal biopsy confirms absent ganglion cells.

Complex/multifactorial or syndromic

  • RET variants (AD with incomplete penetrance) - most common single gene
  • Associated syndromes: Down syndrome, Waardenburg type 4, Mowat-Wilson, CCHS
  • Absent enteric ganglion cells (aganglionic segment)
  • Failure to pass meconium, constipation, abdominal distension
  • Risk of enterocolitis
  • Short segment (rectosigmoid) vs. long segment
  • Contrast enema suggests the diagnosis (transition zone, rectosigmoid index)
  • Rectal suction biopsy is the gold standard (absent ganglion cells, hypertrophied nerve fibers)
  • Anorectal manometry shows absent rectoanal inhibitory reflex
  • Surgical pull-through (resect aganglionic segment, anastomose ganglionic bowel to anus)
  • Decompression and bowel irrigation pre-operatively to relieve obstruction
  • Monitor for and treat Hirschsprung-associated enterocolitis (a major source of morbidity)
  • Long-term follow-up for constipation, soiling, and continence

RET = REcTum: RET is the major susceptibility gene for isolated Hirschsprung disease (RET = REcTum). Hirschsprung disease results from failure of neural crest cell migration. "His-chsprung" is more common in males (5:1 M:F ratio), a "He" disease (like Hemochromatosis and Hemophilia).

Hirschsprung disease is a useful example to group with other non-X-linked disorders that more commonly affect males. "His-chsprung" contains "His," highlighting the male predominance.

Four non-X-linked disorders more common in males: Hemochromatosis, LHON (HOmbres), Brugada (Bros), Hirschsprung disease (His-chsprung)
Four non-X-linked disorders more common in males: Hemochromatosis, LHON (HOmbres), Brugada (Bros), Hirschsprung disease (His-chsprung)