Abstract:
BACKGROUND:Total colonic and small bowel aganglionosis (TCSA) occurs in less than 1% of all Hirschsprung's disease patients. Currently, the mainstay of treatment is surgery. However, in patients with TCSA, functional outcomes are often poor. A characteristic transition zone in TCSA can be difficult to identify which may complicate surgery and may often require multiple operations. CASE PRESENTATION:We present the case of a male infant who was diagnosed with biopsy-proven total colonic aganglionosis with extensive small bowel involvement as a neonate. The patient was diverted at one month of age based on leveling biopsies at 10 cm from the Ligament of Treitz. At 7 months of age, during stoma revision for a prolapsed stoma, intra-operative peristalsis was observed in nearly the entire length of the previously aganglionic bowel, and subsequent biopsies demonstrated the appearance of mature ganglion cells in a previously aganglionic segment. CONCLUSIONS:TCSA remains a major challenge for pediatric surgeons. Our case introduces new controversy to our understanding of aganglionosis. Our observations warrant further research into the possibility of post-natal ganglion maturation and encourage surgeons to consider a more conservative surgical approach.
journal_name
BMC Pediatrjournal_title
BMC pediatricsauthors
Salimi Jazi F,Chandler JM,Thorson CM,Sinclair TJ,Hazard FK,Kerner JA,Dutta S,Dunn JCY,Chao SDdoi
10.1186/s12887-019-1456-0subject
Has Abstractpub_date
2019-04-05 00:00:00pages
93issue
1issn
1471-2431pii
10.1186/s12887-019-1456-0journal_volume
19pub_type
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