Literature DB >> 19082831

Low incidence of enterocolitis and colonic mucosal inflammation in Norwegian patients with Hirschsprung's disease.

Yasser Rehman1, Kristin Bjørnland, Kjetil Juul Stensrud, Inger Nina Farstad, Ragnhild Emblem.   

Abstract

BACKGROUND: Hirschsprung's disease (HD) may be associated with inflammation in the colon. Further, the etiology of Hirschsprung-associated enterocolitis (HEC) is unclear. To learn more about these features, we examined our cohort of HD patients during a period of 6 years for inflammation in their colonic mucosa as well as for signs of HEC.
MATERIALS AND METHODS: Rectal suction biopsies and operative full thickness aganglionic and ganglionic colonic specimens from 36 patients were examined. Signs of inflammation were recorded in hematoxylin/eosin/saffron (HES)-stained sections and with fluorescence conjugated polyclonal antibodies to IgA and IgG applied on serial sections. The suction biopsies were also evaluated for the presence of mucus inspissation and crypt dilatation. Clinical signs of HEC were recorded from medical files of the same 36 patients.
RESULTS: HES-staining revealed that seven patients had inflammation in the suction biopsies; these patients were significantly older than the patients without inflammation. Slight mucus inspissation was identified in suction biopsies of five out of 33 patients, but crypt abscesses or ulcerations were not found in any specimens. Virtually all very young patients (<3 months) had slight crypt dilatation. We identified inflammation in resected colonic segments from 17 out of 36 patients. Thirteen of these 17 had a diverting colostomy, and only one out of 14 patients with colostomy had no inflammation. Inflammatory changes were similar in ganglionic and aganglionic bowel. By immunofluorescence (IF) staining, inflammation was found in resected colonic segments from five patients. Four of these had a colostomy. HEC was diagnosed in three patients, and inflammation detected in resected specimens from only one of these three.
CONCLUSIONS: We have not been able to identify particular characteristics in the colonic or rectal mucosa that are linked to development of HEC. Inflammation in the resected specimen was mainly found in patients with a diverting colostomy, and then in both ganglionic and aganglionic colon.

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Mesh:

Year:  2008        PMID: 19082831     DOI: 10.1007/s00383-008-2300-z

Source DB:  PubMed          Journal:  Pediatr Surg Int        ISSN: 0179-0358            Impact factor:   1.827


  24 in total

Review 1.  Recent advances in the management of Hirschsprung's disease.

Authors:  A G Coran; D H Teitelbaum
Journal:  Am J Surg       Date:  2000-11       Impact factor: 2.565

Review 2.  New insights into the pathogenesis of Hirschsprung's associated enterocolitis.

Authors:  Feilim Murphy; Prem Puri
Journal:  Pediatr Surg Int       Date:  2005-09-30       Impact factor: 1.827

3.  Are the long-term results of the transanal pull-through equal to those of the transabdominal pull-through? A comparison of the 2 approaches for Hirschsprung disease.

Authors:  Mohamed I El-Sawaf; Robert A Drongowski; Jennifer N Chamberlain; Arnold G Coran; Daniel H Teitelbaum
Journal:  J Pediatr Surg       Date:  2007-01       Impact factor: 2.545

4.  Immunoglobulin- and J chain-producing cells associated with lymphoid follicles in the human appendix, colon and ileum, including Peyer's patches.

Authors:  K Bjerke; P Brandtzaeg
Journal:  Clin Exp Immunol       Date:  1986-05       Impact factor: 4.330

5.  Distribution of immunoglobulin producing cells is different in normal human appendix and colon mucosa.

Authors:  K Bjerke; P Brandtzaeg; T O Rognum
Journal:  Gut       Date:  1986-06       Impact factor: 23.059

6.  Natural history and pathophysiology of enterocolitis in the piebald lethal mouse model of Hirschsprung's disease.

Authors:  T Fujimoto
Journal:  J Pediatr Surg       Date:  1988-03       Impact factor: 2.545

7.  A decade of experience with the primary pull-through for hirschsprung disease in the newborn period: a multicenter analysis of outcomes.

Authors:  D H Teitelbaum; R E Cilley; N J Sherman; D Bliss; N D Uitvlugt; E J Renaud; I Kirstioglu; T Bengston; A G Coran
Journal:  Ann Surg       Date:  2000-09       Impact factor: 12.969

8.  Enterocolitis associated with Hirschsprung's disease: a clinical histopathological correlative study.

Authors:  E A Elhalaby; D H Teitelbaum; A G Coran; K P Heidelberger
Journal:  J Pediatr Surg       Date:  1995-07       Impact factor: 2.545

9.  Impaired gastrointestinal mucosal defense in Hirschsprung's disease: a clue to the pathogenesis of enterocolitis?

Authors:  D Wilson-Storey; W G Scobie
Journal:  J Pediatr Surg       Date:  1989-05       Impact factor: 2.545

10.  MUC-2 mucin production in Hirschsprung's disease: possible association with enterocolitis development.

Authors:  Aladdein F Mattar; Arnold G Coran; Daniel H Teitelbaum
Journal:  J Pediatr Surg       Date:  2003-03       Impact factor: 2.545

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