Intestinal neuronal dysplasia is a possible cause of persistent bowel symptoms after pull-through operation for Hirschsprung's disease

Kobayashi, Hiroyuki; Hirakawa, Hitoshi; Surana, R.; O’Briain, D. S.; Puri, Prem

doi:10.1016/0022-3468(95)90570-7

Cited by 118 publications

(74 citation statements)

References 12 publications

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“…Meier-Ruge found IND in 34.4% of patients with HD in his specialized laboratory [13]. Meanwhile, the existence and relevance of associated IND is generally accepted even by groups with initial doubts about this entity [8,21]. However, most studies were retrospective, and no data were given as to the length of the segments aected with IND.…”

Section: Discussionmentioning

confidence: 94%

“…Fadda et al [1] reported motility disorders in one-third of children with HaIND and Kobayashi et al [8] found defecation disorders in all of their patients with HaIND. Hanimann et al [3] described constipation in the early postoperative course in 18.2% of their patients with HaIND and 8.3% with isolated HD.…”

Section: Discussionmentioning

confidence: 97%

“…Morphologic mapping during the initial operation has been recommended [8,15]. A search for dysganglionosis seems to be indispensable at the latest when early and severe obstructive symptoms occur after intestinal resection.…”

Section: Discussionmentioning

confidence: 99%

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Hirschsprung's disease and intestinal neuronal dysplasia - a frequent association with implications for the postoperative course

Schmittenbecher

Sacher

Cholewa

et al. 1999

Pediatric Surgery International

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Between 1991 and 1993, 106 newly diagnosed cases of Hirschsprung's disease (HD) were prospectively analyzed for the association of HD and intestinal neuronal dysplasia (IND) at ten pediatric surgical departments in central Europe. Hirschsprung-associated IND (HaIND) was found in 40% of cases. IND was disseminated in one-third and localized in two-thirds of the patients. Initial clinical symptoms were related to the length of the aganglionic segment, but not to the presence of HaIND. An enterostomy performed in 72 cases (67.9%) was located in a segment of pathologically innervated bowel in 50% of all cases, but in 72% of cases of HaIND. The proximal margin of the resected bowel showed pathological innervation in 44% of cases. Supplemental biopsies from the intestine (apart from diagnostic suction biopsies and biopsies at the enterostomy site) led to the first identification or definition of length of associated IND in 17.9% of cases. Postoperatively, the presence of long-segment aganglionosis or associated IND implied a delay in the restoration or normal defecation. Persistent constipation was found in 40% of patients with associated disseminated IND at follow-up at 6 months, compared to 20.6% in patients with isolated HD. These children needed secondary interventions more often than patients with associated localized IND or isolated HD. HaIND thus has clinical implications for the postoperative course if IND is disseminated.

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Section: Discussionmentioning

confidence: 94%

Section: Discussionmentioning

confidence: 97%

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Hirschsprung's disease and intestinal neuronal dysplasia - a frequent association with implications for the postoperative course

Schmittenbecher

Sacher

Cholewa

et al. 1999

Pediatric Surgery International

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“…The results indicate to us that the lesions caused by ischemia/reperfusion give rise to decreased numbers of ganglionic cells in the myenteric plexus. This may have the consequence of alterations in intestinal motility, since these cells are essential for transmitting nerve impulses for generating peristaltic movement 23,24 .…”

Section: Discussionmentioning

confidence: 99%

“…We utilized histological analysis since this is the method most utilized for evaluating the degree of tissue lesion caused by the phenomenon of ischemia/reperfusion. When there is a loss of vascularization, there is generally an associated loss of innervation 23,24 . In our experimental model for provoking ischemia/reperfusion, we were able to observe this, especially when comparing the group GCEI7 (control) with the group GIREI7, on the 7 th day after undergoing ischemia/ reperfusion.…”

Section: Discussionmentioning

confidence: 99%

Do lesions of the enteric nervous system occur following intestinal ischemia/reperfusion?

et al. 2007

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Purpose:To evaluate tissue lesions, especially those of the intestinal innervation, in an excluded jejunal loop subjected to ischemia and reperfusion in rats. Methods: To evaluate the role of ischemia and reperfusion lesions in an excluded intestinal loop, four groups of 20 rats were set up: control group (GCEI7) and three experimental groups (GIREI7, GIREI14 and GIREI28). They were all subjected to exclusion of an intestinal segment of six centimeters in length, at a distance of 10 centimeters from the Treitz angle. The 60 animals in the three experimental groups were additionally subjected to ischemia of the vascular pedicle for 30 minutes. The control group and the experimental group GIREI7 were evaluated on the 7 th day after the operation. The groups GIREI14 and GIREI28 (which also underwent ischemia) were utilized to evaluate the evolution of the lesion over time, on the 14 th and 28 th days after the operation, respectively. From the intestinal excluded loop, we take one ring of 0,5 cm distal and proximal, that were fixed in formaline 10% solution in order to do histological (HE) and immuno-hystochemial (PS-100) evaluation (enteric nervous system.) The distal loop was exteriorized in stoma and the proximal part closed with polipropilene 6-0. Results: It was observed a decrease in the number of ganglionic cells in the myenteric plexus in the group subjected to ischemia and reperfusion (GIREI7), in relation to the control group (GCEI7) at the 7 th post-operative day (Mann-Whitney test: p = 0.0173 *. Comparing the numbers of ganglionic cells in the myenteric plexus before and after jejunal loop exclusion GCEI7 -(Wilcoxon test: p = 0.0577). GIREI7 -Comparing the numbers of ganglionic cells in the myenteric plexus before and after ischemia (*p = 0.0399). Comparing the percentage variations in ganglionic cells in the myenteric plexus on the 7 th , 14 th and 28 th days after the procedure, in the groups GIREI7, GIREI14 and GIREI28, it was observed that there were no significant alterations. Kruskal-Wallis test: p = 0.6501. Conclusion: There was a decrease in the number of ganglionic cells in the myenteric plexus due to ischemia and reperfusion that did not recover in the late post-operative period. Key words: Intestine, small. Ischemia. Reperfusion. Enteric nervous system. RESUMO Objetivo: Avaliar lesões teciduais, especialmente aquelas da inervação intestinal em alça jejunal excluída submetida à isquemia e reperfusão em ratos. Métodos: Para avaliar o papel da isquemia e reperfusão nas lesões em uma alça intestinal exclusa, quatro grupos de 20 ratos foram criados: Grupo controle (GCE17) e 3 Grupos experimentais (GIRE!7, GIREI14) e GIREI28) Todos foram submetidos à exclusão de um segmento intestinal de seis centímetros de extensão, a 10 centímetros do ângulo de Treitz . Os 60 animais dos 3 grupos experimentais foram também submetidos a isquemia do pedículo vascular por 30 minutos.O grupo controle e o grupo experimental GIREI7 foram avaliados no 7º. Dia após a operação. Os grupos GIREI14 e GIREI28 também ...

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Neuronal dysplasia: A controversial pathological correlate of intestinal pseudo‐obstruction

Kapur

2003

American J of Med Genetics Pt A

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The infant or child with intestinal pseudo-obstruction poses many challenges for geneticists and other specialists. Although a well-defined anatomic diagnosis (e.g., Hirschsprung disease) can be established for a subset of patients, the pathological correlates for many patients are non-existent or controversial. Intestinal neuronal dysplasia (IND) is frequently considered in the differential diagnosis, despite the fact that existence and significance of the abnormal histopathological features that characterize IND are hotly debated. This review highlights some of the concerns regarding this diagnosis including problems with the diagnostic criteria, the manner in which these criteria are applied in contemporary pathology practices, and the likelihood that many of the pathological findings are secondary consequences of impaired motility with no other clear clinical significance. Possible genetic and developmental bases for IND are also discussed.

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Intestinal neuronal dysplasia is a possible cause of persistent bowel symptoms after pull-through operation for Hirschsprung's disease

Cited by 118 publications

References 12 publications

Hirschsprung's disease and intestinal neuronal dysplasia - a frequent association with implications for the postoperative course

Hirschsprung's disease and intestinal neuronal dysplasia - a frequent association with implications for the postoperative course

Do lesions of the enteric nervous system occur following intestinal ischemia/reperfusion?

Neuronal dysplasia: A controversial pathological correlate of intestinal pseudo‐obstruction

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