Cholinergic innervation and ganglion cell distribution in Hirschsprung's disease.


Journal

BMC pediatrics
ISSN: 1471-2431
Titre abrégé: BMC Pediatr
Pays: England
ID NLM: 100967804

Informations de publication

Date de publication:
24 08 2020
Historique:
received: 01 09 2019
accepted: 17 08 2020
entrez: 26 8 2020
pubmed: 26 8 2020
medline: 15 5 2021
Statut: epublish

Résumé

The diagnostic gold standard of Hirschsprung's disease (HD) is based on the histopathological assessment of colorectal biopsies. Although data on cholinergic innervation and ganglion cell (GC) distribution exist, only few studies have examined these two key features together. We assessed the pattern of cholinergic innervation and the amount of GCs in colorectal specimens of 14 HD patients. We established a semi-quantitative score for cholinergic innervation using acetylcholinesterase (AChE) enzyme histochemistry and quantitatively analyzed the number of GCs via NADH tetrazolium reductase (NADH) enzyme histochemistry. We examined both the entire length of the resected specimens as well as defined areas of the transition zone of both pathological and healthy appearing segment. High AChE score values were associated with absence of GCs, and AChE scores were inversely correlated with the number of GCs. Nevertheless, we observed several cases in which one of the two features revealed a normal distribution pattern, whereas the other still displayed pathological features. Our data support the need for transmural colon biopsies, to enable the best evaluation of both cholinergic innervation and GCs for a reliable assessment of HD.

Sections du résumé

BACKGROUND
The diagnostic gold standard of Hirschsprung's disease (HD) is based on the histopathological assessment of colorectal biopsies. Although data on cholinergic innervation and ganglion cell (GC) distribution exist, only few studies have examined these two key features together. We assessed the pattern of cholinergic innervation and the amount of GCs in colorectal specimens of 14 HD patients.
METHODS
We established a semi-quantitative score for cholinergic innervation using acetylcholinesterase (AChE) enzyme histochemistry and quantitatively analyzed the number of GCs via NADH tetrazolium reductase (NADH) enzyme histochemistry. We examined both the entire length of the resected specimens as well as defined areas of the transition zone of both pathological and healthy appearing segment.
RESULTS
High AChE score values were associated with absence of GCs, and AChE scores were inversely correlated with the number of GCs. Nevertheless, we observed several cases in which one of the two features revealed a normal distribution pattern, whereas the other still displayed pathological features.
CONCLUSIONS
Our data support the need for transmural colon biopsies, to enable the best evaluation of both cholinergic innervation and GCs for a reliable assessment of HD.

Identifiants

pubmed: 32838761
doi: 10.1186/s12887-020-02299-z
pii: 10.1186/s12887-020-02299-z
pmc: PMC7445925
doi:

Substances chimiques

Cholinergic Agents 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

399

Subventions

Organisme : Luxembourg National Research Fond
ID : P16/BM/11192686
Pays : International

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Auteurs

Anne K Braczynski (AK)

Department of Neurology, RWTH Aachen University Hospital, Aachen, Germany.
Department of Physical Biology, Heinrich-Heine University, Düsseldorf, Germany.
Institute of Biological Information Processing (IBI-7: Structural Biochemistry, Forschungszentrum Jülich, Jülich, Germany.
Institute of Neurology (Edinger Institute), Goethe University, Frankfurt, Germany.

Stefan Gfroerer (S)

Department of Pediatric Surgery, Helios Hospital Berlin-Buch, Berlin, Germany.

Rudi Beschorner (R)

Institute of Pathology and Neuropathology, Eberhard-Karls University, Tuebingen, Germany.

Patrick N Harter (PN)

Institute of Neurology (Edinger Institute), Goethe University, Frankfurt, Germany.
German Cancer Consortium (DKTK), Heidelberg, Germany.
German Cancer Research Center (DKFZ), Heidelberg, Germany.
Frankfurt Cancer Institute (FCI), Frankfurt am Main, Germany.

Peter Baumgarten (P)

Institute of Neurology (Edinger Institute), Goethe University, Frankfurt, Germany.
Department of Neurosurgery, Goethe University, Frankfurt, Germany.

Udo Rolle (U)

Department of Pediatric Surgery, University of Frankfurt am Main, Frankfurt, Germany.
University Children's Hospital, Goethe University, Frankfurt, Germany.

Michel Mittelbronn (M)

Institute of Neurology (Edinger Institute), Goethe University, Frankfurt, Germany. Michel.Mittelbronn@lns.etat.lu.
Department of Oncology (DONC), Luxembourg Institute of Health (LIH), Strassen, Luxembourg. Michel.Mittelbronn@lns.etat.lu.
Luxembourg Centre for Systems Biomedicine (LCSB), University of Luxembourg, Luxembourg City, Luxembourg. Michel.Mittelbronn@lns.etat.lu.
National Center of Pathology (NCP), Laboratoire national de santé (LNS), 1, Rue Louis Rech, L-3555, Dudelange, Luxembourg. Michel.Mittelbronn@lns.etat.lu.
Luxembourg Center of Neuropathology (LCNP), 1, Rue Louis Rech, L-3555, Dudelange, Luxembourg. Michel.Mittelbronn@lns.etat.lu.

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