Pediatric Surgery International

, Volume 33, Issue 8, pp 883–886 | Cite as

Sensory innervation of the anal canal and anorectal line in Hirschsprung’s disease: histological evidence from mouse models

  • Masahiro Takeda
  • Katsumi MiyaharaEmail author
  • Chihiro Akazawa
  • Geoffrey J. Lane
  • Atsuyuki Yamataka
Original Article



We used non-Hirschsprung’s disease (HD) Sox10-Venus Transgenic mice (non-HDSV-mice), an endothelin receptor-B knockout mouse model of HD (HD-mice), and C57B6C3 wild controls (C-mice) to identify the correlation between the anorectal line (ARL) and successful transanal pull-through (TAPT).


In non-HDSV-mice, intestinal neural crest-derived cells can be visualized with Venus,—a green fluorescent protein—without histochemical staining. We exposed the anal canal in each non-HDSV-mouse and marked the ARL directly with red ink. Specimens of anus and rectum from HD- and C-mice were immunostained with sensory nerve markers substance P and calcitonin gene related peptide (CGRP) and Hematoxylin and Eosin.


Stereoscopic microscopy confirmed a squamous-columnar epithelial junction corresponding to the red ink in non-HDSV-mice. Fluorescence microscopy showed intense Venus expression proximal to the ARL and little enteric nerve expression distally. Substance P and CGRP expression were strong in the basal layer of the anal transitional zone (ATZ) in both HD- and C-mice; i.e., distal sensory innervation was normal in HD-mice.


The ARL delineated a distinct demarcation in sensory innervation that is normal even in HD-mice. Thus, the initial incision during TAPT should be based on the ARL because it is readily identifiable and intimately involved with bowel function.


Hirschsprung’s disease Transanal pull-trough Anorectal line Anal transitional zone Substance P Calcitonin gene related peptide Sox10-Venus/C57BL GFP transgenic mice 



This work was supported by JSPS (Japan Society for the Promotion of Science) KAKENHI Grant numbers 15K10929 & 26462715.


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Copyright information

© Springer-Verlag GmbH Germany 2017

Authors and Affiliations

  • Masahiro Takeda
    • 1
  • Katsumi Miyahara
    • 1
    Email author
  • Chihiro Akazawa
    • 2
  • Geoffrey J. Lane
    • 1
  • Atsuyuki Yamataka
    • 1
  1. 1.Department of Pediatric General and Urogenital SurgeryJuntendo University School of MedicineTokyoJapan
  2. 2.Department of Biochemistry and Biophysics, Graduate School of Health Care SciencesTokyo Medical and Dental UniversityTokyoJapan

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