Botulinum toxin injection for internal anal sphincter achalasia after pull-through surgery in Hirschsprung disease

Abstract Background Most children have a successful outcome after a pull-through for Hirschsprung’s disease. Some may have persisting symptoms after the pull-through. They could be managed conservatively, need minor surgical procedures, or a redo pull-through will be required. In this study, we presented our results in the management of the obstructive complications after pull-through for Hirschsprung’s disease. Results During the specified time period from January 2011 to December 2015, 21 patients presented to our department with persistent constipation or recurrent enterocolitis after a pull-through for Hirschsprung’s disease. Their age ranged between 4 months and 5 years (mean 2 years, median 2.5 years). They were 13 males and 8 females. Eleven patients underwent initial trans-anal endorectal pull-through, 4 underwent Duhamel procedure, and 6 underwent abdominal Soave technique. Three of the 11 patients with initial trans-anal endorectal pull-through had a tight anastomotic stricture which responded well to dilatation, 2 had a long muscular cuff which was incised laparoscopically, 4 had spasm of the internal anal sphincter which was relieved by sphincterotomy, and 2 had residual aganglionosis which required a redo pull-through. Two of the 4 patients who underwent initial Duhamel procedure had a long spur which was divided using a stapler, and the other 2 patients had residual aganglionosis which required a redo pull-through. One of the 6 patients who underwent abdominal Soave technique developed a long tight stricture and required a redo pull-through; in 1 patient, biopsy confirmed hypoganglionosis of the whole colon and was managed medically, and 4 patients had spasm of the internal anal sphincter which was relieved in 1 of them by sphincterotomy and in 2 by botulinum toxin injection while the remaining patient did not improve by either sphincterotomy or botulinum toxin injection. Conclusion Persistent constipation or recurrent enterocolitis after pull-through for Hirschsprung’s disease should be managed according to the cause; they could be managed medically by simple surgical procedures, or a redo pull-through may be required.

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Comparison of posterior internal anal sphincter myectomy and intrasphincteric botulinum toxin injection for treatment of internal anal sphincter achalasia: a meta-analysis

Pediatric Surgery International ◽

10.1007/s00383-012-3123-5 ◽

2012 ◽

Vol 28 (8) ◽

pp. 765-771 ◽

Cited By ~ 24

Author(s):

Florian Friedmacher ◽

Prem Puri

Keyword(s):

Botulinum Toxin ◽

Anal Sphincter ◽

Internal Anal Sphincter ◽

Meta Analysis ◽

Botulinum Toxin Injection ◽

Internal Anal Sphincter Achalasia

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EUS-Guided Botulinum Toxin Injection of the Internal Anal Sphincter in Anorectal Outlet Obstruction

The American Journal of Gastroenterology ◽

10.1038/ajg.2014.138 ◽

2014 ◽

Vol 109 (8) ◽

pp. 1293-1294 ◽

Cited By ~ 1

Author(s):

Arvind J Trindade ◽

Robert Hirten ◽

Ronald E Greenberg ◽

Divyesh V Sejpal

Keyword(s):

Botulinum Toxin ◽

Anal Sphincter ◽

Internal Anal Sphincter ◽

Botulinum Toxin Injection ◽

Outlet Obstruction

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Distribution of Interstitial Cells of Cajal in the Internal Anal Sphincter of Patients With Internal Anal Sphincter Achalasia and Hirschsprung Disease

Archives of Pathology & Laboratory Medicine ◽

10.5858/2003-127-1192-doicoc ◽

2003 ◽

Vol 127 (9) ◽

pp. 1192-1195 ◽

Cited By ~ 1

Author(s):

Anna Piaseczna Piotrowska ◽

Valeria Solari ◽

Prem Puri

Keyword(s):

Anal Sphincter ◽

Internal Anal Sphincter ◽

Interstitial Cells Of Cajal ◽

Internal Sphincter ◽

Smooth Muscles ◽

Hirschsprung Disease ◽

Interstitial Cells ◽

Nerve Fibers ◽

Internal Anal Sphincter Achalasia ◽

Cells Of Cajal

Abstract Context.—Interstitial cells of Cajal (ICCs) are pacemaker cells in the smooth muscles of the gut. The internal anal sphincter (IAS) is the most caudal part of gastrointestinal tract. It has the important function of maintaining fecal continence. It has been proposed that ICCs in the IAS mediate the inhibitory innervation of the recto-anal reflexes. Objective.—To investigate the distribution of ICCs in the normal IAS and in the IAS of children diagnosed with internal anal sphincter achalasia (IASA) and Hirschsprung disease (HD). Methods.—At the time of IAS myectomy, specimens of the IAS were taken from 8 patients with IASA, 4 patients with HD, and 4 normal controls. All specimens were examined using anti–c-Kit and antiperipherin antibodies; immunolocalization was detected with light microscopy. Density of the ICCs was graded by computerized image analysis. Results.—There was strong peripherin immunoreactivity in the ganglia cells and nerve fibers in the normal IAS. The number of peripherin-positive nerve fibers was markedly reduced in the IAS in patients with IASA. In HD patients, there was lack of peripherin immunoreactivity in the IAS, but hypertrophic nerve trunks stained strongly. Many c-Kit–positive ICCs were present among the muscle fibers and between the muscle bundles in the normal IAS. In HD and IASA patients, ICCs were absent or markedly reduced. Conclusion.—Altered distribution of ICCs in the internal sphincter in IASA and HD may contribute to motility dysfunction in these patients.

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