Abstract
Introduction
Stasis from obstruction at the level of the internal anal sphincter (IAS) can lead to Hirschsprung-associated enterocolitis (HAEC) and may be improved by botulinum toxin (BT) injections. Our aim was to determine if BT injection during HAEC episodes decreased the number of recurrent HAEC episodes and/or increased the interval between readmissions.
Methods
A retrospective review was performed of patients admitted for HAEC from January 2010 to December 2019. Demographics and outcomes of patients who received BT were compared to patients who did not receive BT during their hospital stay.
Results
A total of 120 episodes of HAEC occurred in 40 patients; 30 patients (75%) were male, 7 (18%) had Trisomy 21 and 10 (25%) had long-segment disease. On multivariate analysis, patients who received BT during their inpatient HAEC episode had a longer median time between readmissions (p = 0.04) and trending toward an association with fewer readmissions prior to a follow-up clinic visit (p = 0.08).
Conclusion
The use of BT in HD patients hospitalized for HAEC is associated with an increased time between recurrent HAEC episodes and trended toward fewer recurrent episodes. The use of BT should be considered in the management of patients admitted with HAEC.
References
Levitt MA, Dickie B, Pena A (2010) Evaluation and treatment of the patients with Hirschsprung disease who is not doing well after a pull-through. Semin Pediatr Surg 19(2):146–153
Svetanoff WJ, Dekonenko C, Osuchukwu O et al (2020) Inpatient management of Hirschsprung’s associated enterocolitis treatment: the benefit of standardized care. Pediatr Surg Int. https://doi.org/10.1007/s00383-020-04747-4
Langer JC, Rollins MD, Levitt M et al (2017) Guidelines for the management of postoperative obstructive symptoms in children with Hirschsprung disease. Pediatr Surg Int 33:523–526
Han-Guerts IJM, Hendrix VC, de Blaauw I, Wijnen HWA, van Heurn ELW (2014) Outcome after anal intersphincteric botox injection in children with surgically treated Hirschsprung disease. JPGN 59(5):604–607
Gosain A, Frykman PK, Cowles RA et al (2017) Guidelines for the diagnosis and management of Hirschsprung’s-associated Enterocolitis. Pediatr Surg Int 33(5):517–521
Langer JC, Birnbaum E (1997) Preliminary experience with intrasphincteric botulinum toxin for persistent constipation after pull-through for Hirschsprung’s disease. J Pediatr Surg 32(7):1059–1062
Langer JC (2004) Persistent obstructive symptoms after surgery for Hirschsprung’s disease: development of a diagnostic and therapeutic algorithm. J Pediatr Surg 39:1458–1462
Koivusalo AI, Pakarinen MP, Rintala RJ (2009) Botox injection treatment for anal outlet obstruction in patients with internal anal sphincter achalasia and Hirschsprung’s disease. Pediatr Surg Int 25:873–876
Thakkar HS, Bassett C, Hsu A et al (2017) Functional outcomes in Hirschsprung’s disease: a single institution’s 12-year experience. J Pediatr Surg 52:277–280
Church JT, Gadepalli SK, Talishinsky T, Teielbaum DH, Jarboe MD (2017) Ultrasound-guided intrasphincteric botulinum toxin injection relieves obstructive defecation due to Hirschsprung’s disease and internal anal sphincter achalasia. J Pediatr Surg 52:74–78
Louis-Barrone C, Faure A et al (2019) Neurostimulation-guided anal intrasphincteric botulinum toxin injection in children with Hirschsprung disease. JPGN 68:527–532
Roorda D, Abeln ZAM, Oosterlaan J, van Heurn LWE, Derikx JPM (2019) Botulinum toxin injections after surgery for Hirschsprung disease: systematic review and meta-analysis. World J Gastroenterol 25(25):3268–3280
Nataraja RM, Ferguson P, King S, Lynch A, Pacilli M (2019) Management of Hirschsprung disease in Australia and New Zealand: a survey of the Australian and New Zealand Association of Paediatric Surgeons (ANZAPS). Pediatr Surg Int 35:419–423
Patrus B, Nasr A, Langer JC, Gerstle JT (2011) Intrasphincteric botulinum toxin decreases the rate of hospitalization for postoperative obstructive symptoms in children with Hirschsprung disease. J Pediatr Surg 46:184–187
Wester T, Granström AL (2015) Botulinum toxin is efficient to treat obstructive symptoms in children with Hirschsprung disease. Pediatr Surg Int 31:255–259
Chumpitazi BP, Fishman SJ, Nurko S (2009) Long-term clinical outcome after botulinum toxin injection in children with nonrelaxing internal anal sphincter. Am J Gastroenterol 104(4):976–983
Kastenberg ZJ, Wall N, Malhotra N et al (2020) The effect of multidisciplinary colorectal center development on short-term hospital readmissions for patients with anorectal malformations or Hirschsprung disease. J Pediatr Surg 55:541–544
Author information
Authors and Affiliations
Contributions
Study conception and design: WJS, RMR. Acquisition of data: WJS, PA, RJH, RMR. Analysis and interpretation of data: WJS, JJL, PA, RJH, TAO, RMR. Drafting of manuscript: WJS, JJL. Critical revision of manuscript: WJS, JJL, PA, RJH, TAO, RMR.
Corresponding author
Ethics declarations
Ethical approval
This article does not contain any studies with human participants or animals performed by any of the authors.
Conflict of interests
All authors have no conflicts of interest.
Informed consent
Due to the retrospective nature of the study, informed consent was waived by our institutional review board.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Svetanoff, W.J., Lopez, J., Aguayo, P. et al. The impact of botulinum injection for hospitalized children with Hirschsprung-associated enterocolitis. Pediatr Surg Int 37, 1467–1472 (2021). https://doi.org/10.1007/s00383-021-04966-3
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00383-021-04966-3