Anal Botulinum Toxin in Children with Hirschsprung Disease and Functional Constipation: A Prospective Cohort study

Abstract

Introduction Anal sphincter botulinum toxin injections (BTIs) are used in the treatment of children with severe defecation disorders, including Hirschsprung disease (HD) and functional constipation (FC). Our objective was to evaluate the outcomes of BTI in these children.

Materials and Methods We performed a prospective cohort study of children undergoing BTI from July 2018 to December 2018. We recorded perceived effect of the BTI, including effectiveness ranging from 0 (not at all effective) to 4 (extremely effective). In addition, we recorded symptoms and the Cleveland Clinic Constipation Score (CCCS). Data were collected at baseline and at 2 weeks, 2 months, and 4 months post-injection.

Results Forty-two children (HD = 25, FC = 17) were included in the study (median age 4.3 years, IQR 2.4–7.2, 52% male). Twenty-two (88%) children with HD and eight (47%) children with FC had previously undergone a BTI. BTIs were perceived effective in 16 (76%) and 12 (71%) children with HD and eight (47%) and seven (47%) children with FC at 2-week and 2-month follow-up, respectively. Effectiveness was not rated differently between groups except at the 2-month follow-up, when patients with HD rated the BTI more effective compared to those with FC (median 2 [HD] vs. median 1 [FC], p = 0.022). Over the course of the study, 17/39 (44%) children reported self-limiting adverse effects such as fecal incontinence and pain at the injection site.

Conclusion Anal sphincter BTIs can be effective in the treatment of constipation in both HD and FC patients.

Keywords

constipation - Hirschsprung disease - child - fecal incontinence

Publication History

Received: 02 May 2022

Accepted: 18 May 2022

Article published online:
19 July 2022

• References

• 1 Chumpitazi BP, Fishman SJ, Nurko S. Long-term clinical outcome after botulinum toxin injection in children with nonrelaxing internal anal sphincter. Am J Gastroenterol 2009; 104 (04) 976-983
• 2 Bobkiewicz A, Francuzik W, Krokowicz L. et al. Botulinum toxin injection for treatment of chronic anal fissure: is there any dose-dependent efficiency? A meta-analysis. World J Surg 2016; 40 (12) 3064-3072
• 3 Ma J, Elsaidi GA, Smith TL. et al. Time course of recovery of juvenile skeletal muscle after botulinum toxin A injection: an animal model study. Am J Phys Med Rehabil 2004; 83 (10) 774-780 , quiz 781–783
• 4 Roorda D, Abeln ZA, Oosterlaan J, van Heurn LW, Derikx JP. Botulinum toxin injections after surgery for Hirschsprung disease: systematic review and meta-analysis. World J Gastroenterol 2019; 25 (25) 3268-3280
• 5 Langer JC, Rollins MD, Levitt M. et al; American Pediatric Surgical Association Hirschsprung Disease Interest Group. Guidelines for the management of postoperative obstructive symptoms in children with Hirschsprung disease. Pediatr Surg Int 2017; 33 (05) 523-526
• 6 Halleran DR, Lu PL, Ahmad H. et al. Anal sphincter botulinum toxin injection in children with functional anorectal and colonic disorders: a large institutional study and review of the literature focusing on complications. J Pediatr Surg 2019; 54 (11) 2305-2310
• 7 Church JT, Gadepalli SK, Talishinsky T, Teitelbaum DH, Jarboe MD. Ultrasound-guided intrasphincteric botulinum toxin injection relieves obstructive defecation due to Hirschsprung's disease and internal anal sphincter achalasia. J Pediatr Surg 2017; 52 (01) 74-78
• 8 Zar-Kessler C, Kuo B, Belkind-Gerson J. Botulinum toxin injection for childhood constipation is safe and can be effective regardless of anal sphincter dynamics. J Pediatr Surg 2018; 53 (04) 693-697
• 9 Han-Geurts IJ, Hendrix VC, de Blaauw I, Wijnen MH, van Heurn EL. Outcome after anal intrasphincteric Botox injection in children with surgically treated Hirschsprung disease. J Pediatr Gastroenterol Nutr 2014; 59 (05) 604-607
• 10 Basson S, Charlesworth P, Healy C, Phelps S, Cleeve S. Botulinum toxin use in paediatric colorectal surgery. Pediatr Surg Int 2014; 30 (08) 833-838
• 11 Tabbers MM, DiLorenzo C, Berger MY. et al; European Society for Pediatric Gastroenterology, Hepatology, and Nutrition, North American Society for Pediatric Gastroenterology. Evaluation and treatment of functional constipation in infants and children: evidence-based recommendations from ESPGHAN and NASPGHAN. J Pediatr Gastroenterol Nutr 2014; 58 (02) 258-274
• 12 Harris PA, Taylor R, Thielke R, Payne J, Gonzalez N, Conde JG. Research electronic data capture (REDCap)–a metadata-driven methodology and workflow process for providing translational research informatics support. J Biomed Inform 2009; 42 (02) 377-381
• 13 Lewis SJ, Heaton KW. Stool form scale as a useful guide to intestinal transit time. Scand J Gastroenterol 1997; 32 (09) 920-924
• 14 Garra G, Singer AJ, Taira BR. et al. Validation of the Wong-Baker FACES Pain Rating Scale in pediatric emergency department patients. Acad Emerg Med 2010; 17 (01) 50-54
• 15 Agachan F, Chen T, Pfeifer J, Reissman P, Wexner SD. A constipation scoring system to simplify evaluation and management of constipated patients. Dis Colon Rectum 1996; 39 (06) 681-685
• 16 Hallagan A, Pearlstein H, Ryan K. et al. Tu1604—efficacy and safety of anal sphincter botulinum toxin (BOTOX) injection in children with anorectal and colonic disorders. Gastroenterology 2019; 156: S-1059
• 17 Radwan AB, Gadallah MA, Shahawy MR, Albagdady AA, Talaat AA. Can botulinum toxin help in managing children with functional constipation and obstructed defecation?. J Pediatr Surg 2021; 56 (04) 750-753
• 18 Brisighelli G, Macchini F, Consonni D, Di Cesare A, Morandi A, Leva E. Continence after posterior sagittal anorectoplasty for anorectal malformations: comparison of different scores. J Pediatr Surg 2018; 53 (09) 1727-1733
• 19 Koppen IJN, Saps M, Lavigne JV. et al. Recommendations for pharmacological clinical trials in children with functional constipation: the Rome foundation pediatric subcommittee on clinical trials. Neurogastroenterol Motil 2018; 30 (04) e13294