Eur J Pediatr Surg 2023; 33(03): 241-248
DOI: 10.1055/s-0042-1751048
Original Article

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

1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
2   Department of Pediatric Gastroenterology, Amsterdam UMC, Emma Childrens' Hospital UMC, University of Amsterdam, Amsterdam, the Netherlands
,
Alexandra Hallagan
1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
3   College of Medicine, The Ohio State University, Columbus, Ohio, United States
,
Devin R. Halleran
4   Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Danielle K. Orsagh-Yentis
1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Marc A. Levitt
4   Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Richard J. Wood
4   Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Marc A. Benninga
2   Department of Pediatric Gastroenterology, Amsterdam UMC, Emma Childrens' Hospital UMC, University of Amsterdam, Amsterdam, the Netherlands
,
Neetu Bali
1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Karla H. Vaz
1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Desale Yacob
1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Carlo Di Lorenzo
1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
,
Peter L. Lu
1   Department of Pediatrics, Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, Ohio, United States
› Author Affiliations
Funding Desiree F. Baaleman received financial support from the Prins Bernhard Cultuurfonds with support from the Jadefonds, and the VSBfonds to conduct this research.

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.



Publication History

Received: 02 May 2022

Accepted: 18 May 2022

Article published online:
19 July 2022

© 2022. Thieme. All rights reserved.

Georg Thieme Verlag KG
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