Neurostimulation-Guided Anal Intrasphincteric Botulinum Toxin Injection in Children with Hirschsprung's Disease

Objectives: In Hirschsprung's disease (HD), despite successful surgical treatment, 50% of children experience long-term functional gastrointestinal problems, particularly chronic functional obstructive symptoms. We report our experience regarding clinical effects of neurostimulation-guided anal intrasphincteric botulinum toxin (BT) injections on postoperative obstructive symptoms attributed to a non-relaxing anal sphincter complex in HD patients. Methods: In this monocenter cohort study, 15 HD patients with postoperative functional intestinal obstructive symptoms received neurostimulation-guided anal intrasphincteric BT injections. Short, medium and long-term effects were evaluated. The Bristol stool form scale was used to assess stool consistency, and the Jorge-Wexner (JW) score to assess fecal continence. Results: The median age at first injection was 4 years. In the short-term, a significant improvement in stool consistency was noted in 12/14 patients (p = 0.0001) and JW score decreased for 14/15 patients (p = 0.001). In the medium-term, JW score significantly decreased for all patients (p = 0.0001), with an improvement of 50% or more for 10 patients (66.7%). In the long-term, 83.3% of patients had normal stool consistency and JW score was less than 3 for all. Recurrent enterocolitis decreased from 86.7% to 8.3%. A complete resolution of all symptoms without further medication was observed in 66.7% of patients in the long-term. Conclusions: Intrasphincteric BT injection was a safe, effective and durable option for the management of postoperative functional intestinal obstructive symptoms in HD. The use of neurostimulator guidance for specific delivery of BT to muscular fibers of non-relaxing anal sphincter complex takes into consideration the variability of patient's anatomy secondary to curative surgery. Address correspondence and reprint requests to Claude Louis-Borrione, MD, Pediatric Surgery Department, Hôpital La Timone-Enfants, 264 rue Saint Pierre, 13385 Marseille, France (E-mail: claude.louis@ap-hm.fr); Anne Dariel, MD, Pediatric Surgery Department, Hôpital La Timone-Enfants, 264 rue Saint Pierre, 13385 Marseille, France (e-mail: anne_dariel@yahoo.fr). Received 22 April, 2018 Accepted 30 August, 2018 Supplemental digital content is available for this article. Direct URL citations appear in the printed text, and links to the digital files are provided in the HTML text of this article on the journal's Web site (www.jpgn.org). Conflicts of interest and source of funding: none declared. Author Contributions: Claude Louis-Borrione is a pediatric surgeon with an expertise in colorectal surgery in children with Hirschsprung's disease. She designed and conducted the entire study: patient's inclusion, botulinum toxin injections, patient's follow-up and acquisition of data. She supervised the study. Anne Dariel analyzed and interpreted the data, then wrote the paper. Sarah Garnier did the bibliographic research. Julien Labreuche performed the statistical analysis. Claude Borrione, Géraldine Héry, Alice Faure, Jean-Michel Guys and Thierry Merrot were the pediatric surgeons in charge of the primary surgical management of the included patients. They also participated in the critical reading of the manuscript for important intellectual contents. © 2018 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology,

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