J Pediatr Gastroenterol Nutr. 2023 May 16. doi: 10.1097/MPG.0000000000003833.Online ahead of print.

Desiree F Baaleman ^1 2, Mana H Vriesman ^1 2, Peter L Lu ^1 3, Marc A Benninga ², Marc A Levitt ³, Richard J Wood ³, Desale Yacob ^1 3, Carlo Di Lorenzo ^1 3, Ilan J N Koppen ^1 2

Author information

¹Division of Gastroenterology, Hepatology, and Nutrition, Nationwide Children's Hospital, Columbus, OH, USA.

²Department of Pediatric Gastroenterology and Nutrition, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam & VU University, Amsterdam, The Netherlands.

³Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH, USA.

Abstract

Objectives: To investigate long-term outcomes of antegrade continence enema (ACE) treatment in children with constipation or fecal incontinence.

Methods: Prospective cohort study including pediatric patients with organic or functional defecation disorders who started ACE treatment. Data were collected at baseline and at follow-up (FU) from 6 weeks until 60 months. We assessed parent and patient-reported gastrointestinal- health-related quality of life (HRQoL) using the Pediatric Quality of Life Inventory Gastrointestinal Symptoms Module (PedsQL™-GI), gastrointestinal symptoms, adverse events, and patient satisfaction.

Results: Thirty-eight children were included (61% male, median age 7.7 years, IQR 5.5-12.2). Twenty-two children (58%) were diagnosed with functional constipation (FC), 10 (26%) with an anorectal malformation, and 6 (16%) with Hirschsprung disease. Follow-up questionnaires were completed by 22 children (58%) at 6 months, 16 children (42%) at 12 months, 20 children (53%) at 24 months, and 10 children (26%) at 36 months. PedsQL™-GI scores improved overall with a significant increase at 12- and 24-month FU for children with FC and a significant increase in parent reported PedsQL™-GI score at 36-month FU for children with organic causes. Minor adverse events, such as granulation tissue, were reported in one third of children, and 10% of children needed a surgical revision of their ACE. The majority of all parents and children reported that they would "probably" or "definitely" choose ACE again.

Conclusion: ACE treatment is perceived positively by patients and parents and can lead to long-term improvement in gastrointestinal health-related quality of life in children with organic or functional defecation disorders.