Abstract

AimsThe study prospectively assessed motor development from infancy to adolescence in patients with esophageal atresia (EA). MethodsAt one year of age motor performance was evaluated with the Psychomotor Developmental Index (PDI) of the Bayley Scales of Infant Development, Second Edition (BSID-II), and as adolescents reevaluated with Motor Assessment Battery for Children, Second Edition (MABC-2). Associations to clinical factors were assessed. Results23 EA patients were followed from infancy to adolescence. The median total PDI score in infancy was 102 (56–118) and the corresponding mean z-score was −0.006 (SD 0.995) and not significantly different from the reference values (p = 0.48). The median total MABC-2 score in adolescence was 75 (32–93) and the corresponding mean z-score −0.43 (SD 0.998) which is significantly below normal (p = 0.03). Children with impaired motor function in adolescence underwent significantly more rethoracotomies than those with normal motor performance (p = 0.037); whereas the two groups did not differ with respect to other clinical characteristics. ConclusionFrom infancy to adolescence the motor performance in the group of EA patients deteriorated from within normal range to significantly impaired compared to reference values. Interdisciplinary follow-up programs from infancy to adolescence with close monitoring for motor function is necessary to detect motor impairments.

Highlights

  • Anorectal malformations (ARM) are rare congenital defects which affect 1 in 5000 infants [1]

  • One hundred and forty-nine (67.1%) patients were treated with enemas and 73 (32.9%) with oral stimulant laxatives

  • On further review of the enema group, there was a slight predilection to patients on antegrade enemas, with 79 (53.0%) patients using either a cecostomy or an appendicostomy, compared to 70 (47.0%) who were on rectal enemas (Table 1)

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Summary

Introduction

Anorectal malformations (ARM) are rare congenital defects which affect 1 in 5000 infants [1]. The first publications on the utility of bowel management in the treatment of fecal incontinence and severe constipation in ARM patients appeared in the literature in 2009 [3, 4]. El-Gohary et al / Journal of Pediatric Surgery 56 (2021) 1689–1693 continent with the effective use of stimulant laxatives and water soluble fiber to add bulk [4] This combination allows them to pass formed stools on a regular basis. Many patients with anorectal malformations (ARM) need a bowel management program (BMP) to manage lifelong problems of fecal incontinence or severe constipation. Conclusion: An intensive BMP offers significant benefits in the treatment of fecal incontinence in ARM It appears to improve urinary incontinence and urinary voiding as well as the patient’s quality of life.

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