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Abstract
Introduction Despite surgical reconstruction in infancy, children with anorectal malformations (ARM) commonly have physical (faecal incontinence and constipation) and psychosocial morbidity. Traditional dogma is that high ARM have more morbidity compared with low ARM. Children with ARM have poorer sphincteric function demonstrated using anorectal physiology (ARP), yet there is limited knowledge of other extra-sphincteric parameters. In the absence of normal ARP values in children, it is difficult to interpret ARP levels for any condition such as ARM, in isolation.
Aim Our aim was to investigate the pathophysiological findings in children with ARM and compare these with children with functional constipation and faecal incontinence (FCFI).
Methods Patients cohorts were derived from the Children’s Anorectal Physiology Service prospectively kept database over a five-year period (September 2016 – October 2021). Primary outcome measures were: (1) ARP parameters, (2) transit marker study (TMS), (3) bowel scores (St Marks and Cleveland) (4) presence of urinary incontinence and (5) psychology scores (PIED and PedQoL). Patient demographics (sex and age) and type of ARM (Krickenbeck classification) are also reported.
Outcomes for children with ARM were compared with children with FCFI using SPSS v27 software. Categorical variables are presented as proportions and continuous variables as mean with standard deviation. Statistical tests used are presented and a p value <0.05 was considered statistically significant. Patients with missing data were not included in comparative analysis.
Results The overall cohort consisted of 305 children (40 (13.1%) had ARM and 265 (86.9%) had FCFI). Of the ARM cohort, 10 (25%) had high, 14 (35%) had intermediate, 8 (20%) had low malformations, 4 (10%) had anterior anus and 1 (2.5%) had cloaca. There were no differences between ARM types across all outcomes.
The ARM and FCFI cohorts were similar with regard to sex distribution and age at time of performing ARP (table 1). Children with ARMs have lower resting and squeeze sphincter pressures compared to children with FCFI. There were no differences in rectal sensation and dyssynergia between the two groups. Overall, there were no statistical differences in the TMS comparison, however there were proportionally more ARM patients with slow transit than FC/FI (30% cf. 18% respectively) and less ARM patients with rectal evacuatory disorder than FCFI (22% cf. 34% respectively). The severity of reported faecal incontinence (St Mark’s score), proportion with urinary incontinence and psychological scores were not different between the two groups, however children with FCFI have higher Cleveland scores.
Summary and Conclusions The aetiology of symptoms in children with constipation and faecal incontinence is complex and multifactorial (figure 1). The measured outcomes do not differ by ARM subtype. There are physiological differences in children with ARM and FCFI, but symptoms are similar. We suggest that multimodal management for ARM and FCFI should be similar. Normal physiological and motility results usefully direct management to reducing existing treatment or to focusing on behavioural intervention.