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Review Article

Functional Fecal Incontinence in Children: Epidemiology, Pathophysiology, Evaluation, and Management

Rajindrajith, Shaman; Devanarayana, Niranga Manjuri; Thapar, Nikhil; Benninga, Marc Alexander§

Author Information
Journal of Pediatric Gastroenterology and Nutrition: June 2021 - Volume 72 - Issue 6 - p 794-801
doi: 10.1097/MPG.0000000000003056
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Abstract

What Is Known/What Is New

What Is Known

  • Functional fecal incontinence is subclassified into two distinct disorders; constipation-associated fecal incontinence and functional nonretentive fecal incontinence.
  • Both are common problems across the globe leading to considerable suffering, and healthcare burden.
  • Therapeutic interventions including toilet training and laxative therapy have been used successfully in children with functional fecal incontinence.

What Is New

  • Evolving investigations such as high-resolution anorectal and colonic manometry have identified more precise pathophysiological mechanisms for functional fecal incontinence.
  • Novel nonpharmacological interventions including pelvic floor physiotherapy, neuromodulation, and intersphincteric injection of botulinum toxin have shown therapeutic advantages.
  • Surgical interventions such as antegrade continence enema, and segmental resection of bowel may be effective in children with refractory symptoms.

Fecal incontinence (FI) in children indicates the passage of stools into the underwear, either as an unintentional seepage of small amounts of liquid stools (generally referred to as “soiling” or “leakage”) or the complete evacuation of formed stools after the age of 4 years. Irrespective of the amount of stools, it is one of the most embarrassing and distressing events for a growing child. Consequently, they have a poor health-related quality of life (QoL) (1) and are socially isolated, subjected to bullying, rejected by peers, and frequently face child maltreatment (2). In addition, FI has a negative connotation on behavior, upbringing, and learning, all of which are detrimental to a developing child's physical and psychosocial well-being (3).

FI in children is classified into two major groups: functional and organic. The largest group consists of functional FI (ie, FI without an organic cause), further subdivided into retentive and nonretentive FI and defined according to Rome IV criteria (4). Organic FI is usually related to detectable structural or physiological abnormalities within the gastrointestinal tract or involving the nervous systems that provide intrinsic or extrinsic control of continence mechanisms (Fig. 1). This review focuses on functional FI in children.

F1
FIGURE 1:
Classification of fecal incontinence.

EPIDEMIOLOGY OF FUNCTIONAL FECAL INCONTINENCE

Functional FI

Several studies have shown that the majority of children either presenting to the hospital (75–90%) or in the community (80%) have FI due to fecal retention (5–7). Functional FI (both retentive and nonretentive) is more common in males than in females (1.5–3.5 to 1) (5,8,9). A recent meta-analysis of functional defecation disorders reported a global pooled prevalence of functional nonretentive fecal incontinence (FNRFI) of 0.4%, ranging from 0% to 1.8% (10). Unfortunately, this systematic review failed to provide the prevalence of retentive FI associated with constipation.

PATHOPHYSIOLOGY OF FUNCTIONAL FI

Constipation-Associated (Retentive) FI

Inadequate toilet training and withholding behavior are the most common factors that underlie the development of constipation in children.

Stool withholding is a natural reflex mechanism of young children who experience painful defecation, for example, due to hard stools. These children contract their anal sphincters and gluteal muscles to withhold stools, leading to a cascade of physiological changes that further augment stool retention and the formation of a large fecal mass in the rectum with dilatation of both the rectum and the sigmoid colon. Retained fecal matter becomes harder as the water gets absorbed through the colorectal mucosa and eventually become excessively firm and challenging to be propelled by the high-amplitude propagatory contractions (HAPCs). The stools in the upper part of the fecal mass get liquefied due to bacterial action, seeps over the distal hard fecal mass, and out of the anal sphincter, leading to fecal incontinence.

Functional Nonretentive FI

The exact pathophysiological mechanisms responsible for functional nonretentive FI (FNRFI) are not entirely clear. Several studies have suggested that these children have normal physiological function of the anorectal unit including normal resting and squeeze pressure of the anal sphincter complex and a normal rectal sensation to balloon distension (11–14). Children with FNRFI commonly report that they either have no time to go to the toilet or reluctant to leave activities they are engaged with, for example, computer games. Therefore, it is hypothesized that they deny or neglect the normal urge to defecate (13).

IMPACT OF FI

Functional FI and HRQoL

Children with constipation-associated FI and FNRFI have lower HRQoL scores compared to children with constipation alone in both community-based (1,15) and hospital-based studies (16,17). Unsurprisingly, the social and emotional functioning components were more negatively influenced by FI than other aspects when QoL was assessed using disease-specific questionnaires (17).

Psychological IMPACT of FI

Irrespective of etiology, it has been shown that FI affects the psychological well-being of children. Episodic leakage of stools into the underwear leads to significant stress in children and adolescents (1). Psychological problems, including emotional problems, learning difficulties, separation anxiety, phobias, and depressive, and anxiety disorders were reported to be associated with FI (2,3). In addition, behavioral issues, such as internalization and externalization, are also reported more frequently in children with FI (18); however, a causal association between FI and psychological problems has not been proven. Most parents, incorrectly, believe that FI in children is due to laziness or indifference or even an intentional phenomenon. After trying to sort the problem through largely ineffective or inadequate medical interventions, caregivers may have confrontations with the children and ultimately abuse them verbally or physically. Therefore, children with FI more often become victims of child maltreatment (emotional, sexual, and physical) compared with healthy peers (1,3).

CLINICAL EVALUATION

Clinical evaluation often unravels the diagnosis and associated complications in children with FI. The process includes a detailed history and thorough physical examination. Because most children and their parents find it embarrassing to discuss bowel habits with their pediatrician, it is imperative to put them at ease and build an excellent working clinical relationship at the outset. Clinical history should include a detailed description of bowel habits. The nature of the stools is best obtained using the Bristol Stool Form Scale or its modifications (19,20). A child with constipation-associated FI classically has a defecation frequency of <2 per week with hard and painful stools whereas children with FNRFI have regular stools every day in the toilet and occasional (at least once a week) passage of stools into the underwear. The amount of stools noted in underwear is also crucial; typically, a small amount of liquid type stools is most likely due to constipation-associated FI whereas children with FNRFI usually pass a large(r) amount of stools into the underwear. Importantly, the nocturnal loss of feces supports severe constipation, whereas children with FNRFI tend to have incontinence towards the afternoon (7).

Upon physical examination, the presence of an abdominal mass indicates constipation-associated FI. Examination of the lower spine should note asymmetry of the gluteal region, and presence of a sacral dimple (large or close to the anal margin), a tuft of hair as well as scars of repaired myelomeningocele, which may indicate underlying neurological defects (21,22). Anal inspection may note fissures suggestive of associated constipation. Digital examination of the rectum is not needed as general history and examination reveal the diagnosis in most cases; however, in cases of diagnostic uncertainty, digital examination should be performed and can provide information about the integrity of the sphincter complex (eg, lack of tone is a good indicator of a congenitally malformed anal sphincter or neurological dysfunction). The presence of a large fecal mass helps to differentiate between constipation-associated FI and FNRFI (Table 1).

TABLE 1 - Comparison between constipation-associated FI and FNRFI
Feature Constipation-associated FI Functional nonretentive FI
Boys 60% 85%
Nature of usual stools Hard and lumpy Normal formed stools
Amount of incontinent stools Small amounts of either liquid or formed stools Regular or usual amount of stools
Stool frequency <2 stools per week Normal frequency of stools
Large amount of stool Frequent Never
Pain during defecation Frequent Rare
Daytime fecal incontinence Frequent Always
Nighttime fecal incontinence Frequently in severely constipated children Never
Frequent Rare
Abdominal pain May present Not present
Urinary incontinence Often Often
Fecal masses in the abdomen and/or rectum
Perianal examination Perianal feces, redness of skin Perianal feces, redness of skin
Colonic transit time Usually prolonged Normal
Anorectal manometry High sensory threshold Normal
Dyssynergic defecation
Colonic manometry Low frequency of HAPC Normal
No meal-induced contractions
Laxative treatment Usually effective Not effective
FI = fecal incontinence; FNRFI = functional nonretentive fecal incontinence; HAPC = high-amplitude propagatory contractions.

INVESTIGATIONS

Plain Abdominal X-ray

In most clinical settings, it is not uncommon for a plain abdominal radiograph to be performed on encountering a child with a defecation disorder, including FI. Although clinicians believe abdominal radiographs are helpful in diagnosing and directing the management of defecation disorders (23), the sensitivity and specificity are not sufficient to provide the required diagnostic accuracy. Neither the National Institute for Health and Care Excellence nor the European and North American Societies of Pediatric Gastroenterology, Hepatology, and Nutrition guidelines recommend using plain abdominal X-ray for evaluation of defecation disorders (24,25).

Colonic Transit Studies

Children with constipation-associated FI show a variable delay in colonic transit (total and segmental) and anorectal hold-up. In contrast, children with FNRFI have normal colonic transit (11,13,26). A colonic transit study is helpful in differentiating between constipation-associated FI and FNRFI when the diagnosis is uncertain, and the medical history is unreliable (27) (Fig. 2).

F2
FIGURE 2:
Diagnostic algorithm of fecal incontinence.

Anorectal Manometry

Anorectal manometry (ARM) assesses resting and squeezes anal sphincter tone, rectoanal reflexes, rectal sensation, and pressure changes during attempted defecation (28). Children with constipation-associated FI have a higher threshold for rectal sensation than children with FNRFI with no difference in other parameters. This suggests, in the former, a more compliant, dilated rectum with reduced sensation leading to fecal retention (13,14). Contraction of the pelvic floor rather than relaxation during simulated defecation indicates the possibility of dyssynergic defecation (29). The usefulness of ARM in the diagnosis of FI is however limited.

Colonic Manometry

High-resolution colonic manometry is the best available method to study motor and propulsive activity of the colon. Although not widely available, its validity to study the physiological function of the colon in children with defecation disorders is generally accepted. Standard colonic motility patterns in adults have already been described (30). High-resolution colonic manometry in children with severe constipation often reveals generalized colonic hypomotility and the absence of generating high-amplitude propagatory contraction (HAPCs), and an absence of attenuated response to stimulant laxatives. In addition, these children appear to lose the postprandial increase in the cyclic retrograde propagatory motor patterns indicating neuromuscular dysfunction (31). Detecting abnormal wave patterns helps to predict medical and surgical outcomes and plan surgical procedures for segmental motility disorders (30). Children with FNRFI show no abnormalities on colonic manometry (32,33).

Other Investigations

MRI scan of the lower spine is useful to rule out lesions of the spinal cord that could lead to FI; however, most of the time, these lesions are associated with clinically detectable abnormalities (tuft of hair, hemangioma, asymmetry of the gluteal cleft, a scar of a repaired myelomeningocele) of the lower back (21).

MANAGEMENT

General Considerations

Families of children with FI often present to clinicians with a great deal of distress. The clinician must be aware of this fact and provide adequate time for both the children with FI and their families to express their concerns as well as allow for assessment and a management plan to be agreed. At the end of the evaluation, the clinician should provide a detailed explanation, in a nonaccusatory manner, to the child and the parents about the possible reasons for FI, using plain age-appropriate language and simple diagrams (34). This clarity of the context is essential, given both frustration and overprotection are known to associate with constipation and FI (35). This approach further strengthens the therapeutic relationship between the clinician, the child, and the family members and increases successful management (Fig. 3).

F3
FIGURE 3:
Management of functional fecal incontinence (FI) in children.

Dietary Interventions

Dietary Fiber

Children with constipation-associated FI, may benefit from dietary fibers as it acts to soften the stools and reduce its viscosity. Indeed, when the dietary assessment shows inadequate fiber intake (<0.5 g per kg per day), it is advisable to recommend increasing the fiber content to meet the recommended amount as a diet low in fiber could lead to constipation (36,37); however, the European and North American Societies of Pediatric Gastroenterology, Hepatology, and Nutrition guideline does not recommend increasing fiber in children with constipation and sufficient fiber intake (25,38). Similarly, high fiber intake is not recommended for FNRFI (39).

Toilet Training and Cognitive Behavioral Therapy

Inadequate toilet training and poor toilet habits are common contributory factors in developing constipation and associated FI (40). Once established, this aberrant “behavior” leads to painful defecation and voluntary stool retention that further aggravates symptoms of constipation, leading to FI (7). Therefore, regular toilet training is a cornerstone of management (25,39). It is advisable for a child with functional FI to sit on the toilet three times a day, preferably after meals, for 5–10 minutes. Proper seating, relaxed posture, and foot support when appropriate need to be taught to get the maximum benefit from the program. Children with FNRFI should not only sit on the toilet after meals but should also train in the afternoon immediately after coming home from school, because the majority of these children has FI between 3 and 6 pm. Also, there is at least weak evidence that supplementing laxative therapies, the hallmark of treatment, with behavioral interventions such as toilet training, incentive schemes such as star charts, and dietary advice to laxative therapy produces higher success rates in children with constipation-associated FI (41). Approximately 30% of children with functional FI has significant emotional and behavioral problems (42,43). In these children, referral to a clinical psychologist is helpful and increases treatment success.

Biofeedback Therapy

In biofeedback therapy, the patient is taught to understand the normal mechanisms of defecation and helped to gain control over altered physiological mechanisms through instrument-assisted exercises. Comparing conventional treatment alone with conventional treatment plus biofeedback therapy in children with functional FI, biofeedback therapy does not provide an additional advantage of relieving symptoms of both constipation and FI (41). Like constipation-associated FI, biofeedback therapy for FNRFI did not show therapeutic benefits in old studies (18,39); however, a recent study comparing dietary advices and pelvic floor strengthening using Kegal exercises, biofeedback in addition to the dietary advices and Kegal exercises, and transcutaneous posterior tibial nerve stimulation, showed that biofeedback therapy was significantly more successful in decreasing the number of FI episodes and improving the FI score, compared to other two groups.(44). Further studies should explore the value of biofeedback as a therapeutic option for FNRFI.

Pelvic Floor Physiotherapy

Pelvic floor physiotherapy (PFPT) improves children's awareness of bodily sensation while learning to recognize and resolve abnormal functions of the anorectum and the pelvic floor muscles. This therapy aims to teach effective ways of expelling stools, awareness of sensations, and improve pelvic floor muscle function (45). Several studies have reported improvement of symptoms of constipation in clinical trials using PFPT programs (45,46); however, a pragmatic trial in primary care failed to demonstrate the value of PFPT in improving symptoms of constipation (47). The concept and the findings of the initial trials are encouraging, but further evidence is needed to support the use of this therapeutic modality for constipation-associated FI in children.

Pharmacological Interventions

Pharmacological interventions for constipation-associated FI involve two sequential steps: disimpaction and maintenance.

Fecal Disimpaction

Alleviation of fecal impaction in functional FI prevents overflow incontinence. High dose polyethylene glycol is the treatment of choice for fecal disimpaction (48). Sodium phosphate and sodium docusate enemas are also useful to treat fecal impaction. A recent systematic review, including only two trials, found no difference between both interventions for relieving fecal impaction (49).

Maintenance Treatment to Prevent Re-Impaction

Long-term laxative therapy is indicated only for children with constipation-associated FI. Osmotic laxatives are the first-line therapy for the long-term management of constipation-associated FI. Low dose polyethylene glycol is useful in preventing fecal impaction and ensuring the regularity of bowel motions (25). Lactulose is reported to be as effective as polyethylene glycol during the maintenance phase (50). Stimulant laxatives are used in constipation-associated FI in conjunction with osmotic laxatives to enhance bowel motions and prevent fecal impaction. Novel agents such as prucalopride and lubiprostone have not shown clinical benefits in the long-term management of childhood functional constipation (51,52). Laxatives are not indicated in children with FNRFI as the therapy can worsen symptoms (53,54).

Loperamide

Loperamide is a potent synthetic opiate receptor agonist that acts on the μ opioid receptors of the myenteric plexus in the large bowel and slow down gastrointestinal motility. Studies among adults have shown that loperamide reduces stool frequency and urgency, decreases colonic transit and significantly improve resting anal sphincter pressure (55,56). A case study showed the effectivity of loperamide in reducing FI in a child with FNRFI (57). More research is however needed before prescribing loperamide as a standard treatment to children with FI.

Transanal Irrigation

Transanal irrigation systems irrigate the rectum and distal colon to clear accumulated feces. Transanal irrigation is beneficial in children with refractory constipation-associated FI with severe fecal impaction resistant to conventional medical management and FNRFI (58). Using such systems at planned times regularly prevents fecal accumulation and accidental FI. Three studies, including in children with constipation and FI (both organic and functional), have demonstrated improvements in FI when using transanal irrigation (59–62).

Intrasphincteric Injection of Botulinum toxin

In children not responding to oral or rectal laxatives, botulinum toxin injection into the anal sphincter has been used successfully to treat constipation-associated FI. The procedure is safe with minimal complications, including urinary incontinence, pelvic muscle paresis, perianal abscess, pruritus ani and rectal prolapse (63). The downside of botulinum toxin injection is the necessity of general anesthesia and the high costs. In addition, large, randomized placebo-controlled trials are necessary to confirm these positive findings.

Surgical interventions Although most children with FI respond to medical management with counseling, toilet training, and medications, a subset of children, unfortunately, do present with medically resistant FI and need surgical intervention.

Antegrade Continence Enema

With regard to the antegrade continence enema (ACE) procedure, several surgical techniques are used to create a stoma, usually in the cecum, to flush the colon in the proximal to the distal direction. ACE has been shown to be effective in reducing the frequency of FI and improving regular bowel motions in constipation-associated FI (64). The procedure is superior to sacral nerve stimulation for improving bowel frequency (65,66); however, only 15% were able to reverse the cecostomy with complete resolution of symptoms of constipation and hence, constipation-associated FI (67). Complications of ACE are common and include skin excoriation/granulomata, stoma leak, stomal stenosis, surgical site infections, and stomal prolapse (68)

Bowel Resection

Total or segmental resection of the colon has been used as the final therapeutic option for intractable constipation with megarectum (or megasigmoid), recurrent fecal impaction, and associated motility disorders. Two main surgical options have been used; firstly, resection of the dilated sigmoid with colorectal anastomosis or, secondly, resection of the sigmoid and rectum up to the pectinate line with anastomosis of the colon to the lower rectum (69). Other more extensive surgical procedures include colonic resection and ileoanal anastomosis, J pouch ileoanal reconstruction, and the Duhamel procedure. Despite associated complications, these procedures can improve symptoms of intractable constipation and ensure parental satisfaction (66). A systematic review including 83 patients undergoing procedures mentioned above reported 84% of success rate (68).

Other Management Options

Neuromodulation

Neuromodulation is a novel and evolving therapeutic modality in many medical fields, including gastroenterology. A recent systematic review reported a significant improvement in bowel motions with sacral neuromodulation (70). In children with severe, intractable constipation treated with ACE and sacral neuromodulation procedures, the use of sacral nerve stimulation reduced the necessity for antegrade enemas (71). Sacral neuromodulation was also superior to the ACE procedure in reducing FI in children with intractable constipation (65); however, despite reports of success in small studies, more studies involving children in large multicenter RCTs are needed to evaluate the true value of neuromodulation in pediatric practice.

CONCLUSIONS

Functional FI is a significant clinical issue in pediatric gastroenterology. FI carries a considerable physical, psychological, and social burden and reduces the QoL of affected children and their families. The diagnosis of functional FI is based on clinical evaluation and the use of appropriate symptom-based criteria. Sophisticated investigations such as high-resolution ARM and colonic manometry are useful as (research) investigations to shed more light on the pathophysiology of functional FI in children. Oral laxatives are useful in the majority of children with constipation-associated FI whereas, novel therapeutic interventions such as PFPT, intrasphincteric injection of botulinum toxin, and transanal irrigation are being used successfully in more complicated patients who do not respond to pharmacological management. Only in rare cases where all other strategies have failed, surgical interventions are necessary.

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Keywords:

biofeedback therapy; constipation; neuromodulation; nonretentive fecal incontinence; surgical intervention

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