Standard Urotherapy for Children with Neurodevelopmental Disabilities: A Systematic Review

Table 2 summarizes all the included articles based on the following components: (a) participants characteristics, including number of participants, type of incontinence problems and relevant NDs, (b) SU component(s) utilized as described by Schäfer et al. (2018), (c) parental involvement, (d) other intervention procedures utilized, and (e) intervention outcomes.

Table 2 Summary of included articlesParticipant Characteristics

Specific child characteristics including relevant NDs were reported in all five articles. In total, 258 children participated in all five studies combined. The sample size ranged from 3 to 111 participants, which included one case study (n = 3) (Smith et al., 2000), while the remaining four studies had a sample size of 30 or higher (Eliezer et al., 2021; Hanney et al., 2013; Kaye & Palmer, 2010; van Laecke et al., 2010).

Of the 258 participants, 158 (61%) participants were boys and 100 (39%) participants were girls. The ages of the participants ranged from 3.6 to 16.0 years (M = 8.5 years). Of the 258 children, 94 (36%) had ADHD, 73 (28%) had a combined ID and physical disability, 42 (16%) had an ASD diagnosis, 22 (9%) participants were motor disabled, and 16 (6%) participants had ID (i.e., 51% had a mild intellectual impairment, 36% were moderately intellectually impaired, 10% had a severe intellectual impairment, and 3% were profoundly intellectually impaired). The remaining disorders mentioned included ODD (6%), anxiety (5%), non-PDD developmental disability (2%), learning disability (1%), and fragile X syndrome (< 1%).

Three studies reported the child’s disorder specification related to ED (Eliezer et al., 2021; Kaye & Palmer, 2010; van Laecke et al., 2010). Of the 225 participants related to the three studies, 91 (40%) participants had DUI, 41 (18%) children had DUI and nocturnal enuresis (NE), 104 (46%) participants had NE, 75 (33%) children had urgency, 66 (29%) reported frequency, and 3 (1%) children had bladder and bowel dysfunction. Smith et al. (2000) reported that one of the three cases had nocturnal enuresis and no specifics were reported regarding the other two children. Hanney et al. (2013) did not report specific incontinence disorders.

Dependent Variable

As indicated previously, Smith et al. (2000) did not report about the dependent variable measured during the study. Two studies reported their findings using the ICCS guidelines regarding percentage of symptom reduction (Eliezer et al., 2021; Kaye & Palmer, 2010). Symptom reduction was measured by obtaining the post-void residual (PVR), the Akbal score (Eliezer et al., 2021), and Pediatric Incontinence Quality of Life (PinQ) score (Eliezer et al., 2021; Kaye & Palmer, 2010). Van Laecke et al. (2010) measured the maximum voided volume, fluid intake quantity, questionnaire scores, and urinary incontinence. Solely one study measured frequency and latency data regarding the following target behaviors: successes, accidents, accident/success conversions, and initiations (Hanney et al., 2013).

SU Components

According to previous studies, SU interventions should include the following components (Schäfer et al., 2018): (1) disorder specification, (2) instructions to establish better micturition, (3) guidelines for fiber and fluid intake, (4) documentation of all symptoms, and (5) a continuous monitoring by the treatment team.

The range of SU components utilized during the included studies varied from one component (i.e., instructions to establish better micturition) by Smith et al. (2000) to all five components by Eliezer et al. (2021) and van Laecke et al. (2010). Kaye and Palmer (2010) utilized two of the five components (i.e., disorder specification and instructions to establish better micturition), while the remaining study (Hanney et al., 2013) applied four out of the five components, as no information was provided regarding the disorder specification.

Another important finding is that the studies had a different execution regarding some of the components. Eliezer et al. (2021) gave a disorder specification by providing a detailed history regarding the child’s incontinence, completed an examination and uroflow, and obtain PVR, Akbal, and PinQ scores. Kaye and Palmer (2010) executed the disorder specification by completing a physician examination, renal and bladder ultrasonography with PVR, urine volume measurement, urinalysis, uroflowmetry with EMG, and PVR. In addition, the treatment of VD was explained. Van Laecke et al. (2010) provided the maximum voided volume, IQ, mental age, verbal capacity, mobility and postural stability, functional autonomy, clinical examination, uroflowmetry, and PVR.

Regarding the instructions to establish better micturition, Eliezer et al. (2021) gave a review of symptoms and utilized a bladder diary for the children. Furthermore, they gave instructions which included an explanation of how bladder filling and emptying occurs and factors that make it more difficult. They advised children to aim for bladder emptying every 3 h during the day and gave specifics regarding the voiding position for boys (i.e., standing with pants down and making a hissing sound) and girls (i.e., seated with feet flat or elevated and leaning forward). Hanney et al. (2013) used an intensive and progressive sitting schedule, differential reinforcement for urination, urine sensor and alarm, positive practice for accidents, and communication training. Kaye and Palmer (2010) started with behavior modifications including timed voiding, bowel management, and positive reinforcement charts. Children started with small compliance goals and progressed to more difficult goals. Smith et al. (2000) used positive reinforcement, shaping (gradually increasing the proximity to the toilet), fading (reducing the presence of the nappy), and a stimulus control transfer. Van Laecke et al. (2010) described using a daytime alarm, wetting alarm, and the toilet that was adapted to the individual needs of the children to optimize a stable toilet position and maximum pelvic floor relaxation.

The execution of guidelines for fiber and fluid intake entailed increased fluids (Hanney et al., 2013) and a fluid schedule, which was individualized based on giving children a target of drinking 30–50 ml/kg per day, and children were advised to avoid drinking within 2 h of bedtime. To avoid irritants, they advised drinking solely water and milk (Eliezer et al., 2021). Eliezer et al. (2021) also provided recommendations to increasing fiber in the child’s diet with grains, cereal, fruit, and vegetables. They advised adding Metamucil and laxatives if there was no improvement.

The documentation of symptoms was completed by providing a bladder diary (Eliezer et al., 2021), or a description of the maximum voided volume, fluid intake quantity, and degree of incontinence (Van Laecke et al., 2010). Furthermore, Hanney et al. (2013) utilized frequency and latency data on successes, accidents, accident/success conversions, and initiations. Kaye and Palmer (2010) solely indicated that they registered during the first visit, but no further details were provided.

Finally, the monitoring by the treatment team was described by Eliezer et al. (2021) as an initial visit, a 3-month visit, and a 6-month visit. The child’s responses were assessed at the 3-month visit. Children with a “no response” classification received combination therapy, adjunct specific urotherapy, or pharmacotherapy. Hanney et al. (2013) described that they transferred the procedures to parents on the first treatment day and provided phone consultation during the following weekend. The implementation at school happened the following Monday, while all therapeutic support was eventually faded as the clients progressed. Services were provided by 20 graduate students. Van Laecke et al. (2010) described that initially the study consisted of 6 weeks with regularly repeated uroflows and bladder scans. Furthermore, additional treatment procedures were considered after a minimum of 6 weeks of treatment. After 3 months with no change, videourodynamics were performed. Kaye and Palmer (2010) did not specify if and how the treatment team monitored the child, besides mentioning the initial visit. The treatment team consisted of a pediatrician, psychiatrist, and/or psychologist. In addition, Smith et al. (2000) also provided no details regarding continuous monitoring of the treatment team, except stating the involvement of an assistant psychologist.

Additional Intervention Procedures

Three out of the five articles started with SU interventions, and additional interventions were implemented after a specific timeframe if SU alone was not sufficient in obtaining the desired result regarding the child’s incontinence (Eliezer et al., 2021; Kaye & Palmer, 2010; Van Laecke et al., 2010).

First of all, Eliezer et al. (2021) indicated that they completed a review of symptoms at the 3-month visit. Based on the child’s response at that time, the child’s progress was maintained on urotherapy alone or adjuncts were considered. Combination therapy was based on the underlying bladder disorder, availability of treatment and patient, and the preferences of the caretaker(s). The adjunctive therapies included desmopressin, alarm training, physiotherapy, anticholinergics, and alpha blockers. Another evaluation took place at the 6-month visit.

Next, Kaye and Palmer (2010) indicated that they did not use a standard treatment approach. Instead they started with applying behavior modification (i.e., timed voiding, bowel management, and positive reinforcement charts) and moved to more advanced goals as the child progressed. If behavior modifications were successful in the sense of performance, but continence was not yet achieved (with normal EMG-PVR findings), anticholinergic pharmacotherapy (i.e., oxybutynin 0.1 mg/kg 3 times daily) was given. Animated biofeedback was given to children demonstrating DV on the EMG-PVR.

Finally, Van Laecke et al. (2010) indicated that after a minimum of 6 weeks of treatment, medical treatment, anticholinergics, and a wetting alarm were considered as additional therapies. After 3 months with no change, videourodynamics were considered.

The remaining two articles described procedures related to specific behavioral modifications. Smith et al. (2000) used positive reinforcement, shaping, fading, and stimulus control transfers, while Hanney et al. (2013) utilized intensive and progressive sitting schedules, increased fluids, differential reinforcement of urination, urine sensor or alarm, positive practice for accidents, and communication training.

Parent Involvement During SU

Hanney et al. (2013) stated that the implementation of the treatment protocol was gradually transferred from the graduate students to the parents and school staff during the first day of treatment at the clinic. In addition, parents and school staff implemented the treatment protocol during all subsequent days in the home or at school. In order to ensure that parents and school staff were able to implement the procedure, parents and school staff were trained on implementing the protocol by the treatment team using instructions, and modeling and performance feedback was given.

Kaye and Palmer (2010) involved parents during the first treatment sessions by stating clear expectations for treatment by explaining that the treatment of incontinence is a process (especially in children with ADHD), and therefore no immediate results should be expected after one visit or with one sole intervention. They clearly asked parents if they were willing to continue treatment and indicated that if parents were mentally or logistically unable to commit at that time, they could opt to return at a later time. Furthermore, treatment was individualized based on the input of the parents, and they concluded that parent involvement (as well as the involvement of a pediatrician and psychologist) was essential for successful treatment.

Van Laecke et al. (2010) indicated that parents were involved during treatment by giving parents and caretakers guidelines on fluid quality and quantity. The remaining articles (Eliezer et al., 2021; Smith et al., 2000) did not mention if and how they involved parents during treatment.

Intervention Effectiveness

An updated report was released by the ICCS regarding the use of certain terminology and definitions of treatment outcomes (Austin et al., 2016). According to these guidelines, researches should report initial study findings as follows: (a) no response, < 50% symptom reduction; (b) partial response, 50–99% symptom reduction; (c) complete response, 100% reduction. It is important to state that one article reported the findings in accordance with these guidelines which made it possible to compare this study based on a uniform standard (Eliezer et al., 2021). Eliezer et al. (2021) stated the following study outcomes after the 6-month visit: (a) nine children (24%) had a complete response to SU alone and four (11%) with adjuncts, (b) three children (8%) had a partial response with SU alone and seven (19%) with adjuncts, and (c) five children (14%) with SU alone had no response, while four children (11%) with adjuncts had no results. This indicated that at the 6-month follow-up, 32% of the children had a complete/partial response to SU alone. Type of underlying behavioral disorder did not influence the outcome.

Kaye and Palmer (2010) also reported their findings according to the ICCS criteria at the time; however, these requirements have been updated since the publication of that specific study. The results of that study according to the guidelines at the time of publication were as follows: (a) of the 56 patients with ADHD, 17 (30%) achieved a complete response—with behavior therapy alone (n = 7), with anticholinergics (n = 2), and biofeedback (n = 8); (b) 30 patients with ADHD (54%) achieved partial improvements—with behavior therapy alone (n = 9), with anticholinergics (n = 15), and biofeedback (n = 6); finally, (c) 9 of 56 patients with ADHD (16%) had no results even with all three interventions (behavior therapy, anticholinergics, and biofeedback). This entails that at the end of the study, 29% of the children had a complete/partial response to SU alone. The authors suggests that noncompliance by the parents may have been reduced by asking parents if they were mentally and logistically able to commit to treatment and therefore the success of treatment might have been affected by the patient selection bias.

Van Laecke et al. (2010) reported the outcome findings based on the ICCS definitions regarding continence and determined the statistical significance in continence patterns at the start and end of the study. At the end of the study, 69 children (67%) were completely continent (day and night). Fourteen children (14%) remained urinary incontinent (day and night), while five children (5%) had DUI and fifteen children (15%) had NE. No significant differences in continence results were found among the study subgroups (i.e., mentally disabled, motor disabled, and mentally and motor disabled). As this study additionally looked at adequate fluid intake, it was found that 66% of the children drank at least 25% less than the physiologically necessary quantity.

Hanney et al. (2013) described the findings of the study in terms of the level of continence: (a) full continence—independent initiation of successful voids with no accidents, (b) partial continence—inconsistent successful voids, (c) schedule-dependent continence—successful voids based on prompts (every 3–4 h), and (d) unknown—insufficient data. Of the initial 30 participants, 14 children with ASS (47%) obtained full continence with an average of 14.4 days of treatment, 3 children with ASS (10%) obtained partial based continence, and another 3 participants with ASS (10%) were schedule-dependent continent at the end of the study. Overall, for all participants, the mean time off the toilet steadily increased during treatment.

Finally, the results from Smith et al. (2000) should be interpreted with caution as the critical appraisal of this article was insufficient. Individual findings of the three cases were reported in the study. Child 1 completed the program in 54 weeks and at the end of treatment no soiling occurred at home or at school. Child 2 was in treatment for 130 weeks at which point the child was able to defecate on the toilet without a nappy. Finally, child 3 completed the program in 51 days when the child was able to urinate in the toilet without a nappy. Important to note is that this study suggested that children with learning disabilities might be more prone to treatment resistance which may explain the need for a longer treatment time for these children. The hypothesis is that this resistance to change is not solely due to simply failure to learn or a skill deficit; therefore, other factors should be considered during treatment (e.g., anxiety).

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