Toilet refusal syndrome (TRS) was first described by Christophersen and Edwards (1) as a common disorder in toddlers who refuse to use the toilet and insist on being given a diaper for defaecation for a duration of 1 month or more. In contrast, micturitions occur without problem on the toilet (1,2). TRS must be differentiated from toilet phobia, in which toilet is refused both for defaecation and micturition (2). So far, TRS is classified as a disorder neither in the International Classification of Diseases-10th Edition (ICD-10) or in the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition (DSM-IV) nor in the International Children's Continence Society (ICCS) or the Rome III pediatric criteria for functional gastrointestinal disorders (3–6).
Despite being a common disorder, affecting 22% of toddlers ages 18 to 30 months, only a few studies have been conducted (7). A total of 23% to 26% children at the age of 17 to 19 months develop TRS during toilet training (8). TRS is associated with late toilet training beyond the age of 42 months. A total of 56.7% of late trained (>42 months) versus 17.9% of early trained children (≤42 months) are affected by TRS (9). In contrast, only 12% of children trained extremely early in their first year of life had TRS (10). These children also needed a longer time to become continent in contrast to those without TRS. Children using the diaper secretly for defaecation also had a higher risk for TRS (11). TRS is temporary in most of the children, but can persist for several years (12). There are no epidemiological data on the occurrence of TRS in older preschool children. Only 1 case report of a 7.8-year-old girl with chronic TRS and an emotional disorder shows how difficult the therapy at this age can be (13).
There is a close association between TRS and constipation. A total of 54% of 27 children with TRS ages 30 to 48 months had constipation (in contrast to 7% of 27 controls), 60% experienced pain during defaecation (vs 15% of controls) (14). On the contrary, constipation can be a risk factor for TRS (15). In 45% of children, faecal incontinence preceded TRS (16). Behavioural problems were not more common in children with TRS compared to controls in 1 study (14), but other family factors such as younger siblings and lack of clear parental rules were associated (7). In older children, the co-occurrence with oppositional defiant disorder (ODD) has been described (2).
Many children with TRS have a good prognosis. TRS resolved without treatment within 6 months in 74% of toddlers ages 18 to 30 months (7). Generally, reduction of familial stress by giving the diaper for defaecation is recommended (7,17,18). Children are allowed to wear the diaper until they develop regular bowel movements and ask for the toilet themselves. A total of 89% of children started using the toilet within 3 months as a result of these means (7). Only if these simple recommendations do not suffice, more intense treatment is recommended especially in cases with stool retention, constipation and faecal incontinence, chronification, older children, comorbid behavioural disorders, familial conflicts, and lack of toilet training (2,7,8). If constipation is present, disimpaction, oral laxatives (especially polyethylenglycol [PEG]) and relaxed sitting on the toilet after mealtimes is recommended (1,2,17). Luxem et al (19) reported positive results by cognitive-behavioural therapy (CBT), regulation of defaecation and mealtimes, laxatives, changes of diet, and increase of oral fluids.
Although simply defined by the use of diapers and refusal of toilet for defaecation for 1 month or more, previous studies seem to describe different types of TRS: toddlers with temporary, benign TRS versus older children with chronic TRS; those with and without constipation; those with behavioural problems and disorders; and those without TRS. In a study on younger children, no significant differences in behavioural characteristics were found between children with and without TRS, but a trend towards a more difficult temperament in children with TRS was discussed (14). Furthermore, another study shows that children with TRS are more affected by critical family constellations (younger siblings) and have parents with problems to set limits (7). In addition, Taubman and Buzby state (16) that a high incidence of family dysfunction is present in those cases, in which TRS continues after the age of 4. Luxem et al (19) discuss the existence of different subtypes of TRS, which could be the result of medical causes such as constipation as well as behaviourally determined.
Owing to the paucity of studies of this common paediatric disorder, 1 aim of this explorative study was to analyse the complete group of children with TRS presented at a tertiary outpatient clinic for elimination disorders. Specifically, it was hypothesised that the presence of behavioural disorders would differ between boys and girls and between children with and without constipation. Earlier studies showed that at preschool age more boys are affected by psychological problems than girls in general (20,21). A second hypothesis was that children with TRS and constipation would differ from children with TRS without constipation regarding behavioural problems. As discussed by Luxem et al (19) we assume that children with TRS and constipation would show less psychological problems than patients with TRS without constipation.
In addition, 3 typical case vignettes are presented illustrating different clinical presentations of TRS. The case vignettes also show different therapy approaches when psychiatric comorbidities were presented in the child additionally.
All of the patients with TRS presented at a specialised outpatient clinic for elimination disorders at a tertiary University Hospital (Department of Child and Adolescent Psychiatry) during the years 2004–2011 were analysed retrospectively. The principles outlined in the Declaration of Helsinki were followed. The procedure was approved by the ethics committee.
All of the examinations were part of the standard clinical procedure in the specialised unit for elimination disorders. No further assessments for research purposes were carried out. All of the patients and their parents were interviewed by an experienced child psychiatrist. The assessment included a comprehensive medical, behavioural, developmental and family history, questionnaires, fluid-volume charts, physical and neurological examinations, ultrasound, uroflow, and urinalysis. All of the medical assessments were executed by a child and adolescent psychiatrist under paediatric supervision. The medical and psychiatric assessments were performed in 1½ to 2 hours. In addition, behavioural symptoms were assessed by the parental Child Behavior Checklist (CBCL 4/18) (22). The 3 composite scores (internalising, externalising behaviour, and total score) were analysed based on the German norms (23). The recommended cutoff for the clinical range was used at a T value of 63 (90th percentile of the normative population).
Enuresis and daytime urinary incontinence were diagnosed according to the ICCS recommendations; constipation was diagnosed in analogy to the Rome III criteria (4,5). After assessment, ICD-10 diagnoses were given based on symptom checklists (6) and mutual consensus conferences by 2 independent raters. After the assessment, parents were informed about the diagnoses and further treatments were initiated.
All of the statistical analyses were calculated with SPSS version 19.0 (SPSS Inc, Chicago, IL). The Fisher exact test was used for the analysis of sex differences and differences between patients with and without constipation. Also, the Student t test for independent samples was used for interval data. Results were considered significant at a level of P = 0.05.
A total of 25 children (10 boys and 15 girls) were included in this study, representing all of the children with TRS presented in the years 2004–2011 from a total sample of 1001 (2.5%) children with elimination disorders. The mean age was 5.2 years (range 3.4–7.3) and did not differ between boys and girls (Table 1).
A total of 44% (n = 11) had nocturnal enuresis, 24% daytime urinary incontinence, and 60% constipation. Forty percent (n = 10) of children (70% of boys, 20% of girls) fulfilled the criteria for at least 1 additional psychiatric disorder according to ICD-10. The most common disorders were emotional (20%), conduct (16%), specific developmental disorders (12%), elective mutism (4%), and tic disorder (4%). Boys also had significantly higher T value scores on the total and internalising, but not on the externalising problems scale of the CBCL. In addition, boys had a total problem score in the clinical range significantly more often than girls (57% vs 8%). A total of 71.4% of boys, but only 33.3% of girls, had a clinically relevant internalising problem score. Regarding the externalising problem score, 28.6% of the boys, but none of the girls, had values in the clinical range.
Constipation was present in 60% (n = 15) of children with TRS—in both boys and girls. There were no differences regarding ICD-10 diagnoses and CBCL scores between children with constipation and those without (Table 1). The following case vignettes illustrate different treatments of TRS when different types of psychological disorders are present.
Different groups of patients with TRS are presented in 3 exemplary case vignettes: First, “classical” TRS with constipation following painful defaecation. Treatment is usually uncomplicated, children are allowed to wear a diaper, but constipation is treated with laxatives and toilet sessions 3 times per day following mealtimes.
A second group is characterised by ODD. These children show oppositional and even aggressive behaviour in many situations—not just by refusing the toilet for defaecation. In these cases, ODD needs to be treated with general CBT and/or parent trainings to reduce problematic behaviour and increase compliance. In addition, TRS needs to be treated separately.
Sibling rivalry (towards younger siblings), an emotional disorder, is a characteristic of a third group. They attract parental attention through their TRS and can even enjoy wearing the diaper—as their siblings do. Parental attention reinforces the TRS. Toilet sessions can be reinforced positively by attractive activities with parents (and without siblings), thereby transferring parental attention from the toilet to other situations.
A 4.9-year-old girl presented because she insisted on a diaper for defaecation. She had been continent at nighttime at the age of 2½ years and at daytime at the age of 4 years. Following an episode of painful defaecation, she refused to use the toilet and was constipated intermittently, which was treated with enemas. Her development was normal, and no comorbid behavioural disorders were present. Paediatric physical examination and ultrasound revealed no abnormalities. Oral disimpaction with PEG was performed, which was given continuously for 6 months at a lower dose. She was allowed to use the diaper for defaecation and performed regular toilet sessions after meals, which were reinforced positively with rewards. Within 6 months, the TRS resolved completely.
A 5.3-year-old boy presented because he required the diaper for defaecation and micturition. He defaecated only once into the toilet at the age of 2.5 years. Without obvious reason, he showed TRS since then. In addition, he acted extremely oppositional and aggressive towards his mother and was a selective eater. In other situations he was stubborn and refused to be examined physically, for example. The initially enlarged diameter of the rectum normalised under laxative treatment (PEG). Because of persisting and severe ODD, outpatient and even day clinic child psychiatric treatment was needed. Only by intense CBT with shaping and positive reinforcement and accompanying counselling of his mother was he finally able to use the toilet for defaecation. The oppositional behaviour was also reduced, but selective eating remained.
A 3.9-year-old girl presented because of defiant behaviour and TRS. She would scream and cry loudly and refuse to use the toilet for her bowel movements. The parents reported that she imitated her 2-year-old brother and was extremely jealous towards him. She wanted all of the attention for herself. If she did not get her mother's exclusive attention, she would react aggressively towards her brother and oppositionally towards her mother. Her mother was counselled on how to deal with sibling rivalry and CBT with positive reinforcement was introduced. Within a few sessions, the TRS resolved completely and she could use the toilet independently.
Other comorbid disorders than ODD and sibling rivalry can accompany TRS, such as separation anxiety, specific phobias during defaecation, and even autism spectrum disorders. In these cases, a specific diagnosis of the comorbid disorder is needed for the treatment to be effective.
TRS is a common, benign, and temporary disorder in toddlers and has mainly been described in primary paediatric settings. TRS usually subsides by the age of 4 years (7). This study demonstrated that TRS can, indeed, persist into preschool (and even school) age. It is a rarer condition at later age affecting only 2.5% of all of the children with elimination disorders presented at a tertiary university clinic. The population-based prevalence is not known. This subgroup of children follows not only a chronic course, but their TRS is more severe and characterised by constipation, comorbid elimination, and behavioural disorders.
Constipation can precede and act as a risk for TRS (15). Constipation (40%) in our group of preschool children was more common than in previous studies of toddlers (24%) (15). More boys (70%) were affected, whereas both girls and boys are equally affected by constipation according to a meta-analysis of 18 studies (24). Constipation is associated with both urinary and faecal incontinence, as in this study, but also with hard stools, abdominal pain, pain during defaecation, and loss of appetite (25,26); however, neither incontinence nor accompanying behavioural symptoms and disorders were significantly more common in those children with TRS and constipation than in those without constipation. Although constipation precedes TRS in younger children (15), causal associations cannot be assessed in this cross-sectional study. Late toilet training beyond the age of 42 months has been reported in children with TRS (9,7), whereas early training is not associated with constipation, stool retention, or TRS (27).
CBCL total score is significantly higher in children with TRS (26.3%) with 5 to 6 times more boys having clinically relevant behaviour problem scores (57%) in the CBCL questionnaire than the normative population (10%), whereas the rates for girls (8%) were not increased. The internalising but not the externalising problem scores were significantly higher in boys.
Children with TRS had a high rate of comorbid disorders (40%), especially boys (70%). The overall prevalence of psychiatric disorders in preschool children is 14% to 26%, 9% to 12% with daily incapacitation (20). This would suggest that children with TRS do have 2 to 4 times more often comorbid disorders than would be expected in this age group. Overall, this would mean that children with TRS have a heterogeneous spectrum of comorbid disorders, with both internalising and externalising symptoms. Mediating factors could include family interactional problems (16). These comorbid disorders could act as a risk factor for TRS in boys, whereas other factors, such as preceding constipation, are likely to be involved in the development of TRS in girls.
Children with TRS are also more affected by further elimination disorders such as enuresis, daytime incontinence, or encopresis. The correct diagnosis of these is just as relevant as the diagnosis of further psychiatric disorders or intellectual disability. These children require more intensive treatment (13).
The heterogeneity of comorbid disorders implies that chronic TRS is associated with different factors, as exemplified in typical case vignettes. In some children, TRS resolves as a long-term consequence of painful defaecation and constipation. In others, externalising problems such as ODD predominate and the TRS can possibly be understood as 1 (of many other) oppositional symptom. In others, especially younger children, introversive problems such as sibling rivalry may maintain TRS. The different predominance in comorbidities in the age groups may be considered as different subtypes of TRS.
A main asset of this study is that all of the consecutive children with TRS presented at a university hospital clinic were included and that standardised instruments such as the CBCL were used. Limitations are the small numbers, the retrospective nature and selection effects through the specific institutional setting. Moreover, cross-sectional data only allow the identification of associations. The analysis of possible aetiological implications would require a longitudinal design.
In summary, TRS in preschool children is a chronic condition with high rates of constipation, as well as accompanying behavioural and emotional disorders. Paediatricians should be aware of these comorbid disorders when a child with symptoms of TRS is presented. In addition to the assessment of possible constipation, a general screening for behavioural symptoms with standardised parental questionnaires (eg, CBCL) is recommended for all of the children with enuresis, urinary, and faecal incontinence—and should include all children with TRS as well (28).
The first therapy recommendation is to reduce familial stress and offer the diaper for defaecation to the child when he or she asks for it. Constipation must be treated by laxatives, change of diet, and increased oral fluid intake. In most cases, this information is sufficient and most children start going to the toilet after some time. In some cases, relaxed sitting on the toilet after mealtimes (toilet training) and positive reinforcement are needed to support therapy. If clinically relevant behavioural scores are present, child psychiatric or psychological assessment should follow. As demonstrated in the case vignettes, counselling and additional treatment of psychiatric comorbid disorders are needed. This should be specific, evidence-based, and follow guidelines and practice parameters (28).
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