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Examining Sensory Overresponsiveness in Preschool Children With Retentive

Fecal Incontinence

Article  in  The American journal of occupational therapy.: official publication of the American Occupational Therapy Association · July 2017
DOI: 10.5014/ajot.2017.022707

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 Examining Sensory Overresponsiveness in Preschool
Children With Retentive Fecal Incontinence
Isabelle Beaudry-Bellefeuille, MScOT, is PhD
Candidate, University of Newcastle, School of Health
Sciences, Callaghan, New South Wales, Australia, and
Occupational Therapist, private practice, Oviedo, Spain. At
the time of the study, she was MOT Student, Virginia
Commonwealth University, Richmond; ibbergo@gmail.com
Shelly J. Lane, PhD, OTR/L, FAOTA, is Professor,
University of Newcastle, School of Health Sciences,
Faculty of Health and Medicine, Callaghan, New South
Wales, Australia. At the time of the study, she was
Professor, Department of Occupational Therapy, School of
Allied Health Professions, Virginia Commonwealth
University, Richmond.
7105220020p1
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Isabelle Beaudry-Bellefeuille, Shelly J. Lane
The development of bowel control is an important activity of daily living in early childhood, and challenges in
this area can limit participation in key occupations. Retentive fecal incontinence (RFI) is a common disorder in
children. Up to 50% of children do not respond adequately to initial medical intervention, and behaviors
around toileting, some related to sensory overresponsivity (SOR), may be partly responsible. Therefore, this
study investigated the relationship between RFI and SOR and also examined the discriminative validity of the
Toileting Habit Profile Questionnaire (THPQ). Per parent report, children with RFI (n 5 16) showed
significantly more behaviors related to SOR compared with typically developing children (n 5 27). In
addition, results indicated that the THPQ effectively discriminates between children with RFI and typically
developing children. Results are discussed regarding RFI and SOR, the impact of RFI on childhood
occupational engagement, and the role of occupational therapy with this population.
Beaudry-Bellefeuille, I., & Lane, S. J. (2017). Examining sensory overresponsiveness in preschool children with retentive fecal
incontinence. American Journal of Occupational Therapy, 71, 7105220020. https://doi.org/10.5014/ajot.2017.022707
T oileting, including bowel management, is an important activity of daily living
(ADL; American Occupational Therapy Association, 2014), and issues in
this area can limit a child’s independence and social participation. Additionally,
acquiring continence of bowel is considered an important milestone of child-
hood, generally achieved by age 3 (Schum et al., 2002). Therefore, addressing
issues related to bowel management is an important component of occupational
therapy practice.
Background
Retentive fecal incontinence (RFI) is a common disorder in children that often
leads to impaired social acceptance, relationships, and development (Friman,
Hofstader, & Jones, 2006). Of the main symptoms of constipation, RFI occurs
when soft stool from the bowel slips around a hard mass of stool that remains
accumulated in the rectum (Cohn, 2011). Constipation is one of the most
common gastrointestinal complaints in children (Tabbers, Boluyt, Berger, &
Benninga, 2011); worldwide prevalence is estimated to be 12% in the general
childhood population, with peak incidence occurring during toilet training
(Mugie, Benninga, & Di Lorenzo, 2011). Compared with typical children,
children with long-lasting symptoms of constipation are at risk for experiencing
lower health-related quality of life as adults, including ongoing health concerns
and body pain (Bongers, van Dijk, Benninga, & Grootenhuis, 2009).
Although the cause of RFI has not been clearly identified, behaviors have
been identified that seem to be partly responsible for the development and
maintenance of the condition (Benninga, Voskuijl, Akkerhuis, Taminiau, &
Büller, 2004; Cox, Morris, Borowitz, & Sutphen, 2002; van Dijk, Benninga,
September/October 2017, Volume 71, Number 5
 Grootenhuis, & Last, 2010). Stool-withholding behavior
has been recognized as one of the most common causes of
development and maintenance of childhood constipation
(Cohn, 2011; Tabbers et al., 2011; Whitehead, di Lorenzo,
Leroi, Porrett, & Rao, 2009). Stool toileting refusal or
fear of sitting on the potty are other identified behav-
ior problems in children that have been associated with
constipation, stool withholding, late toilet training, and
fecal incontinence (Blum, Taubman, & Nemeth, 2004;
Taubman, 1997; Taubman, Blum, & Nemeth 2003).
Refusal behaviors are also reported to contribute to the
maintenance of RFI because they interfere with conven-
tional medical management (Karagiozoglou-Lampoudi
et al., 2012; Kaugars et al., 2010; Kuhl, Felt, & Patton,
2009; Sullivan, Alder, Shrestha, Turton, & Lambert,
2012; Taubman & Buzby, 1997; Vitito, 2000).
Conventional medical management is the first line of
treatment of constipation and RFI, often consisting of
dietary recommendations; stool softener, laxative medi-
cation, or both; and implementation of toileting routines
(Cohn, 2011). Currently, only about 50% of patients
are free of complaints and off laxatives after 6–12 mo of
medical management (Pijpers, Bongers, Benninga, &
Berger, 2010). Although identified behavioral issues re-
lated to RFI are not necessarily causal, closer examination
of the problem behaviors associated with constipation
and RFI could contribute to developing more effective
treatment programs to augment conventional medical
management.
It has been hypothesized that difficulty processing
and integrating sensory information could be a factor
contributing to the development of certain problematic
behaviors in children (Dunn, 2007; Hazen et al., 2008;
Roberts, King-Thomas, & Boccia, 2007; Schaaf et al.,
2010). For example, exaggerated responses to normal
sensory stimuli, often referred to as sensory overresponsivity
(SOR), have been associated with refusal to comply with
parental demands or atypical habits regarding other types
of self-care activities (Cermak, Curtin, & Bandini, 2010;
Chatoor, 2002; Dunn, 2007; Hazen et al., 2008; Nadon,
Feldman, Dunn, & Gisel, 2011; Schaaf et al., 2010). It
has also been hypothesized that some of the behaviors
typical of children with RFI could be related to SOR
(Beaudry Bellefeuille & Ramos Polo, 2011; Beaudry
Bellefeuille, Schaaf, & Ramos Polo, 2013; Handley-
More, Richards, Macauley, & Tierra, 2009). In addition,
SOR has been documented to be associated with other
gastrointestinal dysfunctions (Bakker, Boer, Benninga,
Koelman, & Tijssen, 2010; Mazurek et al., 2013). For
instance, children with dysfunctional elimination syndrome
(DES), a disorder characterized by abnormal bowel and
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bladder voiding in the absence of anatomical or neuro-
logical disease, have been found to have more sensory
responsivity difficulties than typically developing chil-
dren, providing evidence of a link between toileting issues
and sensory responsivity difficulties (Pollock, Metz, &
Barabash 2014).
At present, there is little documentation of a potential
relationship between SOR and RFI. Therefore, the fo-
cus of this study was to investigate this relationship
and establish initial clinical validity of the Toileting
Habit Profile Questionnaire (THPQ; Beaudry-Bellefeuille,
Lane, & Ramos-Polo, 2016), a tool designed to screen for
sensory-based toileting difficulties.
Method
Research Design
We examined construct validity using a known-group
design to determine the ability of the THPQ to distinguish
between children with identified RFI and typical children.
Subsequently, scores from both the typical children and
the children with RFI were analyzed together using a
correlational study design to explore the relationship
between THPQ scores and SOR scores. Parents of 3- to
5-yr-old children with RFI were asked to complete two
different questionnaires: the THPQ and the Spanish
version of the Short Sensory Profile (SSP; McIntosh,
Miller, Shyu, & Dunn, 1999) revised for Spain (S–SSP–R;
Beaudry-Bellefeuille & Lane, 2015). A comparison
group of parents of typical children, age and gender
matched, was also recruited and asked to complete both
questionnaires.
Participants
A conservative estimate based on clinical experience with
the THPQ showed an expected difference of 7 points in
the mean scores of typical children and children with RFI.
Using this estimate and N-Query Advisor® (STATSOLS,
Boston, MA) sample size calculations for a Wilcoxon
rank–sum test, we determined that a minimum of 19 par-
ents should be included in each group. Regarding the
correlation of TPHQ and SOR scores, sample size cal-
culations for an expected correlation of 0.4, with a
power of 0.8, were done using GpPower version 3.1.5
(Heinrich Heine University, Düsseldorf, Germany), and
a sample size of 46 was recommended.
Accordingly, over a period of 4 mo, efforts were made
to recruit two groups of 25 parents of children ages 36–
71 mo. Local pediatric gastroenterologists and occupational
therapy practitioners from both public and private clinics
7105220020p2
 were contacted for recruitment of parents of children Table 1. Participant Descriptive Statistics
with RFI who had not responded to an initial trial of Frequency, n (%)
conventional medical treatment by the child’s pediatri- Descriptive Variable RFI Group (n 5 16) TYP Group (n 5 27)
cian. Parents of typically developing children were re- Participant
cruited through the parent–student associations of local Father 4 (25.00) 0
schools. The parent of each child signed an informed Mother 12 (75.00)* 27 (100)
consent form. Approval by the Virginia Commonwealth Children
Boy 10 (62.50) 14 (51.85)
University institutional review board was obtained before
Girl 6 (37.50) 13 (48.15)
the beginning of the study. Child’s age, yr
Parents whose 3- to 5-yr-old children experienced fecal 3 6 (37.50) 6 (22.22)
incontinence as part of the symptoms of functional con- 4 7 (43.75) 12 (44.44)
stipation according to Rome III criteria (Drossman, 2006) 5 3 (18.75) 9 (33.33)
and had no other diagnosis were included in the RFI group. Child’s school
Public 11 (68.75) 19 (70.37)
Diagnosis of RFI and screening for medical conditions was
Subsidized private 4 (25.00) 6 (22.22)
done by the child’s referring physician as part of the stan- Fully private 1 (6.25) 2 (7.41)
dard medical management of constipation and fecal in- Annual family incomea
continence. If parents with children with suspected RFI ³$5,500 15 (93.75) 25 (92.59)
were interested in participating in the study and were re- <$5,500 1 (6.25) 2 (7.41)
ferred by sources other than physicians, their children Parent education level
Primary school 1 (6.25) 1 (3.70)
were referred for medical screening by a physician before
Middle school 0 4 (14.81)
participation. High school 4 (25.00) 8 (29.63)
Children with organic causes of RFI, including University 11 (68.75) 14 (51.85)
Hirschsprung’s disease, muscle disorders, prior rectoanal Note. Percentages may not total 100 because of rounding. RFI 5 retentive
surgery, spina bifida, mental retardation, or hypothyroid- fecal incontinence; TYP 5 typically developing.
a
Per family member.
ism, were not eligible. For both the RFI and the typical
*p < .05.
group, parents of children with intellectual disability; neu-
rological conditions, such as cerebral palsy; or psychiatric
to help differentiate between typical defecation behaviors
disorders, such as bipolar disorder or oppositional defiant
and behaviors associated with constipation and fecal in-
disorder, were excluded. In Spain, 3- to 5-yr-old children
continence potentially related to sensory responsivity
are typically enrolled in preschool programs, which screen
concerns. Items are organized into two sections: an SOR
for developmental disorders. Therefore, children who had a
section, which contains 8 items, and a sensory under-
curricular adaptation at school or who qualified for their
responsivity section, which contains 2 items (Figure 1).
school’s special needs program were excluded. Public health
This study focused on questions in the SOR section.
services pediatricians also periodically screen children for
Scored using a 5-point Likert scale (ranging from 1 5
mental health and developmental disorders and refer them
almost always to 5 5 never), this tool underwent face and
to early intervention programs accordingly. Therefore,
preliminary content validation using a panel of experts
children who had been referred to these programs were
consisting of pediatric gastroenterologists and occupa-
excluded.
tional therapists with expertise in Ayres Sensory In-
The recruitment period yielded 16 participants for the
tegration® (Beaudry-Bellefeuille et al., 2016).
group comprising parents of children with RFI. For the
Short Sensory Profile. The S–SSP–R was used to assess
group comprising parents of typically developing children,
sensory responsivity abilities and concerns. The SSP is a
27 participants were recruited. Because sample sizes of 25
38-item condensed version of the original 125-item
were not achieved for both groups, the data have been
Sensory Profile (SP; Dunn, 1999). Both the SP and the
analyzed and treated as a pilot study to provide infor-
SSP are caregiver questionnaires that measure responses
mation for a larger study in the future. Participant de-
to sensory events in daily life in children ages 3–10 yr.
mographics are presented in Table 1.
The validity of these tools is well established. The data
provided by these questionnaires allow clinicians to an-
Measures alyze how certain patterns in sensory processing may be
Toileting Habit Profile Questionnaire. A caregiver report related to performance and participation difficulties in
tool, the THPQ was designed as a screening questionnaire daily occupations (Dunn, 1999). The Spanish version

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Figure 1. Items from the Toileting Habit Profile Questionnaire
(Beaudry-Bellefeuille, Lane, & Ramos-Polo, 2016).
was originally aimed at Spanish speakers in the United
States. Verification of the validity of the Spanish version
in Spain was done through cognitive interviewing of care-
givers and review by an experienced linguistic consultant
(Beaudry-Bellefeuille & Lane, 2015). The newer version of
the SP, the SP2 (Dunn, 2014), was not available at the time
of this study.
Data Analysis
Between-group comparisons (children with and without
RFI) on the THPQ and the S–SSP–R were conducted
using the Mann–Whitney U test. Differences were con-
sidered statistically significant at p < .05. A Spearman
correlation coefficient was calculated to analyze the rela-
tionships between the SOR score and the THPQ score.
Because scores are based on ordinal data, nonparametric
statistical tests were chosen.
Because the purpose of the study was to develop a better
understanding of the relationship between RFI and SOR,
Figure 2. Boxplot of Toileting Habit Profile Questionnaire scores.
Note. RFI 5 retentive fecal incontinence; THPQ 5 Toileting Habit Profile Questionnaire; TYP 5 typically developing.
The American Journal of Occupational Therapy
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data analysis used only a subset of relevant items from the
THPQ and the S–SSP–R. For the THPQ, the analysis
included the score obtained on the SOR section of the
questionnaire. For the S–SSP–R, analysis was done as
described by Mazurek and colleagues (2013). An SOR
score was calculated using the items designed to detect
overresponsivity: tactile sensitivity (Items 1–7), taste/smell
sensitivity (Items 8–11), movement sensitivity (Items 12–
14), and visual auditory sensitivity (Items 34–38).
Results
Between-Group Differences
Lower THPQ scores are more indicative of behaviors
hypothesized to be specific to children with RFI. In the
THPQ 8-item SOR section, 40 represents the maxi-
mum possible score and 8 represents the minimum
possible score. Results of the Mann–Whitney U test
analysis indicated a significant difference between children
with RFI (median [Mdn] 5 23.50, first quartile [Q1] 5
17.50, third quartile [Q3] 5 28.00) and the typical group
(Mdn 5 36, Q1 5 34, Q3 5 38; p 5 .000). Results are
represented in Figure 2.
An SOR score was calculated from the S–SSP–R using
the items designed to detect overresponsivity. Lower SOR
scores are indicative of greater SOR. Results of the Mann–
Whitney U test analysis indicated a significant difference
between median scores for children in the RFI group
(Mdn 5 76.50, Q1 5 71.25, Q3 5 85.25) and the typical
group (Mdn 5 84.00, Q1 5 76.00, Q3 5 90.00; p 5
.0346). Results are represented in Figure 3.
7105220020p4
 Relationship Between Scores
A Spearman correlation between SOR scores and THPQ
scores produced rs 5 .423, which confirms a moderate but
significant (p 5 .005) linear dependency between SOR
and THPQ scores.
Discussion
Fecal incontinence and constipation are two of the most
common gastrointestinal complaints in children (Tabbers
et al., 2011), and success rates for conventional medical
treatment of children with these concerns remains limited
(Pijpers et al., 2010). Identifying underlying factors that have
not previously been considered is crucial to improve treatment
outcomes. Results from this pilot study suggest that SOR is
one of these underlying factors and should be considered in
children with conventional medical treatment–resistant RFI.
In addition, because occupational therapy practitioners have
expertise in occupational engagement and sensory modulation
disorders such as SOR, they have a role to play in identifying
and treating sensory concerns in children with RFI.
The age range of 3–5 yr was chosen for multiple reasons.
First, studies have shown that most children acquire fecal
continence by approximately age 3 yr (Schum et al., 2002;
Wald et al., 2009), and ongoing toileting concerns become
apparent in children in this age range. Second, it has been
hypothesized that the symptoms of feeling pain upon def-
ecation and toilet avoidance may be more significant at
younger ages (Borowitz, Cox, & Sutphen, 1999). Clinically,
it appears that children who have long-standing constipation
habituate to the sensations that they initially found painful
and motivated them to withhold stool. However, once
Figure 3. Boxplot of sensory overresponsivity scores.
Note. RFI 5 retentive fecal incontinence; THPQ 5 Toileting Habit Profile Questionnaire; TYP 5 typically developing.
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caught in the cycle of constipation and atypical bowel habits,
many struggle to reacquire healthy defecation even in the
absence of pain. Third, clinical experience and research have
suggested that feeling pain upon defecation and toilet
avoidance is related to SOR to the sensations associated with
defecation. Thus, these factors are linked to the onset and
maintenance of the difficulties children with RFI have es-
tablishing healthy toileting routines (Beaudry Bellefeuille &
Ramos Polo, 2011; Beaudry Bellefeuille et al., 2013;
Handley-More et al., 2009). Finally, this age range has also
been reported to be the age of highest prevalence for childhood
constipation (van den Berg, Benninga, & Di Lorenzo, 2006).
No previous studies have specifically examined the
link between RFI and SOR, although related evidence
indicates a relationship between SOR and gastrointestinal
problems. Existing evidence indicates that children with
autism with any type of gastrointestinal problem, includ-
ing chronic constipation, show higher levels of SOR than
children without such problems (Mazurek et al., 2013).
A link between the auditory startle reflex, a measure of
hyperarousal, and irritable bowel syndrome and func-
tional abdominal pain syndrome has also been established
(Bakker et al., 2010). In addition, although SOR has
not been specifically addressed in children with DES, the
broader group of sensory responsivity difficulties has been
found to a greater extent in children with DES than in a
typical population (Pollock et al., 2014), reinforcing the
need for consideration of sensory issues in children with
elimination difficulties. Study findings add to this liter-
ature and support the coexistence of gastrointestinal dif-
ficulties and SOR. They help complete the picture of this
relationship in clinical populations and lay a foundation
September/October 2017, Volume 71, Number 5
 for use of alternative interventions when routine medical
interventions fail.
The results of this study substantiate that the behaviors
described in the first section of the THPQ, meant to explore
SOR, are associated with SOR, as determined by select
items from the S–SSP–R. In addition, this pilot study
provides preliminary support for the discriminative val-
idity of the THPQ in distinguishing between the toileting
behaviors of children with RFI and those of typically de-
veloping children. The THPQ is a unique tool, designed
to screen for sensory-based defecation difficulties and to
explore child behaviors that may contribute to ongoing
RFI—no other clinically available tools tap into these
concerns (Beaudry-Bellefeuille et al., 2016). Response to
sensations related to the evacuation of stool seems to in-
fluence the acceptance of toilet training and the reponse to
the urge to defecate in some children, affecting the de-
velopment of age-appropriate toileting habits (Beaudry
Bellefeuille & Ramos Polo, 2011; Beaudry Bellefeuille
et al., 2013; Handley-More et al., 2009). We previously
established content validity for the THPQ (Beaudry-
Bellefeuille et al., 2016), and in the current study, we
further reinforce the usefulness of this tool in distinguishing
between children with and without RFI.
Problems in bowel management have the potential to
significantly limit a child’s independence and often lead to
impaired social acceptance, relationships, and development
(Friman et al., 2006). The unpleasant odors caused by feces
are often the cause of rejection and ridicule by peers, which
can affect social participation and participation in key
activities of childhood (Handley-More et al., 2009).
Therefore, deepened understanding and identification of
unresolved issues regarding bowel management are crucial
and lay the foundation for appropriate occupational
therapy intervention for this important ADL.
Limitations and Future Research
Although this study adds to the existing literature, it has
limitations. First is the small sample size. Second, because
participants were obtained mainly from private pedi-
atric gastroenterologists and occupational therapists in
one Spanish city, sampling heterogeneity was limited.
Thus, this study should be viewed as a pilot investigation,
warranting further consideration and a larger, more di-
verse sample.
Another limitation to consider is that assessment data
relied on caregiver report; objective assessment data were not
collected. Nonetheless, caregiver report is reported to be a
valid method of data collection in other areas (Feldman
et al., 2005; Harvey et al., 2010) and can be easily used to
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collect data from large groups, an important factor to
consider for future studies. Because of the study design,
the lead investigator (Isabelle Beaudry-Bellefeuille) was not
blind to group membership. Thus, future studies with re-
searchers blinded to condition are needed to gain a better
understanding of the discriminative ability of the THPQ,
the specific sensory issues faced by children with RFI, and
how these issues may affect toileting habits and participa-
tion in daily activities.
Implications for Occupational Therapy
Practice
The results of this study have the following implications
for occupational therapy practice:
• Results provide initial validation for the use of the
THPQ to discriminate between the toileting behaviors
of children with RFI and those of typically developing
children and provide pilot support for the hypothesis
that the behaviors described in the first section of the
THPQ are associated with SOR.
• Occupational therapy practitioners, with expertise in
sensory integration and processing, can make valu-
able contributions as part of the interdisciplinary teams
identifying children with RFI and stool-withholding
behavior, potentially complicated by SOR.
• Identifying RFI in conjunction with SOR can alert
health care providers to consider additional treatment
options for these children, addressing their sensory sen-
sitivities, to optimally support their participation in
healthy and socially acceptable toileting routines. s
Acknowledgments
The authors thank Leroy Thacker for his support in the
analysis of the data.
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