ORIGINAL CONTRIBUTION

Quality-of-Life Assessment in Children With Fecal Incontinence Humberto S. Filho, M.D., M.Sc1 • Roberto A. Mastroti, M.D., Ph.D.2 Wilmar A. Klug, M.D., Ph.D3 1 Department Pediatric of Irmandade da Santa Casa de Misericórdia de São Paulo, São Paulo, Brazil 2 Department Pediatric of Santa Casa de São Paulo Medical School, São Paulo, Brazil 3 Department of Surgery of Santa Casa de São Paulo Medical School, São Paulo, Brazil

BACKGROUND:  Fecal incontinence is a clinical condition that causes embarrassment and changes the perception of quality of life. The absence of a specific tool for assessing fecal incontinence in children led us to adapt an instrument originally developed for adults, which has already been validated into Portuguese. OBJECTIVE:  The purpose of this work was to evaluate the quality of life of children with fecal incontinence. DESIGN:  This is a single-center, prospective study based

on the application of survey. The Fecal Incontinence Quality of Life questionnaire was modified by eliminating 2 questions related to sexuality and by substituting the word “depressed” with “sad” in the statement, “I feel depressed.” SETTING:  The study took place at a tertiary academic medical center. PATIENTS:  Forty-one children >5 years of age, with incontinence of organic etiology and preserved cognition but without stomy, were interviewed with the use of the Fecal Incontinence Quality of Life modified questionnaire. To evaluate the discrimination validity, 28 healthy children were interviewed as control subjects. MAIN OUTCOME MEASURES:  As to reproducibility, a test/retest was performed, involving 25 children. For construct validation, the Fecal Incontinence Quality of Life modified was correlated with the generic instrument Autoquestionnaire Qualité de Vie Enfant Imagé with the continence index São Paulo Score of Continence.

Financial Disclosure: None reported. Correspondence: Humberto S. Filho, M.D., M.Sc., Rua Pedrinhas 177, Vila Inah, São Paulo-SP, 05616-09, Brazil. E-mail: scorrsalgado@uol. com.br Dis Colon Rectum 2015; 58: 463–468 DOI: 10.1097/DCR.0000000000000324 © The ASCRS 2015 Diseases of the Colon & Rectum Volume 58: 4 (2015)

RESULTS:  The average values by scale included lifestyle, 3.1; emotional, 2.8; behavior, 2.3; and embarrassment, 1.6. The average values for the control group included lifestyle, 3.7; emotional, 4.0; behavior, 3.6; and embarrassment, 3.6. The instrument showed a general reliability of 0.78, measured by the Cronbach α. Reproducibility was also >0.90 according to the Cronbach α. LIMITATIONS:  The intrinsic characteristics of children include their constant growth, and this presented a challenge in our search for an instrument that permitted us to identify and measure these variations. CONCLUSIONS:  The experiment showed a reduction in all of the scale values, particularly for embarrassment in children with fecal incontinence. KEY WORDS:  Anorectal anomalies; Fecal incontinence; Hirschsprung disease; Quality of life; Quality of life in fecal incontinence.

F

ecal incontinence is an incapacitating and embarrassing clinical condition, of significant socioeconomic repercussion, and, because of the embarrassment it causes, patients hide its symptoms.1,2 As with other chronic diseases, its evolution can be followed up by physiological variables, which has proven insufficient.3 The patient's point of view on how the disease and the treatment interfere with his or her life has been the concern of several medical teams over the past several years.4 Subjective questions that are related to the disease and its treatment and characterized as the sensation of well being are grouped together as quality of life (QoL) related to health.5–7 The lack of a specific instrument for assessing fecal incontinence in children motivated us to adapt the Quality of Life in Fecal Incontinence (FIQL) instrument that was originally developed for adults. It was validated in Brazil by Yusuf et al1 and modified by us with the removal of 2 items that made reference to sexual activity. 463

464

Our objective was to evaluate QoL related to health in children with fecal incontinence by using the FIQL modified for children (FIQLm).

PATIENTS AND METHODS This was a prospective study based on the administration of the questionnaire during outpatient treatment. The consent was expressed in writing, and the project was appraised and approved by the Irmandade da Santa Casa de São Paulo Central Hospital Commission of Ethics in Research. The interviews with 41 children >4 years of age were collected during their follow-up at the Pediatric Coloproctology Health Clinic at the Pediatric Surgery Service at Santa Casa. Regardless of the etiology and proposed treatments, they remained incontinent, but were free of stomas, and had competent cognition to answer the questionnaire. The control group was composed of 28 children, with age and sex distributions similar to those in the study group. The FIQL was modified by suppressing 2 items relative to sexuality and by substituting the word “depressed” with “sad” in the answer/statement, “I feel depressed.” The modified instrument is composed of 27 questions contained in 4 domains or scales. Lifestyle includes 2, 2b, 2c, 2d, 2e, 2g, 2p, 3b, 3l, and 3k, related to leisure and social activities. Behavior includes 2f, 2i, 2j, 2k, 2m, 3d, 3j, and 3, verifying the degree of incontinence and anxiety. Emotional includes 1, 3c, 3f, 3g, 3i, 3h, and 4, related to the evaluation of one’s own health and self-esteem. Embarrassment includes 2l, 3a, and 3e, showing patient isolation. The answer scores vary from 1 to 4 points, except for question 1, which oscillates from 1 to 5, and question 4, which goes up to 6, with both of these questions belonging to the emotional domain. The worse the symptom/ dissatisfaction, the fewer points the question receives. The final result is obtained by calculating the average. To evaluate the QoL health-related instrument, some measurement properties should be tested, including reliability or reproducibility, discriminant validity, and convergent validity.3,8 Reproducibility

The reproducibility of the study was investigated by means of 3 interviews. Twenty-seven children participated in this phase. To evaluate the interobserver reproducibility, 2 interviewers conducted the interview on the same day, with a time interval that varied by ≈40 minutes. The intraobserver reproducibility was evaluated through the readministration of the questionnaire in the following weeks, as long as there was no change in the patient’s clinical state.

Filho et al: QoL in Children With Fecal Incontinence

Convergent Validity

The FIQLm was correlated with the São Paulo Score of Continence (SPSC), an index used at our health clinic for measuring and grading the intensity of fecal incontinence, as well as physiological variables, the need for enemas, and the impact on the lifestyle of the child. This indicator scale varies from lower scorings for the continent to the seriously incontinent, considering incontinent those whose scores are >8 (Fig. 1). Still focusing on the convergent validation, another generic instrument was applied, the Autoquestionnaire Qualité de Vie Enfant Imagé (AUQEI), which was validated in Brazil in 2000 by Assumpção Jr et al.9 This instrument allows for a self-evaluation of the child by using images and limiting the population by age, with a minimum age of 4 years and a maximum age of 12 years. Each question presents a domain and 4 answers represented by faces that express different emotional states. Initially, the children are asked to report an experience from their lives that is pertinent to each of the alternatives, which allows them to understand the situation and express their feelings. Subsequently, the children are asked to mark, without a time limit, the answer that best corresponds with their feeling toward the domain in question.10 The instrument is composed of 26 questions, which grade the answers between 0 (very unhappy) and 3 (very happy), and encompass the child domains and relate to social and school activities, corporal functions, health, and so on. This instrument has a CI of 95% and a cutoff grade of 48, below which there is detriment to the QoL. Discriminant Validity

The FIQLm was also applied to a control group (28 children) divided into 2 groups, including 13 children who were carriers of colorectal diseases, albeit continent, and another 15 healthy children who were being treated at the outpatient ward for reasons other than problems related to colorectal interventions, for example, umbilical hernia surgery. For the children who underwent colorectal surgery but showed no sign of incontinence, the phrase “due to loss of feces” was substituted with the phrase “due to surgery.” In the healthy control group, in which the participants did not undergo surgery, the statement was altered to “in relation to health.”

RESULTS The incontinent group was made up of 15 girls and 26 boys, with an average age of 10.1 years, varying from 6.0 to 17.0 years. The average age of the control group was 9.4 years, varying from 5.5 to 13.0 years. The distribution, in relation to sex, showed 13 girls and 15 boys. A signifi-

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Frequency of evacuations

>4

4

2 times a week Soiling

Enemas

3

Satisfaction

Activities

weekly

2

No restriction

1

Sporadically

1

Avoids contact

4

Avoids just school

3

Rarely goes out

2

2 times a day

4

1 times a day

3

Socially interferes little

1

2 times a week

2

Does not interfere

0

Weekly

1

Occasionally

0

Behavior

Figure 1.  São Paulo Score of Continence. The sum of the values ​ of each subgroup results in a number ranging from 4 to 23. Incontience scale is as follows: severe = 17 to 23; moderate = 13 to 16; minimal = 9 to 12; and continent = 4a to 8.

cant statistical difference was not observed between the groups (Table 1). In relation to the etiology of incontinence, the distribution was as follows: Hirschsprung disease, 15%; anorectal anomalies, 41%; and myelomeningocele, 44%. The administration of the FIQLm resulted in the following average scores: lifestyle, 3.1; depression, 2.8; behavior, 2.3; and embarrassment, 1.6 (Table 2). The reliability, or internal coherence, of the FIQLm was measured by applying the Cronbach α in the group studied (41 children). The general result was α = 0.79, with a statistical significance of p < 0.001.

Discriminant Validity

To evaluate the discriminant validity, an interview was performed in the control group, which was composed of 28 children, subdivided into 2 other subgroups: group A included children who underwent colorectal surgery but who were continent nonetheless, and group B included children without morbidities. Initially, there were no significant differences between the 2 groups. Later on, in comparing the study group with the control group by using the Student t test, there were observed significant statistical differences in all of the scales (Table 2 and Fig. 2). Construct Validity

Reproducibility

To investigate its reproducibility, the instrument was used to evaluate 27 children by different examiners at different times. The application of the Cronbach α among the averages of the domain components in the 3 result sets (A, B, and A1) is shown in Table 3; averages are also shown.

The construct validity, or convergent validity, was investigated by correlating SPSC with the domain (or scale) averages from the FIQLm. By applying the Pearson coefficient, the smallest averages were related to the largest SPSC values in the behavior and lifestyle scales (Table 4). When the comparison was made between the total sum of the SPSC against the total FIQLm by the Pearson

Table 1.   Distribution in relation to sex and age for the incontinent and control groups Incontinent group

Control group

Sex

N

Average age, y

SD

Sex

N

Average age, y

SD

Girls Boys Total

15 26 41

10.0 10.19 10.12

3.566 2.263 1.768

Girls Boys Total

13 15 28

9.69 9.16 9.41

3.276 (NS) 2.485 (NS) 2.836 (NS)

NS = not significant.

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Filho et al: QoL in Children With Fecal Incontinence

Table 2.   Results of the FIQLm for assessing fecal incontinence in the incontinent and control groups Variable

IG

Average

CG

Average

p

Emotional Lifestyle Behavior Embarrassment

41 41 41 41

2.8 3.1 2.3 1.6

28 28 28 28

4.0 3.7 3.6 3.6

>0.05 >0.05 >0.05 >0.05

CG = control group; FIQLm = Quality of Life in Fecal Incontinence modified for children; IG = incontinent group.

Table 3.   FIQLm reliability analysis Variability Emotional Lifestyle Behavior Embarrassment

A

B

A1

α

2.7 3.0 2.3 1.6

3.1 3.0 2.3 1.6

2.7 3.0 2.3 1.5

0.947 0.934 0.952 0.964

Interviews A1 and A stand for the author; B stands for the other interviewer. FIQLm = Quality of Life in Fecal Incontinence modified for children.

coefficient, it showed negative correlation with a statistical significance 48 when confronted with AUQEI. To compare the results between boys and girls, the Student t test was applied, and significant differences were thus identified in 3 domains: depression, behavior, and embarrassment (Table 5). To compare the answers in the different etiologies, the variance analysis (ANOVA) was used. No significant statistical differences among the 3 diseases were observed (Table 6). The correlation between the ages of the children and the FIQLm scale averages was made using the Spearman coefficient (Table 7). It showed a contrast: the older the child, the lower the values of FIQLm in the emotional and behavior domains. The embarrassment domain kept the same trend, which was not observed in the lifestyle domain. Incontinent

GCO

DISCUSSION The real incidence of fecal incontinence in the general population is thus underestimated.11 In children, the difficulties are even greater. Therefore, when we recognize the incontinence, we do not have an available tool to evaluate its impact on child development. A number of affective disorders, social phobia, problems at school of varying degrees, low self-esteem, and isolation are described in children and adolescents with fecal incontinence.12 In pediatrics, the analysis of the characteristics that are pertinent to health-related QoL is hindered by the small number of validated instruments, methodologic variables, and differences found between young and older children.13,14 Trajanovska and Catho-Smith,4 in a revision article detailing the 6 existent instruments for evaluating fecal incontinence in children, affirmed that none is applicable without criticism, leaving only the FIQL that had to be adapted for use in children. Fundamental adaptation sought the subtraction of questions related to sexual activity. Those elements confirmed our choice for the questionnaire that was specifically designed to assess fecal incontinence and validated in Brazil by Yusuf et al.1 Our results showed an average decrease in all the scales, and was most pronounced in the embarrassment domain (1.6). There was also a drop in the averages, accentuated by the emotional (2.8) and lifestyle (2.3) domains. The behavior domain tended to have a smaller decrease in relation to the others (average of 3.1). These findings corroborate the findings of other experiments.15–17 The observation of the measurement properties showed that the reliability of the instrument used was acceptable. The statistical analysis of the group of 41 children also showed a satisfactory internal consistency, measured by the Cronbach α, with a value of 0.78. The reproducibility was investigated with the proposition of the accomplishment of the test/retest, although many authors question the retest in light of the changes in behavior as time passes.9

GCWS

4 3.5 3 2.5 2 1.5 1 0.5 Emotional

Lifestyle

Behavior

Embarrassment

Figure 2.  Comparison of scale scores between children with fecal incontinence and those in the control group. The figure shows a comparison among the children with incontinence with children who underwent colorectal surgery but remain continent and the continent children who did not undergo surgery. GCO = group of children operated; GCWS = group of children without surgery.

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Table 4.   Pearson correlation between SPSC and total sum of FIQLm in 41 incontinent children Parameter

SPSC

FIQLm

SPSC Sign FIQLm Sign

1 – –0.477 0.002

–0.477 0.002 1.000 –

FIQLm = Quality of Life in Fecal Incontinence modified for children; SPSC = São Paulo Score of Continence.

Performed with 27 children, the 2 interviews happened on the same day, with an interval of ≈40 minutes. This time was inferred from observing other tests applied.1 The reproducibility between the first 2 examiners showed a high degree of reliability, with a Cronbach α >0.90, which can be justified by the short time between one interview and the other, thus permitting memorization of the answers. This reasoning does not prove true when the third interview is compared with the first 2, again demonstrating a high score for the Cronbach α (0.964), even with intervals from 1 week to 2 months, with an average of 15 days. Blackwell et al,18 in a study for the validation of a specific instrument for urinary incontinence, came to the conclusion that 1 to 2 weeks would be an adequate period for the patients not to lose the memory of the symptoms. For validation of construct, or convergent validation (in addition to the specific instrument, FIQLm), we used the generic instrument for children (AUQEI), with an age limit that varied from 4 to 12 years.9 Comparison between AUQEI and FIQLm results for the 22 children showed that, in 16 children, there was a decrease in QoL, and the results from AUQEI were also compatible with the drop in QoL. We also correlated the FIQLm with the SPSC incontinence index, which was developed in our department. This revealed an opposite tendency in relation to those 2 indexes. In other words, the larger the SPSC score, the lower the FIQLm average. The study also showed a difference between the averages of scales comparing boys and girls. Except for lifestyle, all of the other 3 areas presented lower values for the boys. This observation was not sustained by other articles in the literature, except for 1 article by Mills et al.19 The reason for this finding is uncertain. Few instruments separately assess the sex of the patients. It is common to Table 5.   FIQLm results among girls and boys (Student t test in 41 children with fecal incontinence) Variable

Boys (N = 26)

Girls (N = 15)

p

Emotional Lifestyle Behavior Embarrassment

2.7 3.0 2.1 1.5

3.2 3.1 2.7 2.1

Quality-of-life assessment in children with fecal incontinence.

Fecal incontinence is a clinical condition that causes embarrassment and changes the perception of quality of life. The absence of a specific tool for...
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