- Email: [email protected]
Quality of life in children with slow transit constipation
Journal of Pediatric Surgery (2008) 43, 320–324
www.elsevier.com/locate/jpedsurg
Quality of life in children with slow transit constipation Melanie C.C. Clarke a , Chee S. Chow b , Janet W. Chase c , Susie Gibb d , John M. Hutson a,b,c , Bridget R. Southwell c,⁎ a
Department of Surgical Research, Royal Children's Hospital, Parkville, Melbourne, Victoria 3052, Australia Department of Paediatrics, University of Melbourne, Melbourne, Victoria, Australia c Murdoch Childrens Research Institute, Parkville, Melbourne, Victoria 3052, Australia d Department of General Paediatrics, Royal Children's Hospital, Parkville, Melbourne, Victoria 3052, Australia b
Received 10 September 2007; accepted 9 October 2007
Index words: Slow transit constipation (STC); Quality of life (QOL); Children; PedsQL
Abstract Background: Slow transit constipation (STC) causes intractable symptoms not readily responsive to laxatives, diet, or life-style changes. Children with STC have irregular bowel motions associated with colicky abdominal pain and frequent uncontrollable soiling. This study assessed the physical and psychosocial quality of life (QOL) in children with long-standing (≥2 years) STC vs healthy controls. Methods: Children (aged 8-18) were recruited from gastrointestinal and surgical clinics and a Scout Jamboree. After informed consent was obtained, the questionnaire (Pediatric Quality of Life Inventory) was administered. This consists of parallel child and parent self-report scales encompassing physical functioning, emotional functioning, social functioning, and school functioning. Higher scores indicate better QOL. P value less than .05 was considered statistically significant. Results: In 51 children with STC (mean, 11.5 years; male/female, 2:1) and 79 controls (mean, 12.1 years; male/female, 1.9:1), Pediatric Quality of Life Inventory QOL score was significantly lower in the STC group (72.90 vs 85.99; P b .0001). In addition, parents of children with STC reported a significantly lower QOL score than their child compared with the child's own report (64.43 vs 72.90; P = .0034). Parents of controls did not (84.25 vs 85.99; P = .12). Conclusions: Slow transit constipation is a debilitating condition affecting both physical and emotional functioning in children. Parental perception of QOL is significantly worse, highlighting the considerable family impact of constipation and uncontrollable soiling. © 2008 Elsevier Inc. All rights reserved.
Since its first description in 1996 [1], slow transit constipation (STC) is slowly becoming accepted as a chronic form of constipation in children [2-6]. It is characterized by intractable constipation that is not readily responsive to
Presented at the British Association of Paediatric Surgeons meeting, Edinburgh, Scotland, July 17-20, 2007. ⁎ Corresponding author. Tel.: +61 3 9345 5805; fax: +61 3 9345 7997. E-mail address: [email protected] (B.R. Southwell). 0022-3468/$ – see front matter © 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2007.10.020
laxatives, diet, or a change in life-style, and there is marked delay in colonic transit time on nuclear transit study [7,8]. The etiology of STC remains unknown with various theories involving neuronal abnormalities [9,10], genetic linkage [5,11], endocrine dysfunction [12], autonomic dysfunction [13], and abnormal colonic pacemaking [14] being postulated. Children with STC have irregular bowel motions, colicky abdominal pain, and frequent uncontrollable soiling. In some children, despite aggressive medical therapy, spontaneous passage of stools is unachievable. Consequently,
Quality of life in children with slow transit constipation Table 1
Demographics of study population
Male/Female Mean age (y) Mean duration of symptoms (y) Appendicostomy Soiling Abdominal pain
STC patients (n = 51)
Controls (n = 79)
2:1 11.5 10.1 14/51 41/51 40/51
1.9:1 12.1 N/A N/A N/A N/A
these children are managed by the surgical formation of an appendicostomy, through which regular antegrade bowel washouts can be performed to improve continence [15,16]. Definitive treatment remains elusive, and as a result, families of children with STC are offered a wide range of management options, many of which are perceived to be unsuccessful. There are many studies that have assessed the quality of life (QOL) in children with chronic disease; however, few studies exist that look at the physical and psychosocial impact of longstanding constipation in children [17]. This study used a validated pediatric questionnaire to assess physical and psychosocial features of life in children with STC.
1. Subjects and methods After the attainment of informed consent, the questionnaire (Pediatric Quality of Life Inventory [PedsQL]) was administered by 1 of 3 investigators (M.C., C.C., or J.C.). The PedsQL 4.0 Generic Core Scales consist of parallel child and parent self-report scales and have been validated in children and adolescents aged 2 to 18 years [18-21]. The questionnaires consist of 23 items encompassing (i) physical functioning (8 items); (ii) emotional functioning (5 items); (iii) social functioning (5 items); and (iv) school functioning (5 items). The categories can then be grouped into physical (i) and psychosocial (ii, iii, and iv) functioning. The questions ask how much of a problem each item has been during the past 1 month. A 5-point response scale is used (0 = never a problem; 1 = almost never a problem; 2 = sometimes a problem; 3 = often a problem; 4 = almost always a problem). Items are reverse-scored (0 = 100, 1 = 75, 2 = 50, 3 = 25, 4 = 0) and linearly transformed to a 0 to 100 scale. Higher scores indicate better QOL. Parent and child completed the questionnaire independently with impartial assistance being provided for any child who had difficulty with comprehension. The local Human Research Ethics Committee approved the study.
321 treated for constipation for a minimum of 2 years. Constipation was defined by the Rome II criteria [22], and all patients had previously had metabolic or hormonal causes excluded. All subjects were diagnosed with STC by radioisotope nuclear transit study [7]. Children with an organic cause for their constipation, cognitive impairment, or anorectal retention/normal transit on their nuclear transit study were excluded. Control subjects were recruited from (i) outpatient surgical clinics and consisted of children (8-18 years) who were attending for routine follow-up after uncomplicated appendicectomy or minor surgical procedure (ie, scrotal exploration, orchidopexy, herniotomy) and (ii) a scout jamboree that took place locally during the recruitment period. All control subjects had a “normal” bowel pattern (between 3 movements a week and 3 movements a day where the stool is brown or golden brown in color, formed, has a texture similar to peanut butter, and a size and shape similar to a sausage). Children who had concurrent comorbidities were excluded along with those who did not describe a normal bowel habit.
1.2. Outcomes The primary outcome measures were self- and parentreported, health-related QOL in children with STC compared with healthy controls.
1.3. Data Analysis Physical, psychosocial, and total QOL scores were compared using independent sample t tests (2-tailed). All P values less than .05 were considered as statistically significant.
2. Results From March 2006 to March 2007, 51 children (34 male, 17 female) with STC and 79 healthy controls (48 male, 31 female) were recruited into the study (Table 1). All children and their parents successfully completed the PedsQL. Child-reported QOL was significantly lower in children with STC compared with normal children (72.90 vs 85.99; P b .0001) (Fig. 1). Parent-reported QOL was significantly lower for children with STC compared with the control
1.1. Study population Study patients were recruited from the gastrointestinal and surgical clinics in a large tertiary pediatric hospital. Patients were between 8 and 18 years of age and had been
Fig. 1
Child-reported PedsQL scores: STC vs control group.
322
Fig. 2
M.C.C. Clarke et al.
Parent-reported PedsQL scores: STC vs control group.
group (64.43 vs 84.25; P b .0001) (Fig. 2). Physical and psychosocial QOL, both child- and adult-reported, were consistently poorer in the children with STC (Figs. 1 and 2). Parents of children with STC reported their child had a significantly lower QOL than the children themselves reported (64.43 vs 72.90; P = .0034) (Fig. 3), whereas parents of controls did not (84.25 vs 85.99; P = .12) (Fig. 4). Parents of children with STC and of controls reported significantly poorer psychosocial QOL for their child compared with the child's own report (Figs. 3 and 4). This was more apparent in the group of children with STC. There was no statistical difference in the reporting of physical QOL (Figs. 3 and 4).
3. Discussion Quality of life can be defined as “an individual's subjective perception of overall wellbeing and satisfaction with life” [23] or “physical, social and emotional aspects of a patient's well being that are important to the individual” [24]. QOL is a multidimensional concept that incorporates the assessment of recognized core domains and indicators. Many studies have looked at the QOL in children with chronic disease; however, there are few studies that have specifically examined the impact of chronic constipation on physical and psychosocial functioning [17]. There are no current publications that address QOL issues in children with proven STC. This is a population of children who have in many instances been managed for several years without a definitive diagnosis and often-ineffectual treatment and for whom social interaction is often limited because of antisocial symptoms (ie, soiling).
Fig. 3
STC group: child- vs parent-reported PedsQL scores.
Fig. 4
Control group: child- vs parent-reported PedsQL scores.
This study found that there appears to be a significant impairment in QOL of children with STC when compared with healthy controls. This is consistently described by the children and their parents and affects both physical and psychosocial QOL (Figs. 1 and 2). Parents of children with STC often describe the onset of their child's symptoms as being at, or shortly after, birth; STC represents as a lifelong problem. These children grow up with the daily problem of difficulty with stooling, chronic abdominal pain, and frequent soiling. Their social interactions are often limited because of the lack of insight of their young peers, and consequently, they can become withdrawn and socially isolated. Although children with STC have no obvious physical anomalies, both the children themselves and their parents report a significant deficit in their physical QOL. Questions included in the physical section of PedsQL ask about participation in sport, ability to take bath/shower by oneself, having hurts/aches, and energy levels. If children experience involuntary passage of stool, or soiling, then participation in sport can often seem daunting. Not only do children have to change in a communal area, where they may inadvertently expose protective clothing or their appendicostomy site [15,25], but they may also experience increased symptoms associated with the increase in physical exertion. Similarly, if children experience severe soiling, then they may require assistance to adequately clean themselves. As previously mentioned, children with STC are often affected by chronic abdominal discomfort. This chronic pain can account for the increased reporting of hurts/aches and also for the consistent reporting of low energy levels. In addition, children affected by the slow passage of stool often feel full and bloated and complain of a poor appetite. Their resultant decreased calorific intake will also contribute to their low energy levels. The psychosocial section of PedsQL is divided into emotional, social, and school functioning. Emotional functioning covers feeling afraid, sad, or angry, as well as troubles in sleeping and worrying about the future. Feeling strong emotions and worrying about the future are in many ways features of childhood in general and cannot always be attributed to chronic disease. Despite this, the subgroup of children with STC reported significantly poorer emotional QOL, and in particular, many were concerned about how their future might be affected by their condition.
Quality of life in children with slow transit constipation Table 2
323
Previous results for PedsQL questionnaire in different study populations
Population
Healthy children [19] Healthy children [21] Healthy children [27] Healthy children Not overweight [26] Overweight Obese Orthopedic (acute) [20] Cardiac disease [20] class Ia class IIb class III, IV ADHD [27] STC Cancer [27] Cerebral palsy [27]
Mean child reported score
Mean parent reported score
Total
Physical
Psychosocial
Total
Physical
Psychosocial
79.6 83.9 84.3 86.0 80.5 79.3 74.0 78.1
80.2 87.8 88.0 92.1 85.7 83.5 77.5 75.3
79.3 81.8 82.3 83.3 77.7 77.0 72.1 79.5
80.9 82.3 79.9 84.3 83.1 80.0 75.0 73.7
81.4 84.1 81.8 92.8 87.8 82.6 76.3 72.7
80.6 81.2 78.9 79.7 77.6 76.1 73.9 74.3
83.6 75.9 60.9 70.2 72.9 69.0 66.3
82.1 78.7 58.2 82.6 78.8 65.8 64.8
84.5 74.3 62.3 63.5 69.2 70.8 67.0
86.5 80.1 67.9 69.5 64.4 60.7 56.3
89.5 82.8 66.7 84.6 74.4 56.8 53.3
84.6 78.5 69.0 61.4 58.6 63.1 57.9
Data in bold represent the results of this study. ADHD indicates attention deficit hyperactivity disorder.
Social functioning inquires about the ability of the child to integrate with others. Questions focus on ability to get on with other children as well as social acceptability (other children wanting to be their friend/teasing) and ability to keep up with their peers. Children are often admired for their honesty and integrity; however, it is an undeniable fact that they can also be incredibly cruel especially when they lack insight. Children who are afflicted with chronic soiling are unfortunately easy targets for social ridicule and bullying. Although the responses of the children with STC suggest that they feel they have the ability to keep up with their peers, they have poor QOL scores for social functioning because of their reduced social integration. The final section of psychosocial QOL concerns school functioning. Subjects are asked about their ability to pay attention, memory, keeping up with schoolwork, and missing school either because of feeling unwell or because of the need to visit a medical professional. As previously discussed, some of these questions will score similar answers when asked to all children, especially those relating to paying attention and forgetting things. However, the children with STC again score significantly lower on this section because of poorer school attendance and frequent medical needs. This study also showed that although children with STC and their parents both describe a significantly poorer QOL than healthy children, the parents report a lower QOL in their children than their own children reported themselves (Figs. 3 and 4). There are several reasons why the parents may report a poorer QOL. Parents have an objective ability to look at their children compared with their peers and become distressed by any apparent differences. Because children with STC often have a family history of constipation, their parents may have had similar experiences at school, thus affecting their scoring. As previously mentioned, STC
usually represents a lifelong condition. Children who have STC grow up with their condition and the life-style that it entails and have no other experiences to which they can relate. Although they have the ability to appreciate that their life may not be ideal, it is all that they have ever known and so they make do with things as they are. Parents of children with STC often comment that their child has only one friend, and they do not actively socially interact with their peers. Although a parent may be terribly concerned by this, their child may simply be happy that at least they have a friend. There are several studies that have used the PedsQL to assess health-related QOL in different populations, such as overweight/obese children [26]; children with differing severity of cardiac disease [20]; children with an acute orthopedic injury [20]; children with cerebral palsy, cancer, or attention deficit hyperactivity disorder [27]; and healthy children [20,21,26,27]. These previous results are summarized in Table 2 along with the results for this study (in bold). These results show that QOL in children STC is comparable to children with other chronic illness, including cardiac disease and cancer. In conclusion, this study shows that QOL is significantly lower in children with STC compared with normal children, with both physical and psychosocial functioning scores reduced. Parental perception of QOL is worse when their child is affected by chronic constipation, particularly when estimating their child's psychosocial QOL.
References [1] Benninga MA, Buller HA, Tytgat GN, et al. Colonic transit time in constipated children: does pediatric slow-transit constipation exist? J Pediatr Gastroenterol Nutr 1996;23(3):241-51.
324 [2] Zaslavsky C, De Barros SG, Gruber AC, et al. Chronic functional constipation in adolescents: clinical findings and motility studies. J Adolesc Health 2004;34(6):517-22. [3] Southwell BR, King SK, Hutson JM. Chronic constipation in children: organic disorders are a major cause. J Paediatr Child Health 2005;41(1-2):1-15. [4] Sutcliffe JR, King SK, Southwell BR, et al. Paediatric constipation for adult surgeons—article 1: targeting the cause. ANZ J Surg 2004; 74(9):777-80. [5] Hutson JM, McNamara J, Gibb S, et al. Slow transit constipation in children. J Paediatr Child Health 2001;37(5):426-30. [6] Wald A. Slow transit constipation. Curr Treat Options Gastroenterol 2002;5(4):279-83. [7] Cook BJ, Lim E, Cook D, et al. Radionuclear transit to assess sites of delay in large bowel transit in children with chronic idiopathic constipation. J Pediatr Surg 2005;40(3):478-83. [8] Zuccarello B, Romeo C, Scalfari G, et al. Scintigraphic evaluation of colonic motility in patients with anorectal malformations and constipation. J Pediatr Surg 2006;41(2):310-3. [9] Porter AJ, Wattchow DA, Hunter A, et al. Abnormalities of nerve fibers in the circular muscle of patients with slow transit constipation. Int J Colorectal Dis 1998;13(5-6):208-16. [10] Hutson JM, Chow CW, Borg J. Intractable constipation with a decrease in substance P-immunoreactive fibres: is it a variant of intestinal neuronal dysplasia? J Pediatr Surg 1996;31(4):580-3. [11] Shirasawa S, Yunker AM, Roth KA, et al. Enx (Hox11L1)-deficient mice develop myenteric neuronal hyperplasia and megacolon. Nat Med 1997;3(6):646-50. [12] El-Salhy M, Norrgard O, Spinnell S. Abnormal colonic endocrine cells in patients with chronic idiopathic slow-transit constipation. Scand J Gastroenterol 1999;34(10):1007-11. [13] Knowles CH, Scott SM, Wellmer A, et al. Sensory and autonomic neuropathy in patients with idiopathic slow-transit constipation. Br J Surg 1999;86(1):54-60. [14] He CL, Burgart L, Wang L, et al. Decreased interstitial cell of Cajal volume in patients with slow-transit constipation. Gastroenterology 2000;118(1):14-21.
M.C.C. Clarke et al. [15] King SK, Sutcliffe JR, Southwell BR, et al. The antegrade continence enema successfully treats idiopathic slow-transit constipation. J Pediatr Surg 2005;40(12):1935-40. [16] Stanton MP, Shin YM, Hutson JM. Laparoscopic placement of the Chait cecostomy device via appendicostomy. J Pediatr Surg 2002; 37(12):1766-7. [17] Youssef NN, Langseder AL, Verga BJ, et al. Chronic childhood constipation is associated with impaired quality of life: a casecontrolled study. J Pediatr Gastroenterol Nutr 2005;41(1):56-60. [18] Varni JW, Seid M, Rode CA. The PedsQL: measurement model for the Pediatric Quality of Life Inventory. Med Care 1999;37(2): 126-39. [19] Varni JW, Seid M, Kurtin PS. PedsQL 4.0: reliability and validity of the Pediatric Quality of Life Inventory version 4.0 generic core scales in healthy and patient populations. Med Care 2001;39(8):800-12. [20] Varni JW, Seid M, Knight TS, et al. The PedsQL 4.0 Generic Core Scales: sensitivity, responsiveness, and impact on clinical decisionmaking. J Behav Med 2002;25(2):175-93. [21] Varni JW, Burwinkle TM, Seid M, et al. The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr 2003;3(6):329-41. [22] Drossman DA. The functional gastrointestinal disorders and the Rome II process. Gut 1999;45(Suppl 2):II1-5. [23] Wilkins AJ, O'Callaghan MJ, Najman JM, et al. Early childhood factors influencing health-related quality of life in adolescents at 13 years. J Paediatr Child Health 2004;40(3):102-9. [24] Connolly MA, Johnson JA. Measuring quality of life in paediatric patients. Pharmacoeconomics 1999;16(6):605-25. [25] Marshall J, Hutson JM, Anticich N, et al. Antegrade continence enemas in the treatment of slow-transit constipation. J Pediatr Surg 2001;36(8):1227-30. [26] Williams J, Wake M, Hesketh K, et al. Health-related quality of life of overweight and obese children. Jama 2005;293(1):70-6. [27] Varni JW, Burwinkle TM. The PedsQL as a patient-reported outcome in children and adolescents with attention-deficit/hyperactivity disorder: a population-based study. Health Qual Life Outcomes 2006;4:26.
www.elsevier.com/locate/jpedsurg
Quality of life in children with slow transit constipation Melanie C.C. Clarke a , Chee S. Chow b , Janet W. Chase c , Susie Gibb d , John M. Hutson a,b,c , Bridget R. Southwell c,⁎ a
Department of Surgical Research, Royal Children's Hospital, Parkville, Melbourne, Victoria 3052, Australia Department of Paediatrics, University of Melbourne, Melbourne, Victoria, Australia c Murdoch Childrens Research Institute, Parkville, Melbourne, Victoria 3052, Australia d Department of General Paediatrics, Royal Children's Hospital, Parkville, Melbourne, Victoria 3052, Australia b
Received 10 September 2007; accepted 9 October 2007
Index words: Slow transit constipation (STC); Quality of life (QOL); Children; PedsQL
Abstract Background: Slow transit constipation (STC) causes intractable symptoms not readily responsive to laxatives, diet, or life-style changes. Children with STC have irregular bowel motions associated with colicky abdominal pain and frequent uncontrollable soiling. This study assessed the physical and psychosocial quality of life (QOL) in children with long-standing (≥2 years) STC vs healthy controls. Methods: Children (aged 8-18) were recruited from gastrointestinal and surgical clinics and a Scout Jamboree. After informed consent was obtained, the questionnaire (Pediatric Quality of Life Inventory) was administered. This consists of parallel child and parent self-report scales encompassing physical functioning, emotional functioning, social functioning, and school functioning. Higher scores indicate better QOL. P value less than .05 was considered statistically significant. Results: In 51 children with STC (mean, 11.5 years; male/female, 2:1) and 79 controls (mean, 12.1 years; male/female, 1.9:1), Pediatric Quality of Life Inventory QOL score was significantly lower in the STC group (72.90 vs 85.99; P b .0001). In addition, parents of children with STC reported a significantly lower QOL score than their child compared with the child's own report (64.43 vs 72.90; P = .0034). Parents of controls did not (84.25 vs 85.99; P = .12). Conclusions: Slow transit constipation is a debilitating condition affecting both physical and emotional functioning in children. Parental perception of QOL is significantly worse, highlighting the considerable family impact of constipation and uncontrollable soiling. © 2008 Elsevier Inc. All rights reserved.
Since its first description in 1996 [1], slow transit constipation (STC) is slowly becoming accepted as a chronic form of constipation in children [2-6]. It is characterized by intractable constipation that is not readily responsive to
Presented at the British Association of Paediatric Surgeons meeting, Edinburgh, Scotland, July 17-20, 2007. ⁎ Corresponding author. Tel.: +61 3 9345 5805; fax: +61 3 9345 7997. E-mail address: [email protected] (B.R. Southwell). 0022-3468/$ – see front matter © 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2007.10.020
laxatives, diet, or a change in life-style, and there is marked delay in colonic transit time on nuclear transit study [7,8]. The etiology of STC remains unknown with various theories involving neuronal abnormalities [9,10], genetic linkage [5,11], endocrine dysfunction [12], autonomic dysfunction [13], and abnormal colonic pacemaking [14] being postulated. Children with STC have irregular bowel motions, colicky abdominal pain, and frequent uncontrollable soiling. In some children, despite aggressive medical therapy, spontaneous passage of stools is unachievable. Consequently,
Quality of life in children with slow transit constipation Table 1
Demographics of study population
Male/Female Mean age (y) Mean duration of symptoms (y) Appendicostomy Soiling Abdominal pain
STC patients (n = 51)
Controls (n = 79)
2:1 11.5 10.1 14/51 41/51 40/51
1.9:1 12.1 N/A N/A N/A N/A
these children are managed by the surgical formation of an appendicostomy, through which regular antegrade bowel washouts can be performed to improve continence [15,16]. Definitive treatment remains elusive, and as a result, families of children with STC are offered a wide range of management options, many of which are perceived to be unsuccessful. There are many studies that have assessed the quality of life (QOL) in children with chronic disease; however, few studies exist that look at the physical and psychosocial impact of longstanding constipation in children [17]. This study used a validated pediatric questionnaire to assess physical and psychosocial features of life in children with STC.
1. Subjects and methods After the attainment of informed consent, the questionnaire (Pediatric Quality of Life Inventory [PedsQL]) was administered by 1 of 3 investigators (M.C., C.C., or J.C.). The PedsQL 4.0 Generic Core Scales consist of parallel child and parent self-report scales and have been validated in children and adolescents aged 2 to 18 years [18-21]. The questionnaires consist of 23 items encompassing (i) physical functioning (8 items); (ii) emotional functioning (5 items); (iii) social functioning (5 items); and (iv) school functioning (5 items). The categories can then be grouped into physical (i) and psychosocial (ii, iii, and iv) functioning. The questions ask how much of a problem each item has been during the past 1 month. A 5-point response scale is used (0 = never a problem; 1 = almost never a problem; 2 = sometimes a problem; 3 = often a problem; 4 = almost always a problem). Items are reverse-scored (0 = 100, 1 = 75, 2 = 50, 3 = 25, 4 = 0) and linearly transformed to a 0 to 100 scale. Higher scores indicate better QOL. Parent and child completed the questionnaire independently with impartial assistance being provided for any child who had difficulty with comprehension. The local Human Research Ethics Committee approved the study.
321 treated for constipation for a minimum of 2 years. Constipation was defined by the Rome II criteria [22], and all patients had previously had metabolic or hormonal causes excluded. All subjects were diagnosed with STC by radioisotope nuclear transit study [7]. Children with an organic cause for their constipation, cognitive impairment, or anorectal retention/normal transit on their nuclear transit study were excluded. Control subjects were recruited from (i) outpatient surgical clinics and consisted of children (8-18 years) who were attending for routine follow-up after uncomplicated appendicectomy or minor surgical procedure (ie, scrotal exploration, orchidopexy, herniotomy) and (ii) a scout jamboree that took place locally during the recruitment period. All control subjects had a “normal” bowel pattern (between 3 movements a week and 3 movements a day where the stool is brown or golden brown in color, formed, has a texture similar to peanut butter, and a size and shape similar to a sausage). Children who had concurrent comorbidities were excluded along with those who did not describe a normal bowel habit.
1.2. Outcomes The primary outcome measures were self- and parentreported, health-related QOL in children with STC compared with healthy controls.
1.3. Data Analysis Physical, psychosocial, and total QOL scores were compared using independent sample t tests (2-tailed). All P values less than .05 were considered as statistically significant.
2. Results From March 2006 to March 2007, 51 children (34 male, 17 female) with STC and 79 healthy controls (48 male, 31 female) were recruited into the study (Table 1). All children and their parents successfully completed the PedsQL. Child-reported QOL was significantly lower in children with STC compared with normal children (72.90 vs 85.99; P b .0001) (Fig. 1). Parent-reported QOL was significantly lower for children with STC compared with the control
1.1. Study population Study patients were recruited from the gastrointestinal and surgical clinics in a large tertiary pediatric hospital. Patients were between 8 and 18 years of age and had been
Fig. 1
Child-reported PedsQL scores: STC vs control group.
322
Fig. 2
M.C.C. Clarke et al.
Parent-reported PedsQL scores: STC vs control group.
group (64.43 vs 84.25; P b .0001) (Fig. 2). Physical and psychosocial QOL, both child- and adult-reported, were consistently poorer in the children with STC (Figs. 1 and 2). Parents of children with STC reported their child had a significantly lower QOL than the children themselves reported (64.43 vs 72.90; P = .0034) (Fig. 3), whereas parents of controls did not (84.25 vs 85.99; P = .12) (Fig. 4). Parents of children with STC and of controls reported significantly poorer psychosocial QOL for their child compared with the child's own report (Figs. 3 and 4). This was more apparent in the group of children with STC. There was no statistical difference in the reporting of physical QOL (Figs. 3 and 4).
3. Discussion Quality of life can be defined as “an individual's subjective perception of overall wellbeing and satisfaction with life” [23] or “physical, social and emotional aspects of a patient's well being that are important to the individual” [24]. QOL is a multidimensional concept that incorporates the assessment of recognized core domains and indicators. Many studies have looked at the QOL in children with chronic disease; however, there are few studies that have specifically examined the impact of chronic constipation on physical and psychosocial functioning [17]. There are no current publications that address QOL issues in children with proven STC. This is a population of children who have in many instances been managed for several years without a definitive diagnosis and often-ineffectual treatment and for whom social interaction is often limited because of antisocial symptoms (ie, soiling).
Fig. 3
STC group: child- vs parent-reported PedsQL scores.
Fig. 4
Control group: child- vs parent-reported PedsQL scores.
This study found that there appears to be a significant impairment in QOL of children with STC when compared with healthy controls. This is consistently described by the children and their parents and affects both physical and psychosocial QOL (Figs. 1 and 2). Parents of children with STC often describe the onset of their child's symptoms as being at, or shortly after, birth; STC represents as a lifelong problem. These children grow up with the daily problem of difficulty with stooling, chronic abdominal pain, and frequent soiling. Their social interactions are often limited because of the lack of insight of their young peers, and consequently, they can become withdrawn and socially isolated. Although children with STC have no obvious physical anomalies, both the children themselves and their parents report a significant deficit in their physical QOL. Questions included in the physical section of PedsQL ask about participation in sport, ability to take bath/shower by oneself, having hurts/aches, and energy levels. If children experience involuntary passage of stool, or soiling, then participation in sport can often seem daunting. Not only do children have to change in a communal area, where they may inadvertently expose protective clothing or their appendicostomy site [15,25], but they may also experience increased symptoms associated with the increase in physical exertion. Similarly, if children experience severe soiling, then they may require assistance to adequately clean themselves. As previously mentioned, children with STC are often affected by chronic abdominal discomfort. This chronic pain can account for the increased reporting of hurts/aches and also for the consistent reporting of low energy levels. In addition, children affected by the slow passage of stool often feel full and bloated and complain of a poor appetite. Their resultant decreased calorific intake will also contribute to their low energy levels. The psychosocial section of PedsQL is divided into emotional, social, and school functioning. Emotional functioning covers feeling afraid, sad, or angry, as well as troubles in sleeping and worrying about the future. Feeling strong emotions and worrying about the future are in many ways features of childhood in general and cannot always be attributed to chronic disease. Despite this, the subgroup of children with STC reported significantly poorer emotional QOL, and in particular, many were concerned about how their future might be affected by their condition.
Quality of life in children with slow transit constipation Table 2
323
Previous results for PedsQL questionnaire in different study populations
Population
Healthy children [19] Healthy children [21] Healthy children [27] Healthy children Not overweight [26] Overweight Obese Orthopedic (acute) [20] Cardiac disease [20] class Ia class IIb class III, IV ADHD [27] STC Cancer [27] Cerebral palsy [27]
Mean child reported score
Mean parent reported score
Total
Physical
Psychosocial
Total
Physical
Psychosocial
79.6 83.9 84.3 86.0 80.5 79.3 74.0 78.1
80.2 87.8 88.0 92.1 85.7 83.5 77.5 75.3
79.3 81.8 82.3 83.3 77.7 77.0 72.1 79.5
80.9 82.3 79.9 84.3 83.1 80.0 75.0 73.7
81.4 84.1 81.8 92.8 87.8 82.6 76.3 72.7
80.6 81.2 78.9 79.7 77.6 76.1 73.9 74.3
83.6 75.9 60.9 70.2 72.9 69.0 66.3
82.1 78.7 58.2 82.6 78.8 65.8 64.8
84.5 74.3 62.3 63.5 69.2 70.8 67.0
86.5 80.1 67.9 69.5 64.4 60.7 56.3
89.5 82.8 66.7 84.6 74.4 56.8 53.3
84.6 78.5 69.0 61.4 58.6 63.1 57.9
Data in bold represent the results of this study. ADHD indicates attention deficit hyperactivity disorder.
Social functioning inquires about the ability of the child to integrate with others. Questions focus on ability to get on with other children as well as social acceptability (other children wanting to be their friend/teasing) and ability to keep up with their peers. Children are often admired for their honesty and integrity; however, it is an undeniable fact that they can also be incredibly cruel especially when they lack insight. Children who are afflicted with chronic soiling are unfortunately easy targets for social ridicule and bullying. Although the responses of the children with STC suggest that they feel they have the ability to keep up with their peers, they have poor QOL scores for social functioning because of their reduced social integration. The final section of psychosocial QOL concerns school functioning. Subjects are asked about their ability to pay attention, memory, keeping up with schoolwork, and missing school either because of feeling unwell or because of the need to visit a medical professional. As previously discussed, some of these questions will score similar answers when asked to all children, especially those relating to paying attention and forgetting things. However, the children with STC again score significantly lower on this section because of poorer school attendance and frequent medical needs. This study also showed that although children with STC and their parents both describe a significantly poorer QOL than healthy children, the parents report a lower QOL in their children than their own children reported themselves (Figs. 3 and 4). There are several reasons why the parents may report a poorer QOL. Parents have an objective ability to look at their children compared with their peers and become distressed by any apparent differences. Because children with STC often have a family history of constipation, their parents may have had similar experiences at school, thus affecting their scoring. As previously mentioned, STC
usually represents a lifelong condition. Children who have STC grow up with their condition and the life-style that it entails and have no other experiences to which they can relate. Although they have the ability to appreciate that their life may not be ideal, it is all that they have ever known and so they make do with things as they are. Parents of children with STC often comment that their child has only one friend, and they do not actively socially interact with their peers. Although a parent may be terribly concerned by this, their child may simply be happy that at least they have a friend. There are several studies that have used the PedsQL to assess health-related QOL in different populations, such as overweight/obese children [26]; children with differing severity of cardiac disease [20]; children with an acute orthopedic injury [20]; children with cerebral palsy, cancer, or attention deficit hyperactivity disorder [27]; and healthy children [20,21,26,27]. These previous results are summarized in Table 2 along with the results for this study (in bold). These results show that QOL in children STC is comparable to children with other chronic illness, including cardiac disease and cancer. In conclusion, this study shows that QOL is significantly lower in children with STC compared with normal children, with both physical and psychosocial functioning scores reduced. Parental perception of QOL is worse when their child is affected by chronic constipation, particularly when estimating their child's psychosocial QOL.
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