The psychosocial implication of childhood constipation on the children and family: A scoping review protocol.

Introduction

Constipation is a defecation disorder that is an increasingly significant and prevalent chronic health problem in children (Walter et al., 2019). Despite this, it is often poorly recognised and inadequately managed (Borowitz et al., 2005; Widodo et al., 2018). Barnes et al.’s (2018) survey on paediatric gastroenterologists and general paediatricians reported a lack of awareness of ROME IV criteria for diagnosis of FC in children. For diagnosis purposes, constipation is categorised as primary or secondary based on its pathophysiology. Primary constipation, also known as functional constipation (FC), has no underlying organic causes and occurs in 95% of children diagnosed with constipation (Koppen et al., 2016; Tabbers et al., 2014). In contrast, secondary constipation has organic pathology such as Hirschsprung’s disease, anorectal malformations, hypothyroidism, diabetes mellitus, hypercalcaemia and hypokalaemia (Rajindrajith & Devanarayana, 2011). Diagnosis of functional constipation in children should be guided by the internationally recognised guideline ROME IV criteria, which includes two or more of the following symptoms occurring at least once per week for a minimum period of 1 month: two or less defecations per week, at least one episode of faecal incontinence per week, a history of excessive volitional stool retention or posturing, history of painful or hard bowel movements, presence of large faecal mass in the rectum and a history of large diameter stools that can obstruct the toilet (Hyams et al., 2016). The diagnosis of primary or FC must exclude secondary constipation, as ‘red flag’ symptoms may point to a more serious underlying disorder requiring further investigations (NICE, 2018). Misdiagnosis and ineffective management of constipation in children can result in chronicity that can last up to ten years (Michaud et al., 2009) and even into adulthood (Bongers et al., 2010; Rajindrajith et al., 2013).

Global epidemiological data on childhood constipation to date is limited. The available data on the prevalence of childhood constipation demonstrates a considerable variation between geographical regions ranging from 0.7% to 32% (Chogle et al., 2016; Játiva-Mariño et al., 2019; Oswari et al., 2018; Udoh et al., 2017; van Tilburg et al., 2015; Wu et al., 2011). The global pooled prevalence is reported at 9.5% and a much higher prevalence of 17.5% in children below four years of age (Koppen et al., 2018). In paediatric gastroenterology clinics, the reported prevalence ranges from 25% to 45% (Benninga et al., 2004; Di Lorenzo, 2000; Loening-Baucke, 2007). This variation may be attributed to a heterogeneity in definitions of constipation, duration of symptoms, methodology of studies, age distribution of children and epidemiological data (Rajindrajith & Devanarayana, 2011). Data on prevalence by age distribution is also limited due to variation in study age groups. Koppen et al.’s (2018) systematic review and meta-analysis found no association between prevalence of constipation in children and age. However, Rajindrajith et al.’s (2012) survey on children and adolescents aged between 10 to 16 years found a negative correlation between prevalence of constipation and age; demonstrating the highest prevalence (24%) in children aged 10 years. In addition, Walter et al.’s (2019) cross-sectional study on children aged between 6.5 and 48 months demonstrated that the highest prevalence (13%) were in children aged between 37 and 48 months. To date, there have been no epidemiological studies to identify the prevalence of childhood constipation in Ireland.

While there is no known underlying cause, there are multiple factors that can lead to the development of constipation in children. Some environmental factors have been linked to a higher risk of this disorder in children for example: a sedentary lifestyle and poor diet, low socioeconomic background, poor maternal education, and cramped living accommodations (Edan & Yahya, 2022; Kilincaslan et al., 2014; Ludvigsson et al., 2006) as well as low levels of interaction with parents (Yamada et al., 2019). Early childhood developmental factors have also been associated with constipation in young children, such as incidents of painful defecation at a young age leading to withholding behaviour (Kilincaslan et al., 2014; Mugie et al., 2011); authoritarian parenting styles in younger age group (Kilincaslan et al., 2014; van Dijk et al., 2015); stressful childhood incidents such as psychological trauma; physical, verbal and sexual abuse; being bullied in school and inadequate or punishment during toilet training (Edan & Yahya, 2022; Inan et al., 2007; Philips et al., 2015; Rajindrajith et al., 2016; Rajindrajith et al., 2021; Thomaz de Almeida et al., 2021; Walter et al., 2019) as well as alcoholism and severe illness within the family (Oswari et al., 2018). Joinson et al.’s (2019) prospective study reported a significant association between psychosocial problems and constipation. This large study involving 8435 children aged between 4 and 10 years found that psychosocial issues at a young age such as behavioural and emotional problems, temper tantrums and sleep problems were linked to higher levels of constipation and soiling. In addition, stressful incidents at ages between 2 ½ and 4 years were associated with higher levels of constipation and soiling. While the limitation reported in this study was the use of parental report of constipation and soiling, the use of latent classes extracted from a large cohort increases generalisability of findings. Confounders in this study such as sex, socio-demographic measures and child’s developmental level were adjusted to increase validity of findings.

Childhood constipation has significant adverse consequences affecting multiple aspects of the life of children. Several studies have reported reduced average weight and height in children with chronic constipation when compared to children without the disorder (Chang et al., 2013; Pawłowska et al., 2018; Yousefi et al., 2019). However, this report is controversial, as other studies have reported on the correlation between chronic constipation and overweight and obesity (Koppen et al., 2016; Olaru et al., 2016; Santos-Andreoli et al., 2019). A recent systematic review and meta-analysis which evaluated eighteen studies involving 33,410 children reported that childhood constipation is associated with the prevalence of overweight and obesity (Lazarus et al., 2022). Children with constipation also demonstrated higher somatic scores when compared to healthy children (Appak et al., 2017; Kilincaslan et al., 2014; Rajindrajith et al., 2013). Rajindrajith et al.’s (2013) cross-sectional survey using Pediatric Quality of Life Inventory and Child Somatization Index on 1792 children aged 13 to 18 reported higher somatisation scores with symptoms including headache, dizziness, lethargy, nausea and vomiting, as well as pain in the abdomen, lower back, chest and limbs/joints when compared to healthy children. These symptoms were found to have a significant effect on the children’s health-related quality of life (HRQOL). The large sample size and use of previously validated tools increases the validity and generalisability of this study. The reported limitation to this study was the diagnostic methodology which did not include a physical examination. Also, a generic HRQOL questionnaire was employed which may lack sensitivity and accuracy. Another common problem associated with chronic constipation in children is bladder symptoms including urinary tract infections (Halder et al., 2021; Hoque et al., 2010; Sarvari et al., 2017), urinary incontinence during the day time (van Engelenburg–van Lonkhuyzen et al., 2017) and lower urinary tract dysfunction which includes symptoms such as increased or decrased urinary frequency and bladder overactivity (Burgers et al., 2013; Queiroz Machado et al., 2015; Muhammad et al., 2015; Sampaio et al., 2016)

High somatic scores was also reported in a prospective, comparative survey by Appak et al. (2017) on children with constipation between 4 and 18 years of age (n=32) and ‘healthy controls admitted to a paediatric gastroenterology outpatient clinic’ (n=31)(pg. 466). The study employed the following child and family assessment tools: Strengths and Difficulties Questionnaire (SDQ), Beck Depression Inventory (BDI), State and Trait Anxiety Inventory and Parental Attitude Research Instrument (PARI). High levels of somatic complaints including recurrent abdominal pain, frequency of micturition and faecal soiling were reported in this study. The study also reported significantly higher scores of anxiety and depression in children and their mothers, compared to healthy children and their mothers. This study has a few limitations which were not reported including small sample sizes and non-randomization of sampling which increases the risk of bias.

The impact of constipation on the psychological and social wellbeing, and overall HRQOL of children and adolescents is reported in numerous studies (Appak et al., 2017; Kaugars et al., 2010; Kilincaslan et al., 2014; Rajindrajith et al., 2013; Rajindrajith et al., 2016; Rajindrajith et al., 2020; Ranasinghe et al., 2017; Wang et al., 2013). Wang et al.’s (2013) cross-sectional, case-control study on children aged 3 to 6 years of age (n=211) using a Chinese version of the Pediatric Quality of Life Inventory^TM, version 4.0 (PEDQL^TM4.0) and Elkhayat et al.’s (2016) cross-sectional, case-control study on children aged 4 to 12 years of age (n=100) using an Arabic versions of the Child Behavior Checklist for 4-18 years old (CBCL/4-18) and the Pediatric Quality of Life Inventory^TM, version 4.0 (PEDQL^TM4.0) found that scores for quality of life of children were lower compared to those of children without constipation. Both studies employed non-experimental observational methods with two groups: children diagnosed with constipation and healthy control group. Both studies also demonstrated that children with constipation had significantly higher anxiety levels, sleep disturbances and social problems. The reported limitation to both these studies was the use of a generic HRQOL questionnaire which lacks sensitivity and accuracy, and the small sample size in Elkhayat et al.’s (2016) study. Both studies also reported the probability of lower scores with parent perception of child’s HRQOL. Neither study identified nor discussed adjustment of potential confounders. Sleep disturbance in children with constipation is also reported in Yildirim et al.’s (2017) quantitative study into health-related quality of life in school aged children. Kilincaslan et al.’s (2014) survey on children aged between 2 and 6 years (n=124) reported that children in the constipation group (n=65) demonstrated higher levels of psychological issues such as anxiety, depression, aggression, externalising and internalising compared to children with normal intestinal habits (n=59). This is supported by Rajindrajith et al.’s (2020) cross-sectional survey which found that adolescents with constipation demonstrated increased risk of significant emotional, behavioural or internalising issues such as withdrawal, unresponsiveness, anxiety and depression in comparison to healthy adolescents.

Childhood constipation also has adverse psychosocial impact on the school environment of children and adolescents. Kaugars et al.’s (2010) qualitative study on children with chronic constipation (n=8) and their caregivers (n=8) found that 62.5% of children in the study reported that school performance was affected by constipation and faecal incontinence. The children in this study also expressed concerns of using toilets in the school and the negative effect of constipation and faecal incontinence at school. This is supported by Rajindrajith et al.’s (2016) survey on adolescents aged between 13 and 18 years, (n=1807) using four validated instruments: ROME III diagnostic questionnaire for the paediatric functional GI disorders, child maltreatment questionnaire, Child Somatization Inventory and Pediatric Quality of Life Inventory (PedsQL). This study found that faecal incontinence associated with chronic constipation is associated with maltreatment of all forms including sexual, emotional and physical abuse. However, the study is unable to determine the direction of this relationship, if maltreatment is the result or cause of faecal incontinence. The study also found that faecal incontinence increased the likelihood of emotional abuse in school by other children and even teachers due to the faecal odour. This study emphasized the significance of the psychological impact of constipation and faecal incontinence on children and adolescents in terms of poor interaction, social phobia and academic performance. A major limitation highlighted in this study was the lack of physical examination to formally diagnose constipation. In addition, authors in this study failed to identify or discuss adjustment of potential confounders. However, the association of abuse and bullying with childhood constipation is also reported in Liyanarachchi et al.’s (2022) systematic review. The review included 15 articles and 2954 children with constipation reported that stressful life events including parental divorce or separation, severe illness in family and parental job loss, as well as stressful school events such as bullying, moved school, separation with best friend and examination failure were all associated with constipation in children. These findings are also supported by a more recent systematic review with similar findings (Gozali et al., 2023).

Constipation with faecal incontinence has a greater adverse effect on the quality of life of the child when compared to childhood constipation without faecal incontinence (Kovacic et al., 2015). This prospective study employed five validated instruments: PedsQL – Parent Report, PedsQL – Family Impact Module (FIM), Functional Disability Inventory (FDI) – Parent Version, Pediatric Inventory for Parents (PIP), and Pediatric Symptom checklist (PSC) – Parent Report. The study included families of children aged between 2 to 18 (n=410) found that quality of life scores worsened as the children’s ages increased. Consequently, adolescents with constipation and faecal incontinence demonstrated the lowest scores. A limitation of this study was the use of parent-report of child’s HRQOL which may result in lower scores and which may be influenced by parental confounders such as their psycho-emotional status, level of stress or other medical conditions. Another limitation identified was the generic HRQOL questionnaire which may lack sensitivity and accuracy. There is also a financial impact associated with childhood constipation. Constipation with faecal incontinence has a significant impact on families in terms of increased cost and time spent on laundry secondary to soiling and faecal incontinence (Kaugars et al., 2010; Thompson et al., 2021a). There is also a significant cost impact associated with the utilisation of health care services such as emergency department (Choung et al., 2011; MacGeorge et al., 2018), behavioural therapy and prescriptions (Kaugars et al., 2010; Liem et al., 2009; Thompson et al., 2021a). Choung et al.’s (2011) nested case-control study on 5,718 children, adolescents and young adults aged between 5 to 21 years of age reported that the mean medical costs for children with constipation were four times more than children without constipation (p=0.055) and anxiety in children with constipation resulted in a 348% increase in inpatient cost (Choung et al., 2011). Childhood constipation also has an indirect cost impact through the days lost at work for the parents (Liem et al., 2009).

Recurrent constipation as a result of treatment failures has a significant effect on the quality of life of the child’s parents and family (Dolgun et al., 2013; Elkhayat et al., 2016; Klages et al., 2017; Thompson et al., 2021b; Wang et al., 2013), resulting in conflict within family members (Kaugars et al., 2010). Parents and families of children with constipation have significantly lower quality of life scores in several aspects including physical, emotional, social, daily activity and relationship (Wang et al., 2013). This is supported by Klages et al.’s (2017) study on caregivers of children aged between 2 and 18 using the Parental Opinion of Pediatric FC (POOPC) (Silverman et al., 2015) which found that caregivers of children with constipation reported high levels of disease burden, distress and psychosocial problems. The study also found that caregivers were dissatisfied with management by healthcare providers which resulted in poor adherence to prescribed treatment. This supports findings from other studies including reports of parental anxiety, frustration, and mistrust of healthcare providers (Kovacic et al., 2015) and Thompson et al.'s (2021b) qualitative study on parental experiences of caring for children with constipation which reported that parents expressed frustration and dissatisfaction with the care received in terms of dismissive and insensitive attitude of care providers, as well as inadequate care, support and information provided.

A review of current literature has highlighted that children with chronic constipation have high levels of behavioural problems such as withdrawal, anxiety, depression, aggression, externalising and internalising. Childhood constipation also has an adverse effect on the quality of life of children and their families, affecting their physical, emotional, social and school life, as well as daily activity and relationship. Parents have expressed anxiety and frustration, as well as dissatisfaction and distrust of healthcare professionals. To date, there has been no studies done to identify the psychosocial implication of childhood constipation on children and their families in Ireland.

The burden of chronic illness is one of the key focuses in the recently published Sláintecare Reports in Ireland, highlighting the need to reform the model of care of children and chronic conditions (Houses of the Oireachtas, 2017). Therefore, addressing and identifying the impact of childhood constipation on children and their family is crucial to current needs and can be aligned with Sláintecare’s vision to improve child health and wellbeing services (Houses of the Oireachtas, 2017). In addition, a recent study has highlighted constipation in children and young people as one of the highest-ranking priorities of research across the UK and Ireland (Cathie et al., 2022).

A preliminary search of databases demonstrated an increasing interest in childhood constipation in recent years. The search was conducted through Cochrane Library on Systematic Reviews, Health Research Board (HRB) Open Research and Joanna Briggs Institute (JBI) Evidence Synthesis, as well as UCD Library Onesearch and Google Scholar where several reviews on constipation were identified. There were 6 reviews registered in PROSPERO: 30 systematic reviews and 2 scoping reviews in Google Scholar and one in HRB Open Research. The majority of all the reviews were focused on pharmacological and non-pharmacological treatments. There were three reviews identified with similar elements: Belsey et al.’s (2010) systematic review is focused on impact on quality of life in adults and children with constipation; Vriesman et al.’s (2019) systematic review is focused solely on the quality of life of children; Thompson et al.’s (2021a) systematic review is focused on experiences and information needs of parents. There are no recent reviews focused on the psychosocial implications of childhood constipation on children or the families of children with constipation. Childhood constipation is a complex health problem that has an adverse effect on multi-dimensional aspects of their life. Therefore, there is a need to identify the extent of these effects on the lives of children. In addition, the identification of how childhood constipation affects the wellbeing of the families is also crucial as the well-being of the child is dependent on family well-being (Newland, 2015).

Methods

This scoping review will be guided by the six stages of Arksey and O’Malley Framework (Arksey & O’Malley, 2005) and the updated and refined version by Peters et al. (2022). In order to ensure rigour in reporting, the review will be carried out and reported using the Preferred Reporting Items for Systematic reviews and Meta-Analyses extension for Scoping Review (PRISMA-ScR) (Tricco et al., 2018). The six stages of the review are described as follows.

Study status

At the time of publication, this study is at the stage of preliminary searches of databases.

Discussion

There is a growing recognition that childhood constipation is an increasing global health problem which results in significant burden on the child and their family, health services, society and economy. Studies to date have identified that childhood constipation has a significant implication on health-related quality of life on the children and their families, affecting multiple domains of children and their families’ lives including physical, emotional, school and psychosocial, as well as daily activity and relationship.

To date, no studies have identified the psychosocial implication of childhood constipation on children and their families in Ireland. Therefore, there is a need for a systematic scoping review to identify and explore all available evidence regarding the psychosocial implication of childhood constipation on the children and their families, as well as to identify gaps in the literature. Findings from the review will be used to direct the focus of subsequent research into childhood constipation in Ireland.

This protocol has been developed to guide a scoping review into the impact of childhood constipation on children and their families. Scoping reviews are inclined to be iterative especially in the search strategy, data extraction and data presentation stages, which can often result in deviance from the protocol (Peters et al., 2022). This is acceptable and recommended to promote the development of a thorough, systematic and comprehensive review. To ensure transparency and rigour, all amendments will be clearly documented and described.

Ethics statement

Ethical approval will not be required as the scoping review outlined in this protocol will review publicly available literature.