Importance of Parental Conceptual Model of Illness in Severe Recurrent Abdominal Pain
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* Department of Paediatrics, Conway Institute for Biomolecular and Biomedical Research, University College, Dublin, Ireland
The Children’s Medical and Research Foundation, Our Lady’s Hospital for Sick Children, Crumlin, Dublin, Ireland
The Department of Liaison Child Psychiatry, Our Lady’s Hospital for Sick Children, Crumlin, Dublin, Ireland
|| Department of Pediatrics, Our Lady of Lourdes Hospital, Drogheda, County Louth, Ireland
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ABSTRACT |
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 TOP ABSTRACT METHODSRESULTSDISCUSSIONREFERENCES |
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Objectives. Recurrent abdominal pain (RAP) affects up to 15% of children. A biopsychosocial approach to the treatment of children with RAP has been proposed as an alternative to the traditional medical model. The aim of this study was to examine whether the parental conceptual model of illness is a factor in the long-term outcome of children with severe RAP.
Methods. The study was undertaken in 2 parts: 1) a review of the medical and psychiatric records (including Child Behavior Checklist [CBCL]) of all children with RAP of sufficient severity to necessitate hospitalization during a 5-year period and 2) a structured telephone interview to collect information on ongoing abdominal pain, other somatic symptoms, school attendance, and the parents’ opinion as to the cause of the child’s pain.
Results. Twenty-eight of 30 children who were identified were available for follow-up. Twenty-three (82%) were tertiary referrals from other pediatric services, and 20 had pain for >6 months. On admission 7 (25%) of 28 had a depressive disorder, and 8 (29%) had an anxiety/depressive disorder. Twenty-one of 28 parents completed the CBCL, and on analysis of the CBCL, 11 (52%) children had scores in the clinical range (>65). At follow-up (mean: 3.56 years; standard deviation: 1.59), 14 (50%) of 28 continued to complain of pain. These children also complained of multiple other somatic complaints and had repeated school absences. Only 1 (7%) of 14 parents of children with ongoing pain believed that there was a psychological cause for their child’s pain, whereas 11 (78%) of 14 parents of the children who had recovered believed that the cause was attributable to psychological factors (odds ratio: 47.67; 95% confidence interval: 3.56–1511.6).
Conclusions. The acceptance by parents of a biopsychosocial model of illness is important for the resolution of recurrent abdominal pain in children.
Key Words: recurrent abdominal pain • children • biopsychosocial model
Abbreviations: RAP, recurrent abdominal pain • IBS, irritable bowel syndrome • CBCL, Child Behavior Checklist • SD, standard deviation • OR, odds ratio • CI, confidence interval
Recurrent abdominal pain (RAP) is one of the most common problems in pediatrics, affecting 15% children.1–8 RAP was first described by Apley in the 1950s, when he defined the condition as at least 3 episodes of pain in the previous 3 months, severe enough to affect the child’s normal activities.1 Neither the natural history of RAP nor the factors that promote a resolution of pain are well understood. Although many children with RAP are treated successfully by their family doctor,9 RAP accounts for up to 25% of referrals to tertiary gastroenterology clinics,10 and severe refractory symptoms may, on occasion, require hospital admission. The outcome for children with severe RAP is uncertain, with only 1 small follow-up study suggesting that at least 50% of such children continue to have symptoms after discharge from the hospital.11
Compas et al12 proposed that RAP is a problem of exposure to psychological stress, individual differences in reactions to stress, and maladaptive attempts to cope with stress. It is suggested that the ways in which a child responds to stress is the critical factor in determining the frequency and duration of RAP.12,13 Engel first14 described a "biopsychosocial" model of illness in the 1970s. He proposed that illness is the product of biological, psychological, and social subsystems interacting at multiple levels.15 Irritable bowel syndrome (IBS) in adults is a functional gastrointestinal disorder affecting up to 20% of the population.16–18 A biopsychosocial model of medical care for the treatment of adults with IBS has been proposed by Drossman19,20 in preference to the traditional medical model. While understanding that both physiologic and psychological factors are important in IBS, Drossman suggested that acceptance by the patient of this conceptual model is critical to a successful outcome for the patient with IBS.19
Hyams and Hyman21 have also promoted a biopsychosocial model of medical care as being more appropriate for the treatment of children with RAP and their families rather than the traditional biomedical approach. However, parents often need to find a cause as well as a cure for their child’s pain. This need, to find a cause rather than a reason, may make it difficult to accept that psychological factors, in particular stress-related disorders, are the cause of the pain. We hypothesized that the parental conceptual model of illness is important in determining their child’s prognosis. Our aim in this study was to examine the outcome of a group of children with severe RAP and to assess whether there was a relationship between the parents’ conceptual model of illness and resolution of symptoms.
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METHODS |
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TOPABSTRACTMETHODSRESULTSDISCUSSIONREFERENCES |
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In our department, all children who have a diagnosis of RAP and require hospital admission are assessed and cared for jointly by the liaison child psychiatrist (M.D.) and the gastroenterology team. The biopsychosocial model of illness is introduced to the child and the parents at the outset by the gastroenterologist and the psychiatrist. Although recognizing that the pain is real and distressing, the psychosocial factors involved in the cause and persistence of pain are explored with the family.
This study was undertaken in 2 parts. In the first part, we identified all children who were admitted with a diagnosis of RAP to the gastroenterology unit at Our Lady’s Hospital for Sick Children and collected demographic, clinical, and psychiatric information from the medical records. In the second part of the study, we used a telephone interview to assess the current well-being of the children.
Part 1: Review of Clinical Information on Admission to the Hospital
Medical records of all children who were admitted with RAP during a 5-year period (1995–2000) were reviewed. Children with concomitant chronic illness such as cystic fibrosis were excluded from the study. RAP was defined as at least 3 episodes of paroxysmal abdominal pain occurring for at least 3 months and interfering with the child’s normal activities. The diagnosis of RAP was made by 1 of 2 pediatric gastroenterologists (B.B. or B.D.) after clinical history, examination, and review of the results of any medical investigations that were clinically indicated to exclude a diagnosis of organic disease. Demographic information, source of referral, specific symptoms and duration of symptoms, previous hospital admissions, investigations, medications, and school absence were abstracted from each child’s medical records.
A detailed semistructured psychiatric interview had been conducted with the child and the parents by the liaison child psychiatrist (M.D.) soon after admission. In addition, the Child Behavior Checklist (CBCL)22,23 was completed by most parents at the time of admission. The CBCL is a questionnaire consisting of 113 descriptive items that is completed by the parents. It documents the presence of somatic symptoms, symptoms of anxiety and depression, and the behavioral manifestations of aggressive and delinquent presentations. The CBCL categorizes the general personality or coping style of the child as either internalizing or externalizing. The internalizing behavior scale reflects fretful, inhibited, overcontrolled behavior, whereas the externalizing scale reflects aggressive, antisocial, undercontrolled behavior. Details of any psychiatric disorder in the child or family members, at the time of admission, were also recorded.
Part 2: Outcome for Children With Severe RAP
Parents of children identified in part 1 were contacted by mail and invited to participate in the follow-up study. Details of the study were explained in the letter. Parents who did not wish to participate in the study were asked to return a letter of refusal. A structured telephone questionnaire was used to collect information on ongoing abdominal pain, other somatic symptoms, medical service utilization, and school attendance. Another section addressed what the parents thought was the cause of the pain and whether they found a joint pediatric and psychiatric approach to the treatment of their child in the hospital helpful or otherwise. A single examiner (E.C.) conducted all telephone interviews. Ethical approval for the study was obtained from the Ethics Committee of Our Lady’s Hospital for Sick Children.
Statistics
Data were summarized using means and standard deviations (SD) and proportions as indicated. Differences between children who had recovered from RAP and those with ongoing pain are presented as odds ratios (ORs) with 95% confidence intervals (CIs). All tests were considered significant at P < .05.
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RESULTS |
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TOPABSTRACTMETHODSRESULTSDISCUSSIONREFERENCES |
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Part 1: Review of Clinical Information on Admission to the Hospital
Thirty children with RAP required hospital admission between January 1995 and December 2000. One family could not be contacted for follow-up, and 1 family refused to participate. Details of the 28 children included in the study are presented in Table 1. Twenty-three (82%) of the 28 children were tertiary referrals from other pediatric services (Table 1). The mean duration of pain at admission was 2 years (SD: 2.49 years; range: 3 months–12 years). Twenty (71%) children had pain for >6 months.
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Review of the clinical psychiatry assessment performed at the time of admission revealed that 7 (25%) of 28 children had a major depressive disorder. Four of these children were commenced on antidepressant medications, and the parents of 1 other child refused such treatment. Eight (29%) children had anxiety/depressive states. Ten (36%) children had other psychiatric diagnoses, including stress, denial, and somatization disorders. Only 3 children had no psychiatric disorder. Despite that 23 of these children were tertiary referrals, only 2 of the 28 had previously been assessed by a child psychiatrist.
Twenty-one (75%) of 28 children had a CBCL completed by their parents on admission to hospital. Analysis of the CBCL revealed that all 21 children had internalizing characteristics, with 11 having internalizing symptom scores in the clinical range (score >65). Coexisting externalizing characteristics were found in only 2 children. All of the 11 children who had CBCL scores in the clinical range also had a depressive or an anxiety/depressive disorder.
Part 2: Outcome for Children With Severe RAP
This part of the study was conducted in 2001, 1 to 6 years (mean: 3.56; SD: 1.59 years) after the initial admission to Our Lady’s Hospital for Sick Children. Interviews were conducted with 26 mothers and 2 fathers. Of the 28 children, 14 (50%) continued to have intermittent pains, with 4 of these still having pain several times a week. The mean duration of pain at the time of admission to the hospital was significantly longer for the group with ongoing pain than for the recovered group (3.2 years [SD: 3.0] vs 0.9 years [SD: 0.9]; P < .05). In addition, those with persistent pain tended to be younger (12.9 years [SD: 3.4] vs 14 years [SD: 2.3]; P < .05) at follow-up than the recovered group. The group with ongoing pain had a slightly longer time to follow-up (3.7 years [SD: 1.4] vs 3.4 years [SD: 1.5]), suggesting that recovery was not related to duration of follow-up. Children with continuing symptoms of RAP were much more likely to have multiple somatic symptoms at follow-up compared with those whose abdominal pain had resolved (Table 2). Furthermore, repeated school absence continued to be a problem in 64% of children with unresolved pain, whereas this was not a problem for those whose pain had resolved (Table 2).
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Although a follow-up psychiatric assessment was beyond the scope of this project, 10 (66%) of 15 of the children with a major psychiatric impairment such as depression or anxiety disorders at the time of initial hospital assessment had improved in terms of abdominal pain compared with only 4 (31%) of 13 of those with a somatization disorder or no psychiatric diagnosis (OR: 4.5; 95% CI: 0.7–31.4; Table 2). Furthermore, 12 children who had a parent with a history of psychiatric illness (depression; n = 10) were more likely to do well. Eight (66%) of these 12 children had recovered compared with 4 (25%) of 16 children from families who did not have such a diagnosis.
Six (54%) of 11 children who had internalizing scores in the clinical range on the CBCL had recovered from their pain, whereas 5 children continued to have pain. Two of the 3 children with no psychiatric impairment had pain at follow-up.
During the follow-up interview, parents were asked what they thought was the cause of their child’s illness. Thirteen of 14 parents of the group with ongoing pain cited a physical cause; only 1 of 14 cited psychological factors. In contrast, 11 of 14 parents of the children in the recovered group said that the pain was primarily attributable to psychological factors (OR: 47.7; 95% CI: 3.56–1511.6), and 8 of these parents said that identification of stressors was an important factor in their child’s recovery.
Parents were asked whether they had found the involvement of the pediatric gastroenterologist and the psychiatrist to be helpful in the treatment of their child. The parents of only 3 of 14 children in the group with ongoing pain stated that the involvement of the psychiatrist was helpful in the treatment of their child’s symptoms; 10 of 14 said that it had not been helpful, and 1 was uncertain whether it had been beneficial or not. In contrast, 10 of 14 of the parents of the recovered group said that the psychiatry consultation was helpful, whereas just 2 reported that it had not been helpful, and 2 were uncertain (OR: 10.8; 95% CI: 1.35–11.6). To control for bias in this reporting, parents were asked whether the gastroenterology input had been helpful in their child’s care or not; there was no difference between the groups, with 9 (64%) of 14 from each group stating that it had been helpful (Table 2).
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DISCUSSION |
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TOPABSTRACTMETHODSRESULTSDISCUSSIONREFERENCES |
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The aim of this study was to evaluate the long-term outcome of children with severe RAP requiring hospital admission, specifically in relation to the benefit of treatment based on a biopsychosocial model of illness with a joint care approach from the child psychiatry and pediatric gastroenterology services. We found that 50% of children continue to complain of abdominal pain >3 years (mean: 3.7 years; SD: 1.4) after discharge from the hospital and that parental acceptance of a biopsychosocial model of illness is strongly associated with recovery from RAP.
One of the most important requirements for counseling parents of children with RAP is information on the long-term outcome of this condition. Prospective studies on RAP are difficult to carry out because of the length of time required for follow-up, with most studies having a relatively short follow-up.24 Although our study is weakened by the retrospective manner in which patients were identified, that all of the children included had a full child psychiatric assessment at presentation and the majority (21 of 28) completed the CBCL considerably enhances the value of this study.
We believe that this is the first follow-up study of RAP based on a joint pediatric and child psychiatry approach to the child and the family. Hospitalization is rarely indicated for children with RAP,7,10 and the group of children in this study represents the severe end of the spectrum for the condition. This is further confirmed by the fact that 23 (82%) of 28 were referrals from other pediatric or surgical units. We specifically focused on those with severe RAP because we believed that it would be easier to identify differences between those who recovered and those who did not in such a group.
Despite a unified treatment approach by the gastroenterologists and the psychiatrist in promoting a biopsychosocial model of disease from the outset, the outcome was poor for 50% of children with severe RAP. There were marked differences between the parents of those children who recovered and those who did not in their perception of the cause of the pain and in their attitude toward the involvement of a child psychiatrist in the care of their child (Table 2). Parents of children with ongoing pain generally considered the cause to be physical in nature and stated that the psychiatrist’s input was not helpful. It could be suggested that because these children continued to complain of pain, their parents were unlikely to find their overall care satisfactory. However, 64% of parents of children with ongoing pain rated the input from the gastroenterologist as helpful. This suggests an unwillingness to consider a model of illness other than a medical model.
This study could not determine the parents’ conceptual model of illness at the time of admission to the hospital; therefore, the degree to which our approach to the treatment of these children was able to change their conceptual framework could not be assessed. Parents who reject the idea that psychological factors may be associated with the child’s pain will often continue to look for an organic cause for the pain, thus adding further to the burden of illness for the family as a whole, particularly the child and siblings. In an outpatient study, Claar et al25 found that before medical evaluation of children with abdominal pain, the majority of mothers believed that both physical and psychological factors were important causes of their child’s abdominal pain. However, despite medical evaluation and treatment, the mothers’ perception of the cause of the pain remained the same 1 year later. Additional prospective studies are needed to assess whether it is possible to change conceptual frameworks of illness and at what stage such interventions are likely to be successful.
We found that those children who still complain of pain were younger and had the pain for longer at the time of admission than the group that recovered. Apley26 has suggested that age <6 years and pain for >6 months are poor prognostic factors in children with RAP. It is unclear why younger children should be at increased risk for persistence of symptoms. The possibility of earlier intervention to identify the cause of stress for these children or the patterns of illness behavior, which develop into RAP, could diminish the burden of illness for these children and their families.
Psychiatric disorders have been previously associated with RAP, particularly anxiety and depression.27–30 Depression has been reported in 9% to 40% of children with RAP,28–30 whereas anxiety has been reported in 33% to 70%.27–30 It has also been suggested that persistent abdominal pain in childhood is a predictor of psychiatric disorders in adulthood.31 Our findings would suggest that >50% of children with severe RAP in this study have a significant psychiatric disorder, but of interest, 66% of this group had recovered from their abdominal pain at follow-up. The possibility of a referral bias because of psychiatric illness cannot be out ruled; however, a review of the referral letters suggests that the referring physicians were seeking a gastrointestinal rather than a psychiatric assessment.
Recurrent abdominal pain in childhood is more common in families with high rates of reported physical illness.1,31,32 We found that the mothers of children with persistent pain were significantly more likely to report gastrointestinal symptoms than mothers of children who recovered (P < .05; data not shown). Certain family responses are thought to perpetuate pain by inadvertent positive reinforcement.33,34 It is possible that the child may have "learned" to complain of pain or adopt the sick role model rather than to deal with stress.13
The absence of validated criteria to support a pediatrician in making a positive diagnosis of recurrent abdominal pain, without resorting to investigations to rule out organic causes, is a major problem in clinical practice. Pediatricians are understandably pushed toward defensive practice in evaluating these children, although they firmly believe the cause to be functional in nature. Although our study cannot provide a basis for pediatricians’ making a positive diagnosis of recurrent abdominal pain, it does suggest that the concept of a biopsychosocial basis for the pain should be introduced at a very early stage. We are presently undertaking a large study on the presenting features of children with organic and recurrent abdominal pain in an attempt to identify positive diagnostic criteria. The identification of such criteria would be significant, as it would allow pediatricians to reassure parents that the diagnosis of recurrent abdominal pain is based on evidence rather than clinical experience alone.
In summary, this study indicated that children whose RAP is diagnosed at a young age or families who reject a biopsychosocial model of illness are likely to have a poorer long-term outcome compared with families who accept the possibility of stress-related illness. We believe that the early introduction of a biopsychosocial model of illness to both the parents and the children with RAP could improve the outcome for many of these children.
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ACKNOWLEDGMENTS |
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We thank the Children’s Medical and Research Foundation, Our Lady’s Hospital for Sick Children, Crumlin, Dublin, which provided support for this work.
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FOOTNOTES |
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Received for publication Sep 6, 2002; Accepted Feb 19, 2003.
Reprint requests to (M.R.) Department of Paediatrics, Children’s Research Centre, Our Lady’s Hospital for Sick Children, Crumlin, Dublin 12, Ireland. E-mail: marion.rowland{at}ucd.le
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