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Research ArticleOriginal ResearchA

Prognosis of Abdominal Pain in Children in Primary Care—A Prospective Cohort Study

Yvonne Lisman-van Leeuwen, Leo A. A. Spee, Marc A. Benninga, Sita M. A. Bierma-Zeinstra and Marjolein Y. Berger
The Annals of Family Medicine May 2013, 11 (3) 238-244; DOI: https://doi.org/10.1370/afm.1490
Yvonne Lisman-van Leeuwen
1Department of General Practice, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands
2Department of General Practice, Erasmus MC, Rotterdam, the Netherlands
PhD
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  • For correspondence: Y.Lisman-van.Leeuwen@umcg.nl
Leo A. A. Spee
2Department of General Practice, Erasmus MC, Rotterdam, the Netherlands
MD
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Marc A. Benninga
3Department of Paediatric Gastroenterology, Emma Children’s hospital-Academic Medical Center, Amsterdam, the Netherlands
MD, PhD
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Sita M. A. Bierma-Zeinstra
2Department of General Practice, Erasmus MC, Rotterdam, the Netherlands
PhD
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Marjolein Y. Berger
1Department of General Practice, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands
2Department of General Practice, Erasmus MC, Rotterdam, the Netherlands
MD, PhD
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  • Author response: Pediatric Chronic Abdominal Pain. The Trip On The Iceberg
    Yvonne Lisman-van Leeuwen
    Published on: 26 August 2013
  • Pediatric Chronic Abdominal Pain. The Trip On The Iceberg
    Miguel Saps, MD
    Published on: 08 August 2013
  • Author response: 'Commentary for Lisman-van Leeuwen et al article on Prognosis of Abdominal Pain in Children in Primary Care--A Prospective Cohort Study'
    Yvonne Lisman-van Leeuwen
    Published on: 12 July 2013
  • Commentary for Lisman-van Leeuwen et al article on Prognosis of Abdominal Pain in Children in Primary Care--A Prospective Cohort Study
    Natasha A Koloski
    Published on: 28 June 2013
  • Published on: (26 August 2013)
    Page navigation anchor for Author response: Pediatric Chronic Abdominal Pain. The Trip On The Iceberg
    Author response: Pediatric Chronic Abdominal Pain. The Trip On The Iceberg
    • Yvonne Lisman-van Leeuwen, Researcher
    • Other Contributors:

    Saps and Chogl emphasize the importance of conducting studies investigating FGIDS at the primary care level as most patients receive care at this level. We share their thought and we are happy they appreciate our effort of conducting a study on abdominal pain at the primary care level. They state that information on treatment strategies and whether the use of medications affected the prognosis would have been an importa...

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    Saps and Chogl emphasize the importance of conducting studies investigating FGIDS at the primary care level as most patients receive care at this level. We share their thought and we are happy they appreciate our effort of conducting a study on abdominal pain at the primary care level. They state that information on treatment strategies and whether the use of medications affected the prognosis would have been an important addition to the study. The addition of information on treatment strategy would indeed have been a valuable addition to our manuscript. Unfortunately, we have limited data about the GP's treatment strategy. We do know that treatment in these children consisted of advice on eating habits or life style factors, losing weight, medications or keeping a diary on abdominal pain. In approximately one third of patients the GP had a wait-and-see approach.(1) Treatment strategies however, are heterogeneous and therefore prohibit proper comparison.

    Recently, the Dutch College of General Practitioners developed and published the guideline "Abdominal pain in children".(2) This guideline differentiates acute abdominal pain (? 1 week) from chronic abdominal pain (> 1 week) and gives advice on diagnostic and treatment strategy. In children with chronic abdominal pain without indications for a somatic cause the diagnostic strategy is recommended to be limited to urinary analysis. The emphasis should be on treatment. The guideline committee concludes that most children with chronic abdominal pain can be treated by their GP. The management should consist of explanation and advice to the patient and his parents, with the aim of providing reassurance. Taking up normal activities and going to school must be promoted. At least 1 follow- up consultation is advised to check the effect of treatment and answer possible questions. If the abdominal pain persists, a pediatrician can be consulted. Because of a lack of scientific evidence, most treatment recommendations in this guideline are based on expert opinions. We therefore fully agree with Saps and Chogl that further studies evaluating candidate interventions and predictors of their effects are urgently needed.

    References

    1. L.A.A. Spee, Y. Lisman-van Leeuwen, M.A. Benninga, S.M.A. Bierma-Zeinstra, M.Y. Berger. Prevalence, characteristics and management of childhood functional abdominal pain in general practice. In press Scand J Prim Health Care

    2. W. Eizinga, M.J. Gieteling, M.Y. Berger, R.M. Geijer . [Summary of the NHG guideline 'Abdominal pain in children', the 100th NHG guideline]. Ned Tijdschr Geneeskd 2013; 157(15): A6191

    Competing interests: None declared

    Show Less
    Competing Interests: None declared.
  • Published on: (8 August 2013)
    Page navigation anchor for Pediatric Chronic Abdominal Pain. The Trip On The Iceberg
    Pediatric Chronic Abdominal Pain. The Trip On The Iceberg
    • Miguel Saps, MD, Associate Professor in Pediatrics-Gastroenterology, Hepatology and Nutrition
    • Other Contributors:

    Pain predominant functional gastrointestinal disorders (FGIDs) encompass a group of disorders characterized by the presence of chronic or recurrent abdominal pain that cannot be explained by biochemical, anatomical, or structural abnormalities. FGIDs including functional abdominal, dyspepsia and irritable bowel are among the most common conditions in children.1 FGIDs are currently defined according to the third version o...

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    Pain predominant functional gastrointestinal disorders (FGIDs) encompass a group of disorders characterized by the presence of chronic or recurrent abdominal pain that cannot be explained by biochemical, anatomical, or structural abnormalities. FGIDs including functional abdominal, dyspepsia and irritable bowel are among the most common conditions in children.1 FGIDs are currently defined according to the third version of the Rome criteria.2 In children, chronic pain can greatly interfere with daily functioning.3,4 Children experiencing chronic abdominal pain frequently miss school, withdraw from social activities, and are at risk of developing internalizing symptoms.3,5 These factors make pediatric chronic pain a focus of clinical and research interest. Better understanding of the epidemiology of FGIDs their natural course and prognosis may help establish successful treatment strategies and optimize the allocation of public health resources. One of the problems frequently encountered by clinicians and public health planners is the paucity and limited quality of pediatric data. Only a small minority of children with abdominal pain seeks medical attention and those receiving health care services are frequently seen at the primary level.5 Studies have also shown an increasing trend for children with chronic pain symptoms to receive care at the primary level.6 Still, majority of the studies in FGIDs are conducted at academic and tertiary care level institutions that do not represent the level of care that most children receive. Studies have shown that children cared for at the tertiary level undergo more frequent and more costly testing than children seen at the primary care.7 All the aforementioned factors underscore the importance of conducting investigations on the subset of children with FGIDs that receive primary care services. Lisman-van Leeuwen et al. conducted a study on a cohort of 305 children seen at the primary care level for one year. The effort of the authors to conduct a multicenter prospective study should be praised. The primary care based design, the use of standardized questionnaires and low attrition rate are important aspects of this study. The authors evaluated possible selective bias to enhance the external validity of the results. Children in the study were assessed at baseline, 3, 6, 9 and 12 months after initial visit. Repeated evaluation allowed establishing the course and prognosis of this group of disorders.

    The results of this study provide important and rather humbling prognostic information on CAP in children. Forty-six percent of children were diagnosed with CAP at the first visit and 37.1% of children had CAP after one year of medical care and follow-up. Almost 80% of children fulfilled criteria of CAP during the study period. Most of the children reported persistence of abdominal pain at the end of one year and only half of children reported that abdominal pain no longer had an impact in their life after one year (defined in the study by school absenteeism, interference with play, use of medications or moderate to severe pain). Children who were first diagnosed with CAP during follow-up and who were likely to have earlier care than those that already had CAP at recruitment did not have a better prognosis (mean duration CAP diagnosis at follow-up, 8.3 months vs. 7.5 months duration in children with CAP diagnosis at baseline). The results of this study are in agreement with another Dutch study on children undergoing hypnotherapy.8 In this clinical trial, only 25% of children that underwent standard medical therapy (control group) that included multiple medical consultations with pediatric gastroenterologist, medications, fiber and dietary advice were in clinical remission at one year. Together, both studies demonstrate that commonly used conventional therapies provide modest relief and even less of a resolution to the child's problem. Moreover, the broad prognosis of children with chronic abdominal pain in both studies does not overall differ from the worrisome prognosis reported in studies published more than 50 years ago in children with recurrent abdominal pain.9 Since then, the old term of recurrent abdominal pain has been substituted by more modern and updated terminology, multiple iterations of the Rome criteria have been published and new therapies and approaches to management have been proposed. Still, some practitioners continue not to use the Rome criteria and the prognosis of children with chronic abdominal pain continues to be less than optimal.10 The prognosis of children not participating in studies and therefore not being repeatedly contacted by the research team to assess their progress may be even worse as repeated contact may provide some level of reassurance that somebody cares about their problem.

    The high prevalence and impact of CAP in children underscores the needs for an in depth evaluation and revision of currently used treatment strategies.4,5 A previous study found that family physicians commonly prescribe medications for nonspecific abdominal pain despite the lack of evidence for its effectiveness.6 Less than 30% of the cohort of children in Lisman-van Leeuwen et al study received medications at any point of time. The authors do not provide information on how many different children received medications, type of treatments or success of each strategy. The use of the Rome criteria for diagnosis and the inclusion of both, information on treatment strategies and whether the use of medications affected the prognosis would have been an important addition to this study. It is possible that a common approach to treatment in children with different pathophysiological mechanisms to their symptoms, different risk factors, comorbidities, developmental stages and psychological makeup is responsible for the poor prognosis and the disappointing results found in some clinical pharmacological trials.11 Interestingly, the study found that children diagnosed with irritable bowel syndrome and those 10-17 years of age were a particularly vulnerable population. Children in this age group had the longest duration of abdominal pain (10.5 months), the highest cumulative incidence (67.9%) and the highest prevalence impact at one year (61%). The investigation of the risk factors for poor treatment response is key to achieve the goat of instituting tailored treatments for different groups of children.

    In summary, Lisman-van Leeuwen et al. conducted a multisite prospective cohort study using standardized methods and a large sample size that provides relevant information on the prognosis of children with CAP who receive medical attention at the primary care level. Although issues with abdominal pain are frequently portrayed as the tip of the iceberg due to the low rate of formal medical assessment and care received by children, Lisman-van Leeuwen et al. study underscores another important aspect of this problem by showing that even children who receive care at the doctor's office often have a poor prognosis.12 The disappointing prognosis in children who seek medical attention demonstrates that the problem transcends beyond accessibility to medical services and questions the current approach to care. Clinical trials and prospective studies using Rome criteria definition of functional gastrointestinal disorders to assess the results of the different interventions and investigate predictors of treatment outcome may allow identifying the factors responsible for the repeated trips with the same iceberg that result in so many treatment failures.

    References:
    1. Chitkara DK, Rawat DJ, Talley NJ. The epidemiology of childhood recurrent abdominal pain in Western countries: a systematic review. The American journal of gastroenterology. Aug 2005;100(8):1868-1875.
    2. Rasquin A, Di Lorenzo C, Forbes D, et al. Childhood functional gastrointestinal disorders: child/adolescent. Gastroenterology. Apr 2006;130(5):1527-1537.
    3. Palermo TM. Impact of recurrent and chronic pain on child and family daily functioning: a critical review of the literature. Journal of developmental and behavioral pediatrics : JDBP. Feb 2000;21(1):58-69.
    4. Palermo TM, Chambers CT. Parent and family factors in pediatric chronic pain and disability: an integrative approach. Pain. Dec 15 2005;119(1-3):1-4.
    5. Saps M, Seshadri R, Sztainberg M, Schaffer G, Marshall BM, Di Lorenzo C. A prospective school-based study of abdominal pain and other common somatic complaints in children. The Journal of pediatrics. Mar 2009;154(3):322-326.
    6. Gieteling MJ, Lisman-van Leeuwen Y, van der Wouden JC, Schellevis FG, Berger MY. Childhood nonspecific abdominal pain in family practice: incidence, associated factors, and management. Annals of family medicine. Jul-Aug 2011;9(4):337-343.
    7. Lane MM, Weidler EM, Czyzewski DI, Shulman RJ. Pain symptoms and stooling patterns do not drive diagnostic costs for children with functional abdominal pain and irritable bowel syndrome in primary or tertiary care. Pediatrics. Mar 2009;123(3):758-764.
    8. Vlieger AM, Menko-Frankenhuis C, Wolfkamp SC, Tromp E, Benninga MA. Hypnotherapy for children with functional abdominal pain or irritable bowel syndrome: a randomized controlled trial. Gastroenterology. Nov 2007;133(5):1430-1436.
    9. Christensen MF, Mortensen O. Long-term prognosis in children with recurrent abdominal pain. Archives of disease in childhood. Feb 1975;50(2):110-114.
    10. Gieteling MJ, Bierma-Zeinstra SM, Passchier J, Berger MY. Prognosis of chronic or recurrent abdominal pain in children. Journal of pediatric gastroenterology and nutrition. Sep 2008;47(3):316-326.
    11. Kaminski A, Kamper A, Thaler K, Chapman A, Gartlehner G. Antidepressants for the treatment of abdominal pain-related functional gastrointestinal disorders in children and adolescents. The Cochrane database of systematic reviews. 2011(7):CD008013.
    12. Nurko S. The tip of the iceberg: the prevalence of functional gastrointestinal diseases in children. The Journal of pediatrics. Mar 2009;154(3):313-315.

    Competing interests: None declared

    Show Less
    Competing Interests: None declared.
  • Published on: (12 July 2013)
    Page navigation anchor for Author response: 'Commentary for Lisman-van Leeuwen et al article on Prognosis of Abdominal Pain in Children in Primary Care--A Prospective Cohort Study'
    Author response: 'Commentary for Lisman-van Leeuwen et al article on Prognosis of Abdominal Pain in Children in Primary Care--A Prospective Cohort Study'
    • Yvonne Lisman-van Leeuwen, Researcher
    • Other Contributors:

    We are happy to read that Koloski and Talley appreciate our follow-up study of children with abdominal pain presenting in primary care. Koloski and Talley explain the results of our study using the biopsychosocial model and suggest different etiologies in younger and older children. We agree that in some children the abdominal pain could be induced by stressful life events such as starting school and that in other childre...

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    We are happy to read that Koloski and Talley appreciate our follow-up study of children with abdominal pain presenting in primary care. Koloski and Talley explain the results of our study using the biopsychosocial model and suggest different etiologies in younger and older children. We agree that in some children the abdominal pain could be induced by stressful life events such as starting school and that in other children chronic? psychological disorders might be more prominent as a cause. Therefore, our finding that in younger children chronic abdominal pain is more often recurrent, whereas in older children it is more often chronic might suggest different etiology in these age groups, which is a very interesting thought, which needs further evaluation.

    We would like to share a different an alternative explanation. We were interested in the prognosis of functional abdominal pain. Despite its multifactorial origin, one could argue that functional abdominal pain is one 'pathologic' entity. From that perspective, the recurrent abdominal pain in young children might well be the precursor of chronic abdominal pain in older children. Therefore, directing psychological interventions towards the younger children (and their parents) might be more effective than such interventions in the older ones. We appreciate the thought that parents become enmeshed in a negative cycle of reacting to pain by allowing undesirable behavior. This mechanism might also underlie a prolonged course in younger children, which suggests young children and their parents are suitable candidates for psychological (or educational) interventions. Further study is required to evaluate this hypothesis.

    Although we did not present the data in our article, we did collect psychological data using the Child Behavior Checklist (CBCL) parent report. The CBCL provides a global measure of psychopathological symptoms. We analysed the presence of depressive and anxiety problems, but also looked at the presence of other functional symptoms [1]. We can confirm that the prevalence of psychological problems is high in children with functional abdominal pain. At presentation to the general practitioner, the prevalence of depressive and anxiety problems was around 25% and 15%, respectively. More striking was the high number of children (60%) with multiple non-specific somatic symptoms. During the 12 months follow-up the depressive, anxiety or somatic symptoms persisted in about one third of the children. Although the prevalence of these symptoms decreased, children with abdominal pain still had more psychological and non-specific somatic symptoms after 12 months follow-up than children in the general population.

    We therefore strongly support the advice of Koloski and Talley that the general practitioner should take age into account and always consider other concurrent somatic but also psychological diagnoses in their management plan for pediatric abdominal pain. We especially would like to point out the possible presence of multiple non-specific somatic symptoms and recommend the general practitioner to include these in his history taking.

    Reference 1. Gieteling MJ, Lisman-van Leeuwen Y, Passchier J, et al. The course of mental health problems in children presenting with abdominal pain in general practice. Scand J Prim Health Care 2012; 30(2): 114-20.

    Competing interests: None declared

    Show Less
    Competing Interests: None declared.
  • Published on: (28 June 2013)
    Page navigation anchor for Commentary for Lisman-van Leeuwen et al article on Prognosis of Abdominal Pain in Children in Primary Care--A Prospective Cohort Study
    Commentary for Lisman-van Leeuwen et al article on Prognosis of Abdominal Pain in Children in Primary Care--A Prospective Cohort Study
    • Natasha A Koloski, Researcher
    • Other Contributors:

    Unexplained abdominal pain is common in children [1]. The article by Lisman-van Leeuwen et al fills an important gap in the literature as it describes the natural history of abdominal pain in children in general practice. Previous studies have shown that complaints of abdominal pain affect up to 44% of children in general practice [2] but this study is the first prospective investigation to demonstrate that almost half of...

    Show More

    Unexplained abdominal pain is common in children [1]. The article by Lisman-van Leeuwen et al fills an important gap in the literature as it describes the natural history of abdominal pain in children in general practice. Previous studies have shown that complaints of abdominal pain affect up to 44% of children in general practice [2] but this study is the first prospective investigation to demonstrate that almost half of children with abdominal pain will go on to meet the criteria for chronic abdominal pain (CAP) over a 12 month period. In particular, they showed that CAP was more likely to be recurrent in younger children with a peak in 4 to 5 year olds, and more chronic and severe in older children aged 10 to 17 years. While a negative impact on activities was the main reason for presenting to general practice, it is concerning that 50% of children and up to 62% of older children still reported a moderate to severe impact of abdominal pain after 12 months. Although no psychological data were collected, others have shown that the majority of children with abdominal pain have anxiety or depression [3].

    It has been suggested that CAP is best conceptualised within a biopsychosocial model whereby interactions between psychosocial distress and altered gut physiology (sensory and motor) result in functional abdominal pain via the brain-gut axis [4]. This model provides a useful framework for understanding the findings from the current study. Younger children may experience abdominal pain during stressful life events such as starting school, and this could explain the peak in prevalence of abdominal pain seen among the 4 to 5 year olds. Thus it is important for the general practitioner to ask about current stresses and provide reassurance to parents that this abdominal pain is likely be a reaction to stress rather than organic disease. On the other hand older, older children with chronic abdominal pain or symptoms of IBS may need more formalised and earlier psychological intervention. It is possible that psychological disorders are compounded by illness behaviour in older children whereby their parents become emeshed in a negative cycle of reacting to pain by allowing the missing school and repeatedly seeking health care [5] thereby reinforcing the cycle. This study highlights the need for the general practitioner to consider age and concurrent diagnoses in their management plan for paediatric abdominal pain.

    References

    1. Chitkara DK, Rawat DJ, Talley NJ. The epidemiology of childhood recurrent abdominal pain in Western countries: a systematic review. Am J Gastroenterol. 2005;100:1868-1875.

    2. Huang RC, Palmer LJ, Forbes DA. Prevalence and pattern of childhood abdominal pain in an Australian general practice. Paediatr. Child Health 2000;36:349-353.

    3. Campo JV, Bridge J, Ehmann M et al. Recurrent Abdominal Pain, Anxiety, and Depression in Primary Care. Pediatrics 2004;113:817- 824.

    4. Drossman DA. The functional gastrointestinal disorders and the Rome II process. Gut 1999;45(suppl 2):II1-5. Robinson JO. Alverez JH. Dodge JA. Life events and family history in children with recurrent abdominal pain. Journal of Psychosomatic Research 1990;34:171-81.

    Competing interests:   None declared

    Show Less
    Competing Interests: None declared.
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Prognosis of Abdominal Pain in Children in Primary Care—A Prospective Cohort Study
Yvonne Lisman-van Leeuwen, Leo A. A. Spee, Marc A. Benninga, Sita M. A. Bierma-Zeinstra, Marjolein Y. Berger
The Annals of Family Medicine May 2013, 11 (3) 238-244; DOI: 10.1370/afm.1490

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Prognosis of Abdominal Pain in Children in Primary Care—A Prospective Cohort Study
Yvonne Lisman-van Leeuwen, Leo A. A. Spee, Marc A. Benninga, Sita M. A. Bierma-Zeinstra, Marjolein Y. Berger
The Annals of Family Medicine May 2013, 11 (3) 238-244; DOI: 10.1370/afm.1490
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  • Exploring the concept of pain of Australian children with and without pain: qualitative study
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  • The Surprising Chronicity of Abdominal Pain in Children
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