Pain-Related Fear and Its Disabling Impact in Hypermobile Adolescents With Chronic Musculoskeletal Pain.

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Pain-related fear and its disabling impact in hypermobile adolescents with

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chronic musculoskeletal pain

3 1,2,

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Thijs van Meulenbroek, PT, MSc

Ivan P.J. Huijnen, PT, PhD

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Wiertz, MD 1, Jeanine A. Verbunt, MD, PhD 1,2

1,2,

Carlijn M.H.

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Journal of Orthopaedic & Sports Physical Therapy® Downloaded from www.jospt.org at R B Draughon Library on September 26, 2017. For personal use only. No other uses without permission. Copyright © ${year} Journal of Orthopaedic & Sports Physical Therapy®. All rights reserved.

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1

Maastricht University

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Research School CAPHRI

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Research program Functioning and Rehabilitation

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Department of Rehabilitation Medicine

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P.O. Box 616

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6200 MD Maastricht

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The Netherlands

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Adelante

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Centre of Expertise in Rehabilitation and Audiology

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P.O. Box 88

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6430 AB Hoensbroek

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The Netherlands

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This work was funded by Fonds Nuts Ohra, Stichting Vooruit, and Adelante. The

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authors have no financial disclosure and no conflicts of interest relevant to this work.

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Ethical approval for this protocol was granted by the Medical Ethics Committee of the

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Academic Hospital Maastricht/Maastricht University, the Netherlands,

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NL41712.068.12/METC 12-3-052.

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This study was not registered in a registry.


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Thijs van Meulenbroek, MSc

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Department of Rehabilitation Medicine

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Maastricht University

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P.O. Box 616

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6200 MD Maastricht

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The Netherlands

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Phone: 003143-3882168 (Tuesday)

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E-mail: [email protected]

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Study design: Cross-sectional study

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Background: Chronic musculoskeletal pain (CMP) has a negative impact on

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physical functioning. During adolescence joint hypermobility is a potential risk factor

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for developing CMP and pain-related fear contributes to the persistence of CMP.

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Whether pain-related fear and being hypermobile are related and even reinforce

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each other, resulting in a stronger association with perceived level of disability, is still

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unknown.

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Objectives: To evaluate whether pain-related fear has a stronger association with

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disability in hypermobile compared to non-hypermobile adolescents with CMP.

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Methods: The study included 116 adolescents with CMP. The presence of

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hypermobility was assessed using the Beighton scale. Measures of pain intensity,

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age, sex, and pain-related fear were collected and included in the multivariable

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model. A hierarchical regression analysis with disability as dependent variable and

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the interaction between hypermobility and pain-related fear was tested.

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Results: Hypermobile adolescents with CMP do not have more pain-related fear

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compared to non-hypermobile adolescents with CMP. There was no interacting effect

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between hypermobility and pain-related fear in explaining disability (β=0.20, P=0.42).

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Similarly, perceived harmfulness of balance-related activities were not more strongly

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associated with disability in hypermobile adolescents with CMP.

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Conclusion: The association of pain-related fear with the perceived level of disability

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is not more pronounced in hypermobile compared to non-hypermobile adolescents

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with CMP.

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Key words: Beighton, disability, joint hypermobility syndrome, perceived

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harmfulness

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INTRODUCTION

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Chronic pain is a common occurrence among adolescents. Up to 25% of all

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schoolchildren and adolescents, report chronic pain.29,30 Alongside abdominal pain

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and headache, chronic musculoskeletal pain (CMP) is one of the most frequently

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reported pain complaints in adolescents.10,29 In adolescents with CMP, pain

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complaints without a specific diagnosis often relate to muscular, ligamentous, bony,

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or joint structures and last for longer than 3 months.28 The incidence of CMP is

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reported to be higher among girls26,29 and increases with age during adolescence.29

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In approximately 40% of adolescents with CMP, pain has a considerable disabling

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impact.16,21,25 CMP has a negative impact on physical functioning, mood, social

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functioning, and family, reducing the adolescent’s quality of life.9,17,20,21,28 In

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adolescents with CMP, higher pain intensity has been shown to be associated with

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greater disability.14,41

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Although a broad array of medical diagnoses is involved, only 10-30% of pain

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complaints can be explained based on a specific somatic disease.19,23 Because there

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is not always a somatic solution to explain CMP, a biopsychosocial approach in the

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management of CMP should be considered.28 In adults, the fear-avoidance model

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has been proposed and shown to explain the disabling role of pain-related fear.22,45,50

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This model posits that catastrophizing thoughts may lead to the development of pain-

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related fear, thus leading to avoidance of feared activities. Over the long term, this

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avoidance behavior may result in disability, depression, and disuse, further fueling

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the vicious circle of disabling CMP.46-49 Recently, more evidence has become

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available confirming that this model can be applied to explain pain-related disability in

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the pediatric and adolescent population.2,34

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Recent evidence has also shown that hypermobility is a risk factor for developing

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CMP.39,43 Generalized joint hypermobility (GJH) is characterized by generalized joint

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laxity and increased joint mobility. In children with CMP the prevalence of GJH is 40-

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48%.15,33,42 This number far exceeds the prevalence of GJH in the general child

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population (19.2%).7 GJH associated with pain and symptoms such as arthralgia, soft

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tissue injury, and joint instability is referred to as joint hypermobility syndrome (JHS).

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Studies in children and adolescents showed that JHS is characterized by decreased

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muscle strength,11 impaired proprioception,11 disabling fatigue,52 reduced balance,32

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and problems with gait, falls, and coordination.1 In adults also an increased incidence

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of psychiatric symptoms, including anxiety disorders,4,12 panic disorders,13,37 and

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depression,5,37 were found to be associated with JHS.

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A study by Scheper et al31 has shown that young healthy adults with GJH tend to

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avoid sports and outdoor activities and that they prefer more stable activities like

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cycling. In view of their sense of impaired balance, this avoidance behavior is

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hypothesized to be a coping strategy to avoid joint-luxation or injury and thus prevent

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the development of musculoskeletal complaints. Therefore, it could be assumed that

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especially in hypermobile adolescents with CMP, pain-related fear and more

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specifically the perceived harmfulness of balance-related activities are more

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pronounced compared to non-hypermobile adolescents with CMP, possibly resulting

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in avoidance behavior leading to impaired daily functioning. Long term, impaired daily

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functioning may also result in a further decrease in muscle strength. For hypermobile

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adolescents with CMP, loss of muscle strength might act to further reduce joint

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stability, resulting in a higher risk of activities such as walking and problems such as

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falls and poor coordination causing the onset of pain. This might explain the higher

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prevalence of hypermobility in adolescents with CMP15,33,42 compared to the general

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adolescent population.7 Whether hypermobile adolescents with CMP have more

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pain-related fear compared to non-hypermobile adolescents with CMP and whether

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pain-related fear and being hypermobile reinforce each other resulting in a stronger

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association with perceived disability is still unknown.

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Therefore, the aim of this study was to evaluate whether pain-related fear has a

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stronger association with disability in hypermobile compared to non-hypermobile

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adolescents with CMP. In addition, we explored whether the perceived harmfulness

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of balance-related activities had a stronger association with disability in hypermobile

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compared to non-hypermobile adolescents with CMP.

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METHODS

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Participants

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Adolescents (aged 11-21 years) with CMP who were referred to the university

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outpatient rehabilitation clinic specialized in pain rehabilitation (Adelante/Maastricht

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University Medical Center+, the Netherlands) between June 2012 and December

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2015 were included in this cross-sectional study. Inclusion criteria were (a) non-

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specific musculoskeletal pain for longer than 3 months, (b) not diagnosed with a

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specific somatic disorder that could explain the current pain problem (such as

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neurological, orthopedic, or rheumatoid disorders), and (c) sufficient proficiency in the

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Dutch language to complete the questionnaires.

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The age spectrum of 11-21 years was used consistent with the definition of

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adolescents given by the American Academy of Pediatrics,3 which defines 3 stages:

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early adolescence, ranging from 11 to 14; middle adolescence, from 15 to 17; and

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late adolescence, from 18 to 21.

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Procedure

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Adolescents were referred to the university outpatient rehabilitation clinic for a

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consultation with a physician in rehabilitation medicine. All patients were asked to

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complete web-based questionnaires. Questionnaires were part of the regular health

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care procedure. Patients were asked whether their data could be used anonymously

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for research purposes. If they agreed, the data were included in the research

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database. Ethical approval for this protocol was granted by the Medical Ethics

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Committee

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Netherlands, NL41712.068.12/METC 12-3-052. The set of questionnaires retrieved

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information on sociodemographic variables (age, sex, education level, and school

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absences), duration of pain, pain intensity, pain catastrophizing, functional disability,

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and fear of pain. A physician in rehabilitation medicine or physiotherapist assessed

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hypermobility using the Beighton score.

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A subgroup of all participating adolescents received multidisciplinary rehabilitation

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treatment. In this group, the perceived harmfulness of activities was measured during

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the first session of the treatment program, at which time the patients had not yet

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received any educational instructions or multidisciplinary treatment.

of

the

Academic

Hospital

Maastricht/Maastricht

University,

the

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Measures

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Hypermobility

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The presence of hypermobility was assessed using the Beighton scale, which

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consists of 5 functional tests for which a maximum of 9 points can be obtained.

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These 5 tests are listed in TABLE 1. The standardized Beighton protocol is a valid

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instrument for the evaluation of generalized joint mobility in elementary school

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children.38 The full protocol for testing hypermobility is described by Smits-

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Engelsman et al.38 In the current study, this assessment was performed by a

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clinician. Based on the result of this assessment, the patients were assigned to 1 of 2

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groups: patients who had a Beighton score of ≥ 5 were assigned to the group with

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CMP and hypermobility (HM) and patients who had a Beighton score of < 5 were

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assigned to the group with CMP without hypermobility (NHM).18


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Sociodemographic variables

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To describe the study sample, the following variables were recorded: age, sex,

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education level, duration of pain, school absences during the last year, and pain

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intensity. Pain intensity was measured using a 100 mm visual analogue scale (Pain-

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VAS) with the endpoints being ‘no pain’ and ‘worst pain imaginable’. Pain intensity

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was measured for 3 references: (a) the pain at the moment the questionnaire was

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completed, (b) the worst/most severe pain experienced in the last week, and (c) the

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least pain experienced in the last week. The mean of these 3 Pain-VAS scores was

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calculated and used in further analyses. The Pain-VAS seems to be reliable and valid

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in children older than 8 years.24,40

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Functional disability

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Functional disability was measured using the Functional Disability Inventory (FDI).

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The FDI is a questionnaire for children and adolescents that measures the perceived

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difficulty of performing activities at home, at school, and in recreational and social

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domains. It contains 15 items that are rated on a 5-point scale (0 = ‘no trouble’ and 4

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= ‘impossible’). The total on the FDI can range from 0 to 60, with a higher score

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indicating greater pain-related disability. The reliability and validity of the FDI appears

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to be sufficient for assessing pain-related disabilities in children and adolescents.6,51

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Fear of pain

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The Fear of Pain Questionnaire – Child report (FOPQ-C) was used to measure pain-

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related fear in children and adolescents with chronic pain. It contains 24 items and 2

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scales: fear of pain and avoidance of activities. Items are scored on a 5-point scale (0

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= ‘strongly disagree’ to 4= ‘strongly agree’). Total scores can range from 0 to 96.

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Higher scores indicate higher levels of pain-related fear and avoidance. The FOPQ-C

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appears to be a valid and reliable questionnaire.36

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Perceived harmfulness

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The Photograph Series of Daily Activities for youth (PHODA-youth) was used to

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measure perceived harmfulness, or the indication to what extent the patient

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perceives the activity to be harmful for the painful body part. The PHODA-youth

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contains photographs of 51 age-specific daily life activities and social situations

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which are scored on the level of perceived harmfulness of performing these activities.

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The PHODA-youth consists of 3 categories: daily life and household activities

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(PHODA-ADL), intensive physical activities (PHODA-PA), and social activities

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(PHODA-SA). Adolescents rate the photographs from 0 (‘completely harmless’) to 10

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(‘extremely harmful’). Subscale scores for the 3 categories were obtained by

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summing up the scores for all items in that subscale. To obtain a total score, all items

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were summed up. The electronic version of PHODA-youth has been found to be valid

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and reliable.44

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For the current study, a subscale of the perceived harmfulness of balance-related

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activities (PHODA-balance) was developed. Two authors, a physiotherapist (TvM)

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and a physiatrist (JV), each independently marked all 51 age-specific daily life

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activities and social situations ýes´ or ńo´ depending on their relation to balance. If

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both authors marked an activity ýes´ the activity was added to the PHODA-balance.

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In instances of disagreement, a third author (physiotherapist, IH) made the final

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decision whether to include the activity in the PHODA-balance. Seven activities were

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selected as activities related to balance, consisting of 6 items from the PHODA-PA

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and 1 from the PHODA-ADL. To obtain a subscale score on the PHODA-balance,

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scores for the 7 items were summed up as on the traditional subscales. The selected

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activities were: 5. Putting pants on, 20. Participating in physical exercise/gym, 28.

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Dancing/sports, 30. Running, 32. Kicking a ball, 35. Jumping over something, 36.

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Balancing on one leg.

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Pain catastrophizing

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The Pain Catastrophizing Scale for Children (PCS-C) measures catastrophizing

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thoughts about pain. It contains 13 items describing different thoughts and feelings

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that children and adolescents may experience when they are in pain, which are rated

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on a 5-point scale (ranging from 0 = ‘not at all’ to 4 = ‘extremely’). The total score on

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the PCS-C can range from 0 to 52, with a higher score indicating more

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catastrophizing thoughts about pain. The PCS-C has been reported to be reliable

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and valid for children and adolescents.8

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Statistical analysis

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The results were tested for normality using histograms and the Kolmogorov-Smirnov

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goodness-of-fit test. Descriptive data for the HM and NHM group were calculated.

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Where distribution of the data was normal, continuous data were shown as mean

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values (standard deviations [SD]), and where the distribution was non-normal the

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data were shown as medians (interquartile ranges [IQR]); categorical data were

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presented as absolute frequencies or percentages. Differences between the HM and

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NHM group were analyzed using independent sample t-tests, the Mann-Whitney U

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test, the Kruskall-Wallis test, and Chi-square test.

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To test the hypothesis whether pain-related fear has a stronger association with

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disability in adolescents in the HM group compared to the NHM group, a hierarchical

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(blockwise entry) regression analysis was performed. Variables were entered in the

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regression model in 3 blocks. With FDI as the dependent variable in the model, Pain-

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VAS, age, and sex (female 0; male 1) were introduced as independent variables in

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the first step to control for demographic variables and pain intensity. In the second

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step, pain-related fear (FOPQ) and the group variable of hypermobility (HM/NHM;

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dichotomous variable) were entered simultaneously. Finally, the interaction term

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(FOPQ x HM/NHM) was introduced in the third step. In case of a significant

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interaction, additional regression analyses were performed for HM and NHM

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separately to identify influencing factors for disability.

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First the Pearson correlation between FOPQ and PHODA-balance was calculated.

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The regression analysis was then repeated with PHODA-balance instead of FOPQ in

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the model to explore whether the perceived harmfulness of balance-related activities

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has a stronger association with disability in hypermobile compared to non-

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hypermobile adolescents with CMP. For all regression analyses, a collinearity check

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was performed. Collinearity was considered a problem if the variance inflation factor

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(VIF) was above 10.27 Alpha was set at 0.05. All statistical analyses were performed

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in IBM SPSS Statistics for Windows, version 23.0 (IBM Corp, Armonk, NY, U.S.A).

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RESULTS

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Descriptive analyses

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A total of 183 adolescents (162 female/21 male) were asked if their data could be

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used anonymously for research purposes, data from 116 adolescents were used in

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the current study. Sixty-seven adolescents were excluded from the analyses for the

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following reasons. Eight ultimately proved to have a specific somatic disorder

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explaining the current pain problem, 38 did not complete the questionnaires, and the

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Beighton scores of 21 potential participants were unknown. Of the 116 adolescents,

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only 45 received multidisciplinary treatment and for this reason the PHODA-youth

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was only measured in this subgroup (FIGURE).

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The characteristics of each group are presented in TABLE 2. No significant

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differences were found between the HM group and the NHM group with respect to

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sociodemographic variables such as age, sex, education level, and school absences.

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The duration of pain, Pain-VAS, PCS-C, FDI, FOPQ, and all PHODA outcomes also

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did not significantly differ between groups.

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The association between pain-related fear, hypermobility and disability

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TABLE 3 presents the outcome of the regression analysis with disability as the

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dependent variable. The inclusion of Pain-VAS, age, and sex as independent

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variables in the first step showed that no significant association with disability was

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found. In the second step FOPQ and the group variable HM/NHM were added to the

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model, resulting in a significant model (R2=0.32, Adj. R2=0.29, P

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