A two-week inpatient programme with a booster improved long-term management of severe chronic paediatric pain

Abstract

In the context of limited healthcare resources and increasing demands for more cost-effective healthcare solutions, this study assessed the short- and long-term clinical outcomes and resource utilization of a two-week inpatient, interdisciplinary, pain treatment (IIPT) including individual and group cognitive behavioural, occupational, physical and recreational therapy, education and family intervention and a booster in the chronic paediatric pain setting. Using a longitudinal design with a two-year follow-up, two-week IIPT resulted in sustainable improvements in mean and maximum pain intensity, physical functioning and internalization and reductions in the mean number of medical visits, school absence and frequency of pain medication at year 2 following IIPT. While pain-related disability scores did not improve, problem-focused coping became more prevalent, and patient and parent-assessed satisfaction as well as pain experience continued to improve throughout the study. Our results demonstrate that a two-week IIPT with a booster confers meaningful short- and long-term improvements in clinical outcomes and resource utilization among paediatric patients with severe chronic pain.

Keywords

Booster children chronic pain cognitive behavioural therapy inpatient pain management

Background

Chronic paediatric pain defined as persistent (lasting more than three months) or recurrent pain is a multi-causal disease with a distinct pathomechanism (Wojtowicz and Banez, 2015). It frequently manifests itself in headache, abdominal and musculoskeletal pain. Severe chronic pain affects up to 5% of the school-aged population, mostly girls, and has a significant adverse impact on the child (e.g. depression, anxiety, daily functional impairment), parents (e.g. psychological distress, financial hardship) and society (absenteeism, high utilization of healthcare resources) (Hechler et al., 2014; Huguet and Miró, 2008; King et al., 2011; Sleed et al., 2005; Zernikow et al., 2012). The management of severe chronic pain requires an interdisciplinary approach, which addresses

underlying pain pathomechanisms, treats specific symptoms and teaches coping skills to the child and family to restore and improve daily functioning.

Inpatient, interdisciplinary, pain treatment (IIPT) including cognitive behavioural therapy has been advocated as an effective treatment modality for children with severe chronic pain and functional disabilities; however, its widespread adoption into clinical practice is hindered by high costs (lengthy treatments, large treatment teams) and limited access (Hechler et al., 2015; Sieberg et al., 2011).

In Hungary, healthcare resources are limited and expensive; therefore, shortening IIPTs to enhance their efficiency, cost-effectiveness and affordability is a clinical necessity, which may result in improved access to treatment. Despite a growing number of studies with IIPTs in the management of chronic pain, several important research questions remain to be explored including its long-term efficacy and impact on school functioning such as social relationships and learning.

The objectives of this study were to (1) generate research hypotheses and (2) assess the short- and long-term efficacy of a two-week, brief and less resource-intensive IIPT and a booster session to improve relevant clinical outcomes among children and adolescents suffering from severe chronic pain. We hypothesized that our IIPT resulted in persistent and clinically relevant benefits at year 2 following intervention.

Methods

Design

The study was a prospective longitudinal study with self-assessed questionnaires at admission (baseline), month 3 and year 2 for adolescents living with severe chronic pain. The study was approved by the local Ethics Committee of the Bethesda Children’s Hospital. Written informed consent was obtained from patients and their parents.

Patients

Patients attending the outpatient psychological and psychiatric clinic of the HRC Bethesda Children’s Hospital were recruited into the study. Inclusion criteria were based on those developed by the German Paediatric Pain Centre (GPPC) for similar chronic pain programmes (Hechler et al., 2009) and included participants between 10 and 19 years of age, from treatment-resistant chronic pain defined as pain lasting for at least six months, with an average pain intensity ≥5 of 10 in the preceding week and continuous pain or pain peaks ≥8 of 10 at least twice a week. Exclusion criteria included previous organic work-up and disclosure of malignant diseases. Patients with severe psychiatric morbidity, or any other psychiatric condition, which could adversely impact group sessions, have also been excluded. Exclusion criteria included unavailability of parents or patients to attend the three therapy sessions or the two-week inpatient treatment. During the recruitment period of four months, 15 patients have been screened for the study.

Of the 15 patients screened, 5 patients were excluded due to unavailability to attend the two-week programme and one patient due to significant psychiatric morbidity. Of the nine patients enrolled in the study, eight completed the whole two-week programme. One patient was lost to the study (reason unknown). Data from eight patients were analysed for the treatment phase. At the month 3 and year 2 follow-up visits, five and six patients provided data for assessments, respectively. The demographic and disease characteristics of the participants are presented in Table 1.

Table 1.

Demographic and disease characteristics of participants.

Male	1
Female	7
Mean age (SD)	14.75 (2.49)
Living in the capital	4 (50%)
Mean duration of pain in years (SD)	1.73 (2.16)
Site of primary pain
Head	3 (37.5%)
Limb	1 (12.5%)
Abdomen	2 (25%)
Thorax	1 (12.5%)
Eye	1 (12.5%)
Multiple pain site	5 (62.5%)
Persistent pain	4 (50%)
Sleep disturbance	1 (12.5%)

Note: SD: standard deviation.

Interdisciplinary treatment team

The study team consisted of two paediatric psychiatrists, a clinical psychologist/family therapist, a psychologist, a physiotherapist, an occupational therapist, a social worker, a paediatric anaes-thesiologist, paediatric nurses and a paediatrician/family therapist.

The intervention: IIPT

The IIPT was based on systemic therapeutic and operant-cognitive behavioural principles, our cognitive and psychotherapy programmes and other previously published intensive pain management programmes including that of the GPPC (de Blécourt et al., 2008; Eccleston et al., 2003; Hechler et al., 2009, 2010; Lanzi et al., 2007; Maynard et al., 2010; Palermo and Scher, 2001; Sherry et al., 1999). The two-week programme consisted of two to four group psychotherapy sessions (45 minutes each) daily for nine days (a total of 19 hours). In addition, we conducted three family therapeutic sessions, three single psychotherapy sessions, two educational sessions (about the bio-psycho-social model of chronic pain, useful lifestyle changes, time management, setting realistic goals, etc.) and three leisure activities over the two weeks.

A unique feature of our programme was a booster session with the family three months after admission to reinforce the main principles of our programme and newly learnt techniques.

Outcome measures

Outcome measures were assessed by patients and their parents using self-administered questionnaires and open questions developed specifically for this research. Outcome measures and questionnaires were selected based upon our experience with test batteries at our Psychosomatic Department, the practice at the GPPC and the recommendations by the Paediatric Initiative on Methods, Measurement and Pain Assessment in Clinical Trials (McGrath et al., 2008).

Pain intensity

Patients assessed the average and maximal pain intensity they experienced over the previous week by an 11-point visual analogue scale at baseline, month 3 and year 2 following the initial treatment (von Baeyer et al., 2009).

Pain-related disability

The Hungarian version of the Paediatric Pain Disability Inventory was used to measure pain-related disability in daily activities on 12 items (e.g. family life, school attendance, physical activity, etc.) using a 5-point Likert-type scale (1 denoting never and 5 denoting always) at baseline, months 3 and year 2 following intervention. Higher scores indicate higher disability (Hübner et al., 2009).

Depression

Patients completed the Hungarian version of the Children Depression Inventory at baseline and at year 2. Total scores of <13, 13–20 and >20 are considered normal, at risk and clinical depression, respectively (Kovacs, 1992).

Maladaptive behaviour

To assess maladaptive behaviours and emotional states of patients and to rule out any psychopathology before treatment, patients were asked to complete the Hungarian version of the Child Behaviour Checklist Questionnaire at baseline and year 2. The questionnaire has six subscales (social problems, anxiety/depression, somatic problems, attention problems, deviant and aggressive behaviour), which are further categorized into two subscales of internalization (emotional) and externalization (behavioural) problems (Achenbach, 1991). Higher scores indicate more maladaptive behaviour.

Coping

To assess problem and emotion-focused coping, patients were asked to complete the Ways of Coping Inventory at baseline and year 2. Higher scores on the scales indicate higher preference for that coping style (Folkman and Lazarus, 1980).

Subjective pain experience after treatment

Patients were asked to rate how frequently they experienced pain following treatment at month 3 and year 2 on a 5-point Likert-type scale (0 = much less frequently, 4 = much more frequently).

Satisfaction with treatment

Patients and their parents were asked to rate their satisfaction with treatment on an 11-point Likert-type scale (0 = not at all, 10 = very much) at month 3 and year 2. Children were asked to indicate the level of satisfaction that they felt at month 3 and year 2 following the treatment (McGrath et al., 2008).

Use of pain medication and healthcare utilization

Parents were asked to record the frequency of pain medications and medical visits over the preceding two months, respectively. These measures served as surrogates of healthcare burden (including costs) and medication use (McGrath et al., 2008).

Physical functioning. (it is a heading originally)

Parents assessed their children’s physical functioning by recording the number of days with physical impairment in the previous week (impairment due to heavy, moderate and light work).

School absence. (it is a heading originally)

Parents assessed the number of days their children missed at school in the previous month, a surrogate measure of impaired social interactions and learning.

Data analyses and statistical methods

Due to the small sample size, no formal statistical analyses have been performed. Descriptive statistics (mean, standard deviation) were used to assess outcome variables. To enable comparison between our study results and those by Hechler et al. (2015), we also computed Cohen d effect size for the outcome measures in our study at baseline, short- and long-term follow-up (Hechler et al., 2015). Effect sizes above 0.2, 0.5 and 0.8 are considered small, medium and large, according to Cohen (1988). For the data analysis, SPSS software version 21.0 was used (IBM SPSS Statistics; IBM Corporation, Chicago, IL, USA).

Results

Our results on the therapeutic effects of IIPT demonstrated a reduction in pain intensity, school absence, medication as well as healthcare utilization and improvements in daily physical functioning and internalization.

Patients reported severe mean and maximum pain intensity at baseline over the previous week. Mean and maximum pain intensity progressively decreased at month 3 and year 2 following treatment. Pain-related disability scores were similar at baseline and at year 2; however, a slight increase was seen at month 3 following treatment. Depression scores indicated an at-risk population at baseline and did not worsen notably during the follow-up period. At baseline, children had no maladaptive behaviours and emotions; however, an elevated internalization score suggested a higher prevalence of emotional problems. At year 2 following treatment, internalization scores returned to normal levels (Table 2).

Table 2.

Patient-assessed outcome measures of a two-week inpatient, interdisciplinary pain treatment among children and adolescents suffering from chronic pain.^a

Measures	Pre-treatment/baseline (N = 8)	Three months following treatment (N = 5)	Two years following treatment (N = 6)
Pain intensity (last seven days)	4.19 (2.10)	3.60 (2.58)	3.25 (3.68)
Maximum pain intensity (last seven days)	5.69 (1.33)	4.4 (2.95)	3.25 (3.46)
Pain-related disability	24.50 (8.77)	30.80 (7.46)	25.67 (7.39)
Depression	13.00 (5.58)	–	15.00 (7.29)
Maladaptive behaviour
Internalization	8.5 (2.51)	–	5.83 (3.13)
Externalization	5.00 (3.00)	–	5.00 (3.10)
Coping
Problem-focused coping	12.50 (2.78)	–	15.17 (5.60)
Emotion-focused coping	10.38 (5.90)	–	10.67 (2.73)
Pain experience after treatment	–	1.00 (0.71)	0.17 (0.41)
Treatment satisfaction by patients	–	5.2 (2.84)	6.91 (1.49)

^aValues are given as mean (standard deviation).

While emotion-based coping styles did not change much after the intervention, problem-based coping styles became more prevalent at year 2 following treatment. Subjective pain experience improved between month 3 and year 2 after the treatment (Table 2).

Healthcare burden was assessed by calculating the mean number of medical visits in the last two months before each scheduled visit. As shown in Table 3, the mean number of medical visits continued to decrease until the end of the study.

Table 3.

Parent-assessed outcome measures of a two-week inpatient, interdisciplinary pain treatment among children and adolescents suffering from chronic pain.^a

Measures	Pre-treatment/baseline (N = 8)	Three months following treatment (N = 5)	Two years following treatment (N = 6)
Number of medical visits in the last two months	2.72 (2.50)	0.8 (1.10)	0.25 (0.61)
Impaired days in the last seven days
Due to hard physical activity	1.69 (2.50)	0.88 (1.18)	1.5 (2.07)
Due to moderate physical activity	1.00 (2.45)	0.00 (0.00)	0.42 (0.67)
Due to light physical activity	0.13 (0.35)	0.00 (0.00)	0.17 (0.41)
Pain-related school absence in the last four weeks	–	0.61 (1.34)	0.38 (0.63)
Treatment satisfaction	–	6.00 (2.45)	7.00 (2.76)

^aValues are given as mean (standard deviation).

Children’s ability to attend school was assessed by measuring the mean number of pain-related school absences in the last four weeks before the follow-up visits. Our data show that the mean number of school absences almost halved from month 3 to year 2 following treatment. While the mean number of days with functional impairment following hard, moderate and light physical activity decreased at month 3 following treatment versus baseline, the beneficial treatment effect could only be sustained long term in the group with moderate physical activity (see Table 3).

Our results showed a reduction in pain medication intake following IIPT. Before treatment (at baseline), four children had been taking drugs more than 10 times a month, whereas after three months, only one child took medications more than 10 times a month. Similar findings were observed at year 2 following the initial treatment (data not shown). Patients’ and their parents’ satisfaction was maintained throughout the study (see Tables 2 and 3).

The comparison of average pain and pain related disability scores in our study with those published in a systematic review by Hechler et al. (2015) using Cohen’s (1988) effect size methodology showed less pronounced improvements in average pain scores and an increase in pain-related disability scores at the short-term (month 3) follow-up in our study (Supplementary table, Table 4).

Table 4.

Effect size calculations.

Measures	Effect sizes (d) from baseline to short-term follow-up (three months)	Effect sizes (d) from baseline to long-term follow-up (two years)	Effect sizes (d) from baseline to short-term follow-up (Hechler et al. 2015)
Pain intensity (last seven days)	−0.28	−0.35	−1.33
Pain-related disability	+0.82	+0.15	−1.35

Discussion

Our study is the first pilot to show sustainable positive effects of a two-week IIPT on pain-related clinical outcomes among children and adolescents suffering from severe chronic pain in the Hungarian healthcare setting. Although the duration of our IIPT was only nine working days, these results are comparable to other paediatric studies with longer treatment periods where the average treatment duration was 15. 09 working days (Banez et al., 2014; Chalkiadis, 2001; Eccleston et al., 2003; Gauntlett-Gilbert et al., 2013; Hechler et al., 2010, 2014; Hirschfeld et al., 2013; Lanzi et al., 2007; Maynard, et al., 2010; Palermo and Scher, 2001; Sherry et al., 1999; Simons et al., 2012; Weiss et al., 2013).

The comparison of our results with those presented in Hechler et al.’s (2015) systematic review suggested that the effect sizes of the outcome measures in our study at short-term follow-up were smaller. This could be attributed to (1) the shorter mean treatment duration of our study (six days shorter than that presented in the systematic review); (2) differences in baseline patient characteristics between the two studies, namely that our patients were older, had less severe pain intensity and disability as well as shorter pain duration; (3) the timing of study during the summer months with school holidays, which might have biased disability assessments and (4) the small sample size of our pilot.

Data on the long-term impact of interdisciplinary pain treatments are scarce. Our study is among the first ones to demonstrate clinically meaningful improvements in pain-related outcomes that were sustainable over two years after initial intervention. Furthermore, our results suggest that IIPT improved problem-based coping, an effective way of managing pain (Reid et al., 1998).

The strength of our study is the use of a booster session three months after the intervention (Kirchner et al., 2014). Our results suggest that booster sessions in combination with short in-house interventions may play a role in maintaining durable clinical impact.

Our results on the beneficial impact of IIPT on healthcare burden (i.e. number of hospital visits and drug use) are important in the Hungarian setting where healthcare resources are scarce, further increasing the significance and relevance of its utility.

This study has several limitations including the small sample size, the over-representation of girls in the sample, the lack of control group, self-administered questionnaires and resulting bias, and the subsequent lack of causality; however, we have tried to overcome this by using a longitudinal design.

Conclusions

Our two-week IIPT has demonstrated clinically relevant short- and long-term improvements in pain-related outcomes and healthcare utilization among children and adolescents suffering from severe chronic pain. However, these results can only be considered as preliminary due to the limitations mentioned above and further large-scale studies are required to confirm our findings.

This programme design may represent an effective and efficient approach to manage chronic paediatric pain, which is more suitable for the healthcare system in Hungary, and thus improve access to an effective treatment modality of chronic paediatric pain management.

Footnotes

Acknowledgements

The authors wish to thank Prof. Boris Zernikow, Michael Dobe, Tanja Hechler and the German Paediatric Pain Centre for their support. The authors are grateful to Anikó Hirsch, MD, Flóra Sztanó, MD, Ildikó Fléger, Judit Mag, Gabriella Szegleti, Anett Sorompó, Sándor Németh and the whole team in the Department of Psychosomatic Medicine for their work in this study.

Declaration of conflicting interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

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