Background
The International Association for the Study of Pain (IASP) defines pain as “an unpleasant sensory and emotional experience associated with actual or potential tissue damage, or described in terms of such damage” [
1]. Although definitions of what constitutes “chronic” pain vary, the IASP definition of pain lasting more than 3 months is widely accepted. Walsh, Morrison and McGuire [
2] examined chronic pain in adults with an intellectual disability and found that chronic pain was experienced by 15% of adults with an intellectual disability, based on caregiver report. Whilst this is consistent with reports of the frequency of chronic pain in the general population, it has been suggested that this may be an under-estimate of the extent of the issue in those with intellectual disabilities, especially amongst those who are non-verbal or have a more severe level of disability [
3]. As those with more severe intellectual disabilities are not always able to verbally communicate their pain to carers, their pain experience may not always be recognised and reported. Whilst the use of proxy respondents can also be beneficial in gathering information about the pain experience of those with significant intellectual disabilities and communication challenges, this method presents its own challenges including the issue of reliability of carer report. Other methods such as structured behavioural observation offer a reliable and valid alternative [
4] and the use of more than one source of information increases the reliability of the information obtained.
Dysmenorrhea (menstrual pain) is extremely common with as many as 90% of menstruating female adolescents and 50% of women reporting that they suffer from it [
5,
6]. Kyrkou [
7] examined how menstrual pain presents in women with intellectual disabilities as there is anecdotal evidence of an increase in this condition in this population but little research has been conducted in this area. The parents of 24 women with Down Syndrome or Autism Spectrum Disorder (ASD) were surveyed to ascertain how menstrual pain presents in women with intellectual disabilities. Results suggested that two thirds of the women with Autism, three quarters of the women with Down Syndrome and all of the women with Aspergers Syndrome appeared to have problematic period pain. These rates were higher than the 50% rate reported for women in the general population [
6] however, due to the small sample size used in the study, caution is required in the interpretation of these findings.
Dysmenorrhea, defined as pain during menstruation which is severe enough to impact or interfere with daily activities [
8] has recently been the focus of brain-imaging studies which have shown that the brains of otherwise healthy women with moderate-to-severe dysmenorrhea show significant differences in brain structure and function, when compared with non-dysmenorrheic women. Tu, Niddam and colleagues [
9‐
11] identified differences in cerebral metabolism and cerebral structure for the trait of dysmenorrhea and between pain and pain-free states. Differences have also been found in neural activity induced by noxious skin stimulation when applied to areas remote from the pelvic/abdominal region, such as the arm [
12]. An important aspect of these brain-imaging studies is that some of the differences in neural characteristics occurred chronically, throughout the menstrual cycle, even when dysmenorrheic women are not experiencing menstrual pain [
13]. Berkley [
14] suggests that the consistency of these findings along with those from individuals with other chronic pain conditions provides a strong argument that dysmenorrhea should be considered a chronic pain condition.
Given the potential for significant personal, social and economic impact from chronic pain, much research attention has been directed towards pain management and treatment options. In a review of psychological therapies for the management of chronic pain in the general population, Eccleston, Williams and Morley [
15] found that CBT results in improvements in overall functioning and psychological wellbeing. There is also evidence of the effectiveness of such approaches for the treatment of dysmenorrhea [
16].
There has been increasing interest in adapting CBT for use with people with an intellectual disability and a few authors have described the use of modified CBT. McCabe [
17] described the effectiveness of treatment programmes for depression among adults with mild-moderate intellectual disabilities while Lindsay, Neilson and Laurensen [
18] were successful in using this approach to treat anxiety in this population. Willner, Jones, Tam and Green [
19] examined the efficacy of a cognitive-behavioural anger management group for individuals with an intellectual disability using a randomized controlled trial research design. They found that clients in the treated group improved on both self- and carer-ratings, relative to their own pre-treatment scores, and relative to the control group post-treatment. Individuals in the intervention group also showed further improvement relative to their own pre-treatment scores, at 3 month follow-up. McGuire and Kennedy [
4] noted that there has been limited research evaluating CBT for chronic pain in people with an intellectual disability. An important advance in the area was the development of “
Feeling Better – a manual for carers working with people who have intellectual disabilities and chronic pain” [
20]. This modularised programme uses cognitive behavioural principles to teach individuals with intellectual disabilities a range of strategies to manage chronic pain more effectively. In a case series study by McManus and McGuire [
21], some preliminary evidence was provided for the effectiveness of the programme with increases in participant scores on pain management knowledge, wellness-focused coping and effectiveness of coping following the intervention.
In view of the ample evidence that CBT can be used for chronic pain management including the management of dysmenorrhea in the general population, and the preliminary evidence for effectiveness in people with intellectual disability [
21], there is a rationale for evaluating a CBT-based pain management programme for menstrual pain in women with an intellectual disability. This study will be the first controlled clinical trial to address the issue of menstrual pain management with individuals with intellectual disabilities. Research on pain in individuals with intellectual disabilities has largely focused on identification of pain and medical management of pain symptoms. Pain management has largely been ignored and pain management programmes have not routinely been offered to such individuals.
Study aims & objectives
This pilot study aimed to evaluate if a theory-based cognitive-behavioural therapy programme for menstrual pain management (derived from the
Feeling Better manual) is more effective than usual care in helping young women with intellectual disabilities to manage their menstrual pain. In order to answer this question, a small non-randomised controlled trial was conducted comparing the use of menstrual pain management programme with usual care in an intellectual disability service in the west of Ireland. The study included a number of potential secondary outcome measures in addition to the primary outcome measures of pain coping strategies used and pain knowledge. It will enable us to determine if:
1.
Participation in the menstrual pain management group results in an increase in participants’ ratings of pain coping strategies, pain management knowledge and pain self-efficacy and if this is maintained at 3 month follow-up.
2.
Participation in the menstrual pain management group results in a reduction in self-ratings of pain intensity and pain interference by participants and reduced ratings of pain intensity and pain interference amongst participants, as rated by their parents, and if these reductions are maintained at 3 month follow-up.
3.
Participants whose parents score highly on pain-catastrophizing experience greater pain intensity and greater pain interference with quality of life. Catastrophizing – experiencing extremely negative thoughts about one’s plight and interpreting even minor problems as major catastrophes – appears to be a powerful way of thinking that greatly influences pain and disability and is important in determining one’s reaction to pain [
22]. Goubert, Eccleston, Vervoort, Jordan and Crombez [
23] found that parents’ catastrophic thinking about their child’s pain had a significant contribution in explaining the child’s disability and school attendance. For this reason, parental catastrophizing is expected to affect participants’ ratings of pain intensity, pain interference and pain coping strategies used.
4.
Participants in the menstrual pain management group adopt more behavioural than cognitive coping strategies to manage their menstrual pain. Burkitt et al. [
24] found that greater developmental level (rather than chronological age) was associated with the use of more cognitively demanding strategies as may be used in cognitive behavioural therapy treatment approaches. McManus and McGuire [
21] also reported a marked absence of the use of cognitive coping skills. Participants were more likely to use behavioural strategies such as exercise and relaxation.
Process evaluation was conducted via qualitative focus groups with participants, parents and support staff. The purpose of this was to assess the acceptability of the intervention to participants, to explore their experiences including any suggestions they may have to further enhance the programme and to examine which elements of the programme were most successful in promoting change for young women with intellectual disabilities who experience menstrual pain. It was envisaged that this innovative approach would yield valuable information which could form the basis for effective interventions to enrich the quality of life of individuals with intellectual disabilities who experience menstrual pain, as well as enhancing the lives of their carers.
Discussion
In this study, we evaluated the impact on pain coping and pain management knowledge, of a menstrual pain management programme for young women with intellectual disabilities. We expected participants in the intervention group to report the use of a greater number of coping strategies and have greater knowledge of pain management strategies at both T4 and T5, compared to control group participants.
The content of the menstrual pain management programme was adapted from the theory-based cognitive behavioural therapy programme “Feeling Better – A manual for carers working with people who have intellectual disabilities and chronic pain” [
20]. The programme that was developed reflected the input from the parents/guardians of research participants, on both content and delivery methods. The PRP qualitative component ensured that the theory-based programme was specifically tailored to meet the needs of this population. As stakeholders, parents/guardians have insights into the training needs of their daughters and the possible challenges in delivering such a programme. Caregivers control access to medical/health services [
3,
38], so it is very important that they are involved with health interventions for those who are in their care.
A significant advantage to this study was the inclusion of process evaluation to determine which elements of a cognitive behavioural therapy programme work best for individuals with intellectual disabilities. Hunter [
39] identified the need for clarification on effective components of CBT approaches for pre-menstrual symptoms and this is particularly relevant for this population. Moderator analyses of the outcome of the intervention will enable a better understanding of who this type of training is most effective with and under what conditions. Such information will enable us to optimize treatment for each individual into the future. For this reason, a number of variables which are assumed to be related to pain coping were measured in this study.
Competing interests
The authors declare that they have no competing interests.
Authors’ contributions
SK conceived of the study, participated in its design, is responsible for delivery of the intervention, and drafted the manuscript. SOH conducted the programme development focus group and collected outcome measures data. KS participated in the design of study, advised regarding statistical analysis and helped to draft the manuscript. CW participated in the design of the study and helped to draft the manuscript. BMcG participated in the design of the study and helped to draft the manuscript. All authors read and approved the final manuscript.