Introduction

The development of an individual’s sexual identity and sexual expression is a complex biopsychosocial phenomenon with influences ranging from genetics to sociocultural factors. It is well established that chronic illness and disability can have significant deleterious effects upon sexuality and quality of life (QOL) in women [1–3]. Multiple sclerosis (MS) and spinal cord injury (SCI) are two such disabling conditions [4, 5]. In Canada, MS prevalence is known to be high, with recent estimates ranging between 55 and 240 per 100,000 [6]. The prevalence ratio of women to men with MS, as reported by a Scandinavian study is 2.35:1 [7]. SCI prevalence (traumatic and non-traumatic injuries) in Canada is estimated at 2525 per million population and with a minimum male-to-female ratio of 2:1 [8].

These neurological disabilities can profoundly affect one’s sexuality in many ways and at many levels. In the literature on MS and sexuality, the sexual sequelae have been categorized as having three levels of responsible factors: primary, secondary and tertiary [9]. Primary effects refer to any sexual difficulties resulting directly from the impact of the disease or injury on neurophysiology (e.g. altered genital sensation, anorgasmia, and decreased biological desire). Secondary effects include any disruption in sexual health from indirect factors associated with the condition (e.g. fatigue, pain, spasticity, bladder and bowel issues, or side effects of medications). Tertiary effects refer to body image changes and altered perception of “sexual self” due to negative societal stereotypes and myths existing around sexuality and disability. One study did examine the frequency, distribution and interrelation of these primary, secondary and tertiary factors [10]. In a cross-sectional design of 271 women with MS, 172 or 63.5 % of women had sexual dysfunction (SD). Of these 172 women, 52.4 % had SD related to primary factors, 37.5 % had secondary SD, and 41 % had tertiary SD.

The sexual consequences for women with SCI could also be organized in terms of primary, secondary and tertiary factors. Although, perhaps more important to note is the fact the responsible factors are interrelated and the sexual outcomes and possible interventions are complex [11]. In a large-scale survey of both women and men with SCI [12], 83 % agreed that their SCI had altered their sexual sense of self. In the same survey, 83 % said that improving sexual function was important in improving their quality of life. Research on female sexual response and SCI has reported about half of the women experienced vaginal lubrication and only a third experienced orgasm after SCI [13]. More current research revealed that orgasmic experience and genital sexual arousal, although possible even for women with complete injuries, required increased intensity and duration of stimulation [14]. In addition, bladder and bowel issues and autonomic dysreflexia are known to negatively impact sexual activity and intercourse [12]. Furthermore, the length of time required to adjust to sexual health challenges post-SCI may take upwards of one to two decades, and is often reliant on the presence and involvement of a long-term partner and remaining open-minded to sexual pursuits [15].

In order to address the sexual concerns of this population of women (and men) with neurological disabilities, the Sexual Health Rehabilitation Service has offered an interdisciplinary, comprehensive, client-centred approach to health care delivery for over 40 years [16, 17]. The model of service delivery involves the application of restorative and rehabilitation principles and is based on a specialized body of knowledge generated through research and clinical practice evidence. Practitioners of sexual health rehabilitationFootnote 1 offer education, medical and nursing interventions, and emotional support to best address their clients’ sexual concerns. One overarching goal of sexual rehabilitation is facilitating an individual’s (or couple’s) sexual adjustment following a change in health status. In earlier studies on populations of persons with disability, the term “sexual adjustment” was linked to sexual self-concept and adapting to sexual limitations, as well as to partner satisfaction and frequency of sexual activity [18–20]. In this study, the term “sexual adjustment” refers to the changes one might make to move towards a positive sense of sexual well-being. Sexual well-being is defined by Laumann et al. [21] as “the cognitive and emotional evaluation of an individual’s sexuality”. Sexual self-view/self-esteem, are terms also used in sexual rehabilitation, and refer to how a person is able to define themselves as a sexual being in light of their disability [22]. Stevenson and Elliott [23] further elaborate upon this definition adding that: “Illness and the related issues (e.g. continence, hygiene, independence) can adversely affect one’s sense of sex appeal, masculinity or femininity. Previous social roles may be upset or reversed, further undermining sexual confidence”. Furthermore, former personal beliefs about sex and disability may influence how they view themselves post injury or illness and their sexual confidence may be affected as a result [22, 23].

Having an effective intervention to accelerate the sexual adjustment for women with disabilities would greatly enhance their quality of life, however, there have been no such intervention-based studies to date. While existing qualitative research has been explorative in nature, it has offered direction for intervention studies. One example is a study by Ekland and Lawrie [24], who surveyed ten women with SCI and identified common themes around what factors support sexual adjustment. These factors included: access to peer support or mentors, participation in a women’s group, and availability of health care professionals. Forsythe and Horsewell [25] also affirmed that women with SCI wanted to discuss issues with other women who had experienced similar problems.

Purpose of Study

This study was designed to offer an existing intervention tool to women with MS and SCI. The treatment combined cognitive behavioural therapy, mindfulness based skills, and education, and was originally developed and found to be effective for female cancer survivors as well as a more general population of women experiencing sexual difficulties [26]. The purpose of this study was to adapt treatment to a specific population of women with disabilities. We predicted that, compared to pre-treatment, this adapted intervention would result in a significant improvement in the primary endpoint of sex-related distress. We also predicted improvements in secondary endpoints of sexual functioning, relationship functioning, mindfulness skills, and body image satisfaction.

Materials and Methods

Participants

Participants for this study were recruited through advertisements. A research coordinator, trained by a sexual medicine psychologist, conducted the telephone screening interviews to determine participant eligibility. Inclusion criteria were women between the ages of 19 and 65 who had been living with MS or SCI for at least 1 year, were fluent in English, and who met the criteria for Female Sexual Arousal Disorder (FSAD) or Hypoactive Sexual Desire Disorder (HSDD) according to the Diagnostic and Statistical Manual of Mental Disorders, 4th edition [27]. Participants were screened for depression using the Beck Depression Inventory (BDI), a validated measure for depression [28]. Participants were excluded if they had a score >19, given that severe depressive symptoms would have interfered with participants’ ability to complete the homework assignments. Ethical permission to conduct the study was obtained through the university and associated medical center where this research took place. All participants gave their informed consent prior to participating.

Procedure

The study intervention consisted of five 90-min sessions, spaced 2 weeks apart, completed within a 10 week time frame. Questionnaires were administered at three junctures of the study: immediately prior to, immediately following, and 6 months after completing treatment. The groups were co-facilitated by a sexual health clinician (registered nurse specializing in sexual health rehabilitation) and a registered clinical counsellor (M.Ed., R.C.C.). Homework exercises were assigned at the end of every session and discussed at the start of the following group session. Following treatment, a research coordinator conducted a focus group with three study participants and one clinician to obtain qualitative feedback about participating in these groups and on next steps for program development. This research is a triangulation of both qualitative and quantitative measures.

Measures

Primary Endpoint: Sex-Related Distress

Sex-related distress was measured with the 12-item Female Sexual Distress Scale (FSDS), a valid, reliable measure of sexually related distress in women [29].

Secondary Endpoints

Our secondary endpoints focused on sexual functioning and arousal, relationship functioning, mindfulness skills, and body esteem related to having a physical disability. Different domains of sexual functioning were assessed with the Female Sexual Function Index (FSFI), a 19-item validated measure of sexual response in women [30], and the 32-item Detailed Assessment of real-life Sexual Arousal (DASA), an unpublished questionnaire used in the clinical setting to assess self-reported mental sexual arousal, genital tingling, genital wetness, and self-reported pleasure in response to a variety of different types of sexual stimulation [31]. Relationship satisfaction was assessed with the 32-item Dyadic Adjustment Scale (DAS), a reliable, valid and long-utilized measure of relationship adjustment [32], and mindfulness was measured with the Five Facet Mindfulness Questionnaire (FFMQ), a 39-item valid and reliable instrument [33]. Of note, this measure has been used previously with patients with fibromyalgia and was found to be a reliable and valid measure with which to assess mindfulness in that population [34]. We also assessed body esteem with the 10-item Physical Disability Sexual and Body Esteem Scale (PDSBE), a scale with strong psychometric properties, used to measure the capacity to feel positive about one’s sexual self and body esteem while living with a disability [35].

Group Intervention

The group intervention was modeled after a similar intervention that has been developed and found effective for cancer survivors with sexual dysfunction, and more generally for women seeking treatment for low sexual desire [36, 37]. In brief, the intervention combined elements of: education, Cognitive Behavioural Therapy, and Mindfulness Based Therapy. A modified version of this effective treatment was developed for the current population of women who experience a physical disability.

Adaptations to the treatment intervention for this population included: (1) adding information regarding the prevalence of sexual difficulties in MS and SCI populations; (2) incorporating “body-mapping” exercises into the set of existing sensate focus or body exploration exercises (see Table 1). “Body-mapping” involves the creation of a visual diagram of the entire body, followed by application of a sensory rating scale (−5 = very uncomfortable; 0 = no sensation, and +5 = pleasurable). The third addition was to include a description of the female internal reproductive anatomy, in response to previous research findings [38]. The addition of the mindfulness practice of “observing judgements” was the fourth and final adjustment to the existing treatment. The rationale behind this focus on “observing judgements” stemmed from literature which points to more women with disabilities having a negative body image and sexual self-view [39]. The process involved being “mindful” about their subjective judgements when exposed to different objects (ones that typically evoke positive or negative responses). This exercise was meant to give women an opportunity to recognize the impact of negative self-talk on their own sexual self-view. In addition to the aforementioned changes to the treatment, disability specific research and information was also provided to the participants. Throughout this manuscript, we refer to the intervention as d-PED (psychoeducational treatment adapted for women with disability).

Table 1 Description of PED exercises (d-PED additions are highlighted)
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Data Analysis

Given the small sample size, this study was primarily interested in effect sizes, for which we used Cohen’s d. In addition, we calculated a within-subjects repeated measures analysis of variance (ANOVA) comparing participant scores across the three time points. We predicted significant changes from pre- to post-treatment, as indicated by at least moderate effect sizes, and we predicted no significant change (i.e. no loss in improvement) from post-treatment to follow-up, for the primary endpoint of sex-related distress.

Results

Subject Recruitment and Attrition

From 2009 to 2011, a total of twelve women who met the inclusion criteria consented to participate in this study. One woman withdrew before providing any baseline data due to extenuating circumstances that made travelling to group sessions too burdensome. Eleven women provided baseline data (mean age 47.9, SD 11.2, range 25–61 years). Of the remaining eleven subjects, one woman withdrew for unknown reasons before starting the d-PED. Another woman requested to withdraw before she started the d-PED because she was no longer able to make the time commitment. Two women did not receive the d-PED intervention due to insufficient enrolment for a third group offering. These women were referred back to individual counselling with a sexual health clinician. The seven remaining subjects completed all five sessions of the d-PED and the pre- and post-intervention questionnaires. Six out of seven participants completed the final 6-month questionnaire. The seventh participant was unable to complete the final questionnaire due to a severe relapse in her MS that required hospitalization. At the 6-month post d-PED time point, one subject’s information was missing from the FSFI data set due to not being sexually active within the 4 weeks time period stipulated by that particular questionnaire.

During the study period, two separate d-PED group interventions were held. The first group included three participants and the second group included four participants. A third group was cancelled due to low participant recruitment.

Participant Characteristics

Of the seven participants who participated in the d-PED, five women reported being in a long-term, monogamous, heterosexual relationship; one woman was in a new heterosexual relationship (about 3 months), and the seventh participant identified as heterosexual, divorced and currently single. Two of the six women in a relationship reported being satisfied with the level of closeness in their relationship at study entry. All women identified as being Caucasian. Six women had been diagnosed with multiple sclerosis (onset ranged from approximately 2.5 to 42 years) and one woman with spinal cord injury had incomplete paraplegia (almost 3 years post-injury). In terms of severity of disability, one participant rated her disability as mild, three participants rated their disability as moderate and three participants rated their disability as severe. Two subjects with MS were ambulatory and the other five women used some form of mobility aid (electric or manual wheelchairs), one being dependent for transfers. All but one participant had completed a post-secondary education program.

Effects of Treatment on the Primary Endpoint: Sex-Related Distress

An examination of effect sizes on sex-related distress between each of the time points showed a strong effect of treatment from pre- to post-intervention, Cohen’s d = 1.19 (Table 2). However, this effect did not reach statistical significance, F(2,10) = 2.11, p > .05.

Table 2 Female sexual function index (FSFI), detailed assessment of real-life sexual arousal (DASA) and female sexual distress scale (FSDS) subscales score comparison between baseline, 2 weeks post-intervention and at 6 months post-intervention
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Effects of Treatment on Secondary Endpoints

Sexual Functioning and Arousal

There was a significant effect of treatment on FSFI desire scores, F(2,10) = 4.64, p = .038. Among women who reported having participated in “sexual activity” during the previous 4 weeks, a similar significant effect was also found for FSFI arousal, F(2,10) = (2,8) = 10.38, p = .006. The increase in orgasm scores was not significant, F(2,8) = 3.09, p > .05, though there was a very strong effect size from pre- to post-treatment. Reports of genital pain similarly did not significantly change with treatment, F(2,8) = 0.55, p > .05. Sexual satisfaction did not significantly change with treatment, F(2,8) = 1.46, p > .05, though there was a very strong effect size from pre to post-treatment. Overall sexual functioning did significantly increase with treatment, F(2,8) = 6.08, p = .025, and the effect size was very large. Given the established clinical-cutoff for scores on the FSFI total score (26.55), it is evident that women in our sample had a significant improvement with treatment that fell into the non-clinical range, but then some of those gains were lost at the 6-month post-treatment evaluation point. Self-reported disability severity is not correlated with any of the FSFI subscales. Years post disability is negatively associated with sexual desire, but the p value is just short of significant (p = .091). Years post disability is also associated with higher sexual arousal (p = .006). On the measure of sexual arousal, there was no significant increase in DASA mentally excited subscale scores, F(2,10) = 0.80, Genital Tingling, F(2,10) = 1.34, or pleasant genital sensation, F(2,10) = 0.08, p’s > .05; however, the increase in genital wetness was significant, F(2,10) = 5.04 p = .03. For the genital tingling and wetness domains, the effect size corresponded to a very strong effect with treatment. Effect sizes associated with each of the changes between pre and immediately post treatment, and from post-treatment to follow-up, are presented in Table 2.

Relationship Satisfaction

None of the subscales of the DAS reached statistical significance following treatment: consensus subscale, F(2,10) = 3.29, p > .05; satisfaction subscale, F(2,10) = 1.52, p > .05; cohesion subscale, F(2,10) = 0.94, p > .05; affectional expression, F(2,10) = 3.07, p > .05. Consensus and Satisfaction score increases saw strong effect sizes associated with these non-significant changes (Table 3).

Table 3 Dyadic adjustment scale (DAS), five facet mindfulness questionnaire (FFMQ), and physical disability sexual and body esteem scale (PDSBE) subscales score comparison between baseline, 2 weeks post-intervention and at 6 months post-intervention
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Mindfulness

Scores on the FFMQ observing subscale improved (though not-significantly) with treatment, F(2,10) = 2.14, p > .05. Similar non-significant effects were found with FFMQ awareness, F(2,10) = 1.48, p > .05, non-reactivity, F(2,10) = 0.85, p > .05, and describing, F(2,10) = 0.82, p > .05 subscales. Non-judgment significantly decreased with treatment, F(2,10) = 6.08, p = .019 (Table 3).

Body Esteem

On the physical disability sexual and body esteem scale, there were no significant effects of treatment on the sexual esteem subscale, F(2,10) = 1.45, p > .05; Attractiveness, F(2,10) = 1.27, p > .05; Body Esteem, F(2,10) = 1.62, p > .05; or Total score, F(2,10) = 1.62, p > .05. All effect sizes were small to moderate (Table 3).

Qualitative Data Analysis

Participant feedback forms and transcripts from the focus group were read separately by the research coordinator and sexual health clinician and after grouping similar subject matters, themes were identified. A consensus was then reached in identification of major occurring themes. The themes were: (1) The value of mindfulness practice; (2) How exploring sexuality and sexual self-view has a positive effect on self-acceptance; (3) Benefit of peer interaction. The following quotes from the participants were taken from the feedback form and elucidate the key themes.

Value of Mindfulness Practice

… [Mindfulness] was the most difficult thing to remember but probably the most valuable tool.

Mindfulness…finally some ability to let go of the “must do”.

The Effect of Exploring Sexual Self-View

It raised my awareness that even though I am 60 and very disabled, it is not unreasonable to reconsider allowing my sexual being a chance to re-emerge.

Session reinforced a broad, liberal view of sexuality: I may not be perfect, but I am Okay.

… talk yourself into the fact that you are worth it… it is worth taking 2 h out of your life every 2 weeks, just for you.

Benefit of Peer Interaction

discussions with other women in the same situation was helpful.

communication: I feel a lot better equipped to tackle difficult topics.

In addition to the themes, the study participants made comments on the effectiveness of the d-PED format as well as suggestions for improvement to any future group processes. General comments on the d-PED process included:

In the act of doing this [group], it opened a door. It was kinda neat

When I first started this I didn’t really know what to expect… I guess, you know, I was kind of hoping maybe it would like kick start something. And it didn’t really do that, in you know, in my mind, but it helped. It was a beginning.

I wish I would have known this stuff 25 years ago

Study participant suggestions for improving future groups included: more time for discussion; less content in each session; provide audio for mindfulness practice once group ends; more in-session mindfulness exercises (including body image and pain relief); more body image content/discussion as it relates to CBT and Mindfulness; incorporating a partner component (i.e. handouts/guides for partners). One participant suggested that 2–5 years post diagnosis would be the best time to do the exercises in the group.

Discussion

Recruitment Challenges

In terms of recruitment, one significant limiting factor was the DSM-IV criteria for HSDD and FSAD in that subjects needed to report “significant distress either to you personally or in your relationship”. The element of “distress” did not really reflect the women’s subjective experience with desire or sexual arousal. This had implications for the number of participants that were eligible for the study. Some potential subjects did not pass the pre-screening for this study because, although they’d experienced changes in desire and arousal since the diagnosis, they did not perceive these as cause for “distress” per se. In fact, in response to this recruitment challenge, the researchers changed the screening question to read: “does this difficulty bother you or cause strain in your relationship.” Perhaps women with disabilities tend to not fit the “distress” criteria for FSAD and HSDD because they have already experienced losses in many other aspects of their life in the form of secondary and tertiary effects of MS or SCI (body image, mobility, bowel and bladder function). In comparison with the distress caused by these losses, the sexual function changes may be viewed as less significant, or even as an expected consequence of the disease/disability [40, 41]. Despite the fact some of the participants did not fit the strict criteria for FSAD and HSDD, they were indeed interested in working on the sexual part of their lives.

Another issue related to recruitment was the difficulty enrolling women with spinal cord injury in the study. Despite a relatively even number of inquiries from both women with SCI and MS, only two women out of the twelve who consented to participate had a spinal cord injury. Possible barriers to study participation for these women were inability to take time off work, transportation issues, and/or living out of town. The development of an on-line based d-PED intervention may address some of these accessibility issues for this population.

Limitations of Study

We acknowledge that the small sample size is a major limitation of this pilot study. It is, therefore, difficult to attribute study findings to the d-PED intervention directly. While effect sizes suggest that the treatment was effective, or at least trending towards such, caution must be taken in drawing any conclusions given the small sample size. Future studies including an adequately powered sample size are needed in order to verify these findings. The fact there is only one subject with SCI who completed the d-PED, also limits our ability to draw conclusions based on this sample. We opted to use this subject’s data, however, as our clinical experience is that many of the psychological adjustment issues are similar for women with MS and with SCI.

Another limitation of this pilot study is the possible biases created by subject attrition, despite the fact that most women withdrew due to extenuating circumstances unrelated to the treatment. Subject selection bias may also have been a factor given most women were in long-term monogamous relationships. Furthermore, as this is a pilot study, results lack generalizability to the larger population of women with spinal cord injuries and other disabilities who also experience sexual difficulties.

Relevance of Findings

Despite a very small sample size, our quantitative findings suggest that this d-PED intervention may be beneficial for women with MS and SCI in improving their sexual functioning. Self-reported severity and years since disability were not associated with pre-treatment levels of sexual functioning. The only exception was that women with more years since diagnosis tended to have lower desire, but this could simply be a function of age. Interestingly, more years post disability was associated with higher sexual arousal. Perhaps women, despite losing their desire, become more in tune with their bodies and sexual arousal responses.

On the FSFI domains of sexual desire and arousal, although the gains were most significant, they did not sustain into the 6-month post-treatment evaluation point. In contrast, the application of a similar intervention to women with sexual dysfunction secondary to gynaecologic cancer found that significant gains were retained at the 6-month follow-up period [42]. The reasons why these two populations had different long-term responses to a similar treatment are not known; however, it may be that baseline differences in the rates of sexual difficulties may have contributed to this. There is also evidence that women with more depressive symptoms benefit more from this type of intervention that women with low depressive symptoms [43]. Again, it may be that co-occuring mood symptoms may have contributed to the lack of long-term efficacy. Finding a way to maintain these gains over time will be key to the success of future d-PED interventions. In this study it may be that the regular discussion and increased thoughts of sexuality contributed to the positive outcomes in the 2 weeks post scores. Sustaining this discussion and sexual thoughts into the future could be accomplished through periodic electronic connection (with clinicians and/or peers). An on-line version of this intervention with a group chat or peer mentoring portion may be something to consider in future offerings of this intervention. This could also mitigate the issues around accessibility of the groups.

The mindfulness-based skills within the treatment also lead to a significant decrease in judgement of their inner experiences (i.e. they were less likely to place judgment upon their thoughts and feelings). The results from the qualitative data further substantiate the concept of mindfulness as an important part of a woman’s experience of her sexuality. The d-PED allowed women to be less judgmental of their individual experiences as evidenced by this quote from one of the participants: “I may not be perfect, but I am Okay”. As sexual adjustment for women with disabilities can often be delayed up to two decades, [16, 39] this intervention has the potential to be very beneficial to this process. One participant stated: “It raised my awareness that even though I am 60 and very disabled, it is not unreasonable to reconsider allowing my sexual being a chance to re-emerge” and another “wished she had known this stuff 25 years ago”.

The d-PED permitted women to share their experiences with other women who were of similar circumstances. This intervention satisfies the recommendations from previous studies in this area [25, 26, 44] which include: peer support, group format, and access to health care professionals.

Sexual Self-view is an area in sex and disability research that has been given some attention, yet little is known about therapeutic interventions. Tepper [16] describes the journey towards sexual self-acceptance experienced by women after a SCI, which includes the concepts of “cognitive-genital dissociation,” followed by “sexual disenfranchisement,” then “sexual rediscovery.” It is possible that this d-PED intervention might expedite the process of adjusting to a disability/illness and establishing a more positive sexual self-view. This theory informs future practice and interventions geared towards women with SCI and MS. Ultimately the value of the d-PED for women with disabilities/chronic illness may lie in its potential to enhance sexual self-view and quality of life rather than as a treatment for sexual dysfunction.

Conclusion

More research is required to determine the clinical relevance of utilizing the d-PED in the population of women with disabilities. However, this preliminary trial was a first attempt at an intervention-based study in this field and the results were encouraging. In fact, findings suggest this d-PED intervention, specifically the mindfulness-based practices and peer involvement, is beneficial for women with MS and SCI. Given the challenges with recruitment and retention, the authors would consider the following for a future project: removing the DSM-IV criteria for FSAD and HSDD as an inclusion criterion for participation; and the development of an on-line version of the intervention to address the accessibility issues in this population. The authors recommend using these findings to inform the development of future research and clinical endeavors.