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Persistent Genital Arousal Disorder: a Biopsychosocial Framework

Persistent Genital Arousal Disorder: a Biopsychosocial Framework Purpose of Review Persistent genital arousal disorder (also referred to as genitopelvic dysesthesia or PGAD/GD) is a distressing and largely underrecognized condition characterized by persistent, unwanted genital arousal (sensations, sensitivity, vasocongestion) in the absence of subjective/cognitive arousal and sexual desire. The purpose of this review is to summarize recent findings on biological and psychosocial factors in PGAD/GD as they pertain to the assessment and treatment of this condition. These findings will be considered within a biopsychosocial framework, for the purposes of considering next steps for clinical and research efforts. Recent Findings A small number of studies have recently examined potential biological aetiologies for PGAD/GD: pharmaco- logical agents, spinal pathology and peripheral nerve involvement. Recent studies have also found that PGAD/GD is associated with a significant negative impact on psychosocial wellbeing and daily functioning as compared to symptom-free individuals. In addition, these results highlight cognitive/affective responses to symptoms (e.g. catastrophizing of symptoms) that may influence outcomes. However, biological and psychological research are rarely integrated in these studies, despite the interrelationship between these factors. Summary Although PGAD/GD was first described in the scientific literature almost two decades ago, most research on PGAD/GD is presented in the form of case studies. Prospective treatment trials that integrate biopsychosocial factors are needed in order to provide effective and efficient care to this population. This research would be facilitated by the development of a patient-reported outcome measure, as well as greater education/awareness among healthcare providers and the public about this distressing condition. . . . . . Keywords Persistent genital arousal disorder Genitopelvic dysesthesia PGAD/GD Biopsychosocial Catastrophizing Fear avoidance Introduction may last for hours to days, while for others, these symptoms may be continuously present [2]. Symptoms may be triggered First described by Sandra Leiblum and Sharon Nathan in by sexual cues, non-sexual cues or there may be no identifi- 2001, persistent genital arousal disorder (or PGAD) is a highly able trigger [2]. A subset of individuals with PGAD also re- distressing condition, characterized by genital arousal (e.g. port experiencing distressing and unwanted spontaneous or- genital sensations, sensitivity and/or vasocongestion) that oc- gasms [2]. PGAD symptoms are not relieved by any behav- curs in the absence of subjective/cognitive arousal or sexual iour (e.g. masturbation to orgasm) or over-the-counter remedy desire [1]. For some individuals, a ‘flare’ of PGAD symptoms [2, 3]. The symptoms are described as distressing, unwanted and sometimes painful [2, 4, 5] and are associated with a Topical Collection on Female Sexual Dysfunction and Disorder significant negative impact on psychosocial and daily func- tioning [6, 7 ]. * Robyn A. Jackowich Given the similarities between PGAD and other forms of robyn.jackowich@queensu.ca genitopelvic discomfort (e.g. vulvodynia: chronic, idiopathic vulvar pain [8]), it has been suggested that PGAD may be best Caroline F. Pukall considered a type of ‘genitopelvic dysesthesia’ (defined as an caroline.pukall@queensu.ca abnormal, unpleasant sensation) where the primarily unwant- ed sensation is arousal [6, 8, 9� ]. This shift in conceptualiza- Department of Psychology, Queen’s University, 62 Arch Street, Kingston, ON K7L 3N6, Canada tion may help decrease stigma associated with the condition �� 128 Curr Sex Health Rep (2020) 12:127–135 (i.e. that genital arousal is always wanted or pleasurable [9� ]), [2], with 10% reporting lifelong symptoms. While research and it represents a more accurate description of PGAD— has primarily focused on women, there are a growing number specifically that PGAD is a disorder of unpleasant sensations, of case studies of men who experience similar symptoms not high/unwanted sexual desire or high subjective/cognitive [15–20]. arousal. As such, we will refer to the condition as Despite approaching two decades since PGAD/GD was PGAD/genitopelvic dysesthesia (PGAD/GD). More recent first described in the scientific literature, there remains limited and detailed diagnostic criteria for PGAD/GD, based on ex- research on, and awareness of, PGAD/GD. Research has pri- pert opinion, were proposed by ISSWSH in 2016 [10] and are marily been in the form of individual case studies (for a recent summarized alongside other proposed diagnostic criteria in review, see [3]), and no systematic treatment outcome studies Table 1. (i.e. controlled trials) have been published. PGAD/GD was There is limited information available about the prevalence only recently listed in a diagnostic manual—the 11th edition of PGAD/GD. One study found 1% of women attending a of the International Classification of Diseases [21]. This lack sexual health clinic in the UK reported all the Leiblum and of awareness and knowledge of PGAD/GD results in difficul- Nathan (2001) criteria for PGAD/GD, while 33.3% reported ty accessing treatment for these highly distressing symptoms. at least one of the criteria [13]. Another study surveying the Indeed, individuals with PGAD/GD frequently attribute a lack first-year undergraduate cohort in psychology at a Canadian of healthcare provider knowledge of PGAD/GD to be a barrier university (N = 1641) also found approximately 1% of male to receiving timely and effective care [22]. Overall, there is and female students reported experiencing all of the Leiblum need for more research on this distressing condition as well as and Nathan (2001) PGAD/GD criteria at a moderate frequen- a theoretical framework to guide and integrate research and cy or higher [14]. PGAD/GD affects individuals of all ages clinical efforts in the biological and psychosocial domains. Table 1 Proposed diagnostic criteria for persistent genital arousal disorder/genitopelvic dysesthesia (PGAD/GD). Common features of all criteria are noted in italics Citation Proposed PGAD/GD diagnostic criteria Leiblum and Nathan 1. Physiological responses characteristic of sexual arousal (genital and breast vasocongestion and sensitivity) persist for an (2001) [1] extended period of time (hourstodays) and do not subside completely on their own. 2. The signs of physiological arousal do not resolve with ordinary orgasmic experience and may require multiple orgasms over hours or days to remit. 3. These physiological signs of arousal are usually experienced as unrelated to any subjective sense of sexual excitement or desire. 4. The persistent sexual arousal may be triggered not only by a sexual activity but also by seemingly non-sexual stimuli or by no apparent stimulus at all. 5. The physiological signs of persistent arousal are experienced as unbidden, intrusive and unwanted. When feelings of genital arousal persist for days, week or even months, they are experienced as personally distressing and worrisome. Basson et al. (2004) [11] Spontaneous intrusive and unwanted genital arousal (e.g. tingling, throbbing, pulsating) in the absence of sexual interest and desire. Any awareness of subjective arousal is typically but not in variably unpleasant. The arousal is unrelieved by one or more orgasms and the feelings of arousal persist for hours or days. Parish et al. (2016) [10] 1. Characterized by persistent or recurrent, unwanted or intrusive, distressing feelings of genital arousal or being on the verge of orgasm (genital dysesthesia), not associated with concomitant sexual interest, thoughts or fantasies for > or equal to 6 months. Could be associated with: 2. Limited resolution, no resolution or aggravation of symptoms by sexual activity with or without aversive and/or compromised orgasm 3. Aggravation of genital symptoms by certain circumstances 4. Despair, emotional lability, catastrophizing and/or suicidality 5. Inconsistent evidence of genital arousal during symptoms Waldinger et al. (2018) 1. A state of unwanted restless genital sensations that are associated with [12] 2. restless legs, and/or 3. complaints of overactive bladder, and/or 4. urethral hypersensitivity, 5. and not resolving by sexual activity PGAD/GD persistent genital arousal disorder/genitopelvic dysesthesia These criteria are based on the most recent publication by Waldinger and Schweitzer (2018). Previous publications from Waldinger and colleagues include a different set of diagnostic criteria: the combination of PGAD (defined by Leiblum & Nathan, 2001) plus the presence of restless leg syndrome, bladder syndrome and/or urethral hypersensitivity Curr Sex Health Rep (2020) 12:127–135 129 Biological Factors in PGAD and cauda equina syndrome [24]. One study found that 66.7% (n = 12/18) of spinal MRIs collected from individuals Research on biological factors associated with in an online PGAD/GD support group contained a sacral PGAD/GD is beginning to increase, but overall remains Tarlov cyst [25]. Subsequently, Feigenbaum and Boone found relatively limited. This paucity of research leaves that removal of Tarlov cysts in individuals with PGAD/GD healthcare providers who treat PGAD/GD in a position symptoms resulted in improvement in 10/11 women [26]. where they must carefully consider and balance the lack More recently, a retrospective chart review found that 4 of of empirically informed treatments and the invasiveness 10 women presenting with PGAD/GD symptoms at a neurol- or cost of treatments to their patients/clients. ogy clinic had Tarlov cysts [27]. Komisaruk and Goldstein Multiple aetiologies have been hypothesised for the symp- also suggest that irritation (via a herniated intervertebral toms of PGAD/GD including vascular factors, central and disc) of the roots of the genital sensory nerves within the peripheral nervous system factors, pharmacological factors, cauda equina, called cauda equina syndrome, may lead to dietary and psychosocial factors (for a recent summary of case the development of PGAD/GD symptoms [24]which has studies examining causes and treatments of PGAD/GD, see been examined in a series of case studies as well [28]. [3])—suggesting that the aetiology of PGAD/GD may be Although these initial studies suggest spinal pathology multifactorial. In addition, like a diagnosis of vulvodynia, may be one potential aetiology in PGAD/GD, we still re- some cases of PGAD/GD may be idiopathic. Recent research quire systematic studies that include measurement of on biological factors in PGAD/GD has focused on pharmaco- symptomology (i.e. intensity, duration, quality of sensa- logical causes and interventions, peripheral sensory neuropa- tions, associated distress) and functioning (i.e. psychoso- thy and spinal pathology (e.g. cauda equina syndrome, Tarlov cial, sexual and relationship wellbeing) following treat- cysts). Each will be briefly summarized here. ment and at long-term follow-up. Pharmacological Causes and Interventions Small-Fibre Sensory Neuropathy A small number of case reports and self-report studies have observed that pharmacological agents (either initiation or Waldinger and colleagues have hypothesized that small-fibre withdrawal) can induce PGAD/GD symptoms—primarily sensory neuropathy of the pudendal nerve could lead to the seen with antidepressant medications [2, 23]. To date, no dysesthesias that characterize PGAD/GD [12]. In addition to randomised placebo-controlled clinical trials have been under- compression at the nerve root ( [24], see above), PGAD/GD taken to investigate pharmacological treatments for symptoms may result from compression or entrapment of PGAD/GD. Current knowledge of these treatments relies ex- more distal areas of the pudendal nerve, including the dorsal clusively on case reports. Kruger and colleagues provide a nerve of the clitoris [29]. Waldinger and colleagues named recent review of different drug classes (antiandrogens, antide- PGAD/GD ‘restless genital syndrome’ when it occurs with a pressants, anticonvulsants, partial agonist at nicotinic acetyl- common constellation of comorbidities, specifically restless choline receptor subtypes for smoking cessation, dopamine leg syndrome, overactive bladder syndrome and urethral hy- agonists, dopamine antagonists, opioids, toxins and cannabi- persensitivity [12]. While these authors recommend pelvic noids) that have been used to treat PGAD/GD symptoms and and/or lumbosacral MRIs to investigate for spinal or periph- the results of the corresponding case reports [23]. Only a small eral nerve involvement in PGAD/GD, so far, surgical inter- number of cases have examined each medication (i.e. less than ventions have only been evaluated in small samples, and larg- 10 cases per medication), with most finding some improve- er prospective clinical trials are needed [29, 30]. ment in PGAD/GD symptoms (improvement found in 25 of 30 cases summarized; [23]).However,Krugerand colleagues note that there is likely an underreporting of treatment failures in the current case report format Psychosocial Factors in PGAD/GD literature, which would also provide important informa- tion about the effectiveness of pharmacological treat- A small number of recent studies have examined the ments for PGAD/GD [23]. psychosocial wellbeing of individuals with PGAD/GD using validated self-report measures and control group Spinal Pathology comparisons. By definition, PGAD/GD is associated with distress—however, psychosocial factors may poten- Komisaruk and Goldstein recently reviewed two proposed tially increase one’s risk of developing PGAD/GD, me- aetiologies for PGAD/GD resulting from spinal pathology: diate PGAD/GD symptoms or may be negatively im- Tarlov cysts (i.e. fluid-filled sacs at the spinal nerve root) pacted by PGAD/GD. 130 Curr Sex Health Rep (2020) 12:127–135 Psychosocial Risk Factors Associated With PGAD/GD women with higher PGAD symptom severity and higher sex- Symptoms and Distress ual conservatism reported significantly greater distress then women with higher PGAD symptom severity and lower sex- Although psychosocial factors alone are unlikely to cause ual conservatism [35]. Of note, more research is needed to PGAD/GD, they may contribute to the development and understand the directionality among these findings, as this maintenance of the disorder and its associated distress. was a single-timepoint study. Research in this area is minimal, and studies are limited by their cross-sectional designs. Longitudinal and/or intervention Sexual Abuse Two studies have inquired about sexual abuse studies are needed to assess the relationships among psycho- histories in samples of women with PGAD/GD. Both found social factors, PGAD/GD symptoms and PGAD-related dis- high rates of childhood sexual abuse (46.7–52.6%; [4, 36]). tress. However, the associations described below may serve as Direct comparisons are challenging due to differences in the a starting point for our understanding of the ways in which wording of questions about past sexual abuse; however, the psychosocial factors contribute to PGAD/GD symptoms. rates of childhood sexual abuse reported by individuals with PGAD/GD appear to be higher than those reported by indi- Cognitive/Affective Factors In 2007, Leiblum and Chivers viduals with other forms of genitopelvic discomfort (e.g. proposed a psychological model to explain the development vulvodynia, chronic pelvic pain [37–39]). Although more re- of PGAD/GD symptoms [31� ]. They proposed that negative search is needed in larger samples of individuals with appraisals of spontaneous genital arousal, which is a norma- PGAD/GD, sexual abuse experiences may negatively influ- tive part of female sexuality, may lead to increased anxiety ence evaluations of arousal and/or emotional responses to and sympathetic nervous system activity. This in turn could normative or dysfunctional spontaneous genital arousal. increase genital arousal sensitization and narrow one’satten- Indeed, Carvalho and colleagues found that individuals with tion to these sensations, thus creating a feedback cycle of PGAD/GD report more negative thoughts, increased negative increased genital arousal sensations. Supporting this model, affect and decreased positive affect during sexual activity than other studies have found high rates of pre-existing difficulties women without PGAD/GD, which could result from a history with mood, anxiety and stress [7�� , 31� ]inwomen with of abuse and/or from the PGAD/GD symptoms themselves PGAD/GD symptoms. Indeed, many women with [40]. However, the association between sexual abuse and PGAD/GD self-report that stress (33.98%), anxiety PGAD, as well as the timing of these associations, has yet to (29.13%) and loss (13.59%) were the initial triggers of their be investigated. A history of sexual and physical abuse is a symptoms [32]. Jackowich and colleagues found risk factor for other forms of genitopelvic discomfort, such as catastrophizing of vulvar sensations (rumination about symp- vulvodynia [39] and other forms of chronic pelvic pain [41]. toms, magnification of symptoms, hopelessness) was signifi- cantly associated with depression, anxiety, sexual distress, Psychosocial Consequences of PGAD/GD symptom distress and symptom severity [7�� ]. These results highlight the relationship between biological Depression, Anxiety and Stress Three recent studies have and psychological factors. Cognitive and affective reactions noted that individuals with PGAD/GD report significantly toward symptoms (e.g. hypervigilance, catastrophizing) may greater mental health concerns than individuals without mediate the relationship between PGAD/GD symptoms and these symptoms [7�� , 40, 42]. Compared to individuals functional outcomes (e.g. symptom severity, distress, daily without PGAD/GD, an online sample of women with functioning). In addition, these cognitive and affective factors PGAD/GD reported significantly poorer psychosocial may be potential targets of treatment. Facelle and colleagues wellbeing on all subscales of the Brief Symptom have recommended mindfulness-based treatments for Inventory scale, except interpersonal sensitivity [40]. PGAD/GD symptoms, and one case study by Hiller and Another study found that among women with Hekster found a couples-based cognitive-behavioural therapy PGAD/GD, average scores on measures of depression, effective in reducing symptoms and improving the overall anxiety and stress fell above clinical cut-offs [42]. A third relationship [33, 34]. study found that women with PGAD/GD symptoms re- ported significantly greater depression and anxiety symp- Personality Factors Certain personality characteristics have toms than age-matched women without PGAD/GD symp- also been found to be associated with greater PGAD/GD toms [7�� ]. Over half (54%) of women reported some symptom distress. Carvalho and colleagues found that neurot- degree of suicidal ideation—much higher rates than are icism, low openness and sexual conservatism significantly seen in the general population (e.g. from 2.3 to 14.6% predicted distress associated with PGAD/GD symptoms across 5 countries; [43]) and in those with chronic pain [35]. Additionally, sexual conservatism moderated the rela- conditions (prevalence rate of approximately 20% [44]). It tionship between symptom severity and distress, such that is important to note that this research is primarily cross- Curr Sex Health Rep (2020) 12:127–135 131 sectional; therefore, we cannot determine the direction of of the conceptualization of and treatment approaches to causality. These findings highlight the importance of a PGAD/GD is the assumption that PGAD/GD is a purely bio- thorough clinical assessment of psychosocial wellbeing logical condition that can only be treated by medical/surgical with individuals experiencing PGAD/GD, so that appropri- means. Though this biomedical perspective may be supported ate support can be provided. by the existing literature, it is important to stress that the symptoms of PGAD/GD will have an impact on psychosocial Daily Functioning Likely contributing to the psychosocial dif- functioning. The psychosocial experience can, in turn, influ- ficulties associated with PGAD/GD, research has found ence the experience and maintenance of the symptoms; there- PGAD/GDinterfereswithmanyactivitiesofdaily living, in- fore, psychosocial factors can also be targeted for potential cluding social activities and work [6]. PGAD/GD symptoms symptom management. that are described as ‘painful’ additionally interfere with inter- The recognition of the shared importance of the biological mediate activities of daily living (e.g. driving, housework). In and psychosocial components of a condition is well represent- an online sample of women with PGAD/GD symptoms, the ed by the biopsychosocial perspective, a model often used in most commonly reported activities affected by PGAD/GD the chronic pain literature [46]. This model has been applied to symptoms included sitting (85.2%), concentrating (83.5%), vulvodynia [47], which has some shared characteristics with social activities (69.6%), work (67.8%), wearing tight fitting PGAD/GD [8]; for example, their common comorbidities and clothing (67.8%) and sleep (67.8%; [2]). features are consistent with central sensitization. As described above and consistent with this view, Pukall and colleagues Sexual and Relationship Functioning Like other forms of proposed that all forms of genitopelvic discomfort, including genitopelvic discomfort (e.g. vulvodynia, [39]), PGAD/GD vulvodynia and PGAD/GD, be conceptualized as genitopelvic also appears to have negative consequences for sexuality more dysesthesias [9� ]. They further suggested that each form of broadly. Women with PGAD/GD report fewer erotic genitopelvic dysesthesia be classified by cause of symptoms thoughts, greater negative affect and less positive affect during (known or idiopathic), much like the most recent classification sexual activity [40]. Squibb and colleagues found an associa- of chronic vulvar pain and vulvodynia [47]. Importantly, this tion between greater numbers of other medical conditions, approach allows for the future incorporation of ‘potential as- greater depression, greater anxiety and higher levels of sexual sociated factors’ (e.g. pelvic floor musculature, psychosocial distress in women with PGAD/GD [42]. Women with factors)—which are biopsychosocial in nature—to idiopathic PGAD/GD report poorer sexual functioning than women GP/PGAD, in the same manner as they have been applied to without PGAD/GD on all domains of the Female Sexual vulvodynia [47]. Although the research on PGAD/GD is not Function Index, except desire [45]. Many women with yet sufficiently broad or well developed to delineate what PGAD/GD also report avoiding sexual activity in order to biopsychosocial factors may be potentially associated with reduce symptoms [7�� ]. This is reflected in the high levels of PGAD/GD, it is essential to adopt, at the very least, a sexual distress seen in individuals with PGAD/GD across biopsychosocial perspective of PGAD/GD as early as possible multiple studies [7�� , 42]. Little is known about the impact in order to prevent dualistic theoretical and practical ap- that PGAD/GD has on relationships, though women with proaches to PGAD/GD treatment. PGAD/GD reported significantly lower relationship satisfac- A biopsychosocial approach to PGAD/GD is supported by tion than age-matched women without PGAD/GD symptoms two main lines of evidence. First, one’s interpretation of the [7�� ]. sensations of genital arousal can influence one’s distress level. For example, as suggested by Leiblum and Chivers (2007), a negative appraisal of arousal sensations may lead to distress Integrating Mind and Body: a Biopsychosocial associated with these sensations, whereas a positive interpreta- Framework tion may result in no distress to, or even a positive experience of, the sensations [31� ]. Indeed, there is evidence of individuals Most of the research to date on PGAD/GD has focused on who have persistent sensations of genital arousal but who are possible biological underpinnings of the condition and psy- not distressed by the sensations (e.g. [13]), suggesting that one’s chosocial outcomes of having the condition. The predominant reaction to the sensations (as determined by psychosocial influ- literature on intervention has focused on medication and phys- ences, for example) can play a significant role in the distress iological treatments, ranging from transcutaneous electrical experienced. The continuum of possible reactions to the symp- nerve stimulation to surgery (e.g. removal of Tarlov cysts; toms may be primarily influenced by psychosocial factors, [3]). Very little is known about long-term treatment outcomes thereby supporting the importance of an approach that accounts and what options are, in fact, effective even in the short term. for psychosocial factors. Related to this point is the emerging In addition, many cases of PGAD/GD have unknown evidence (summarized above) of cognitive and emotional me- diators of symptoms and distress (e.g. catastrophizing, aetiology. A potential downfall of the early literature in terms 132 Curr Sex Health Rep (2020) 12:127–135 hypervigilance), which again suggest that psychosocial factors specifies that the way pain is interpreted may lead to different play a role in the experience of the symptoms and may be outcomes. If individuals interpret pain as non-threatening and potential targets for treatment (e.g. cognitive skills to decrease maintain their activities, then recovery from pain is promoted. catastrophizing and hypervigilance). Second, the significant Alternatively, if one is hypervigilant to pain, catastrophizes, psychosocial impact of PGAD/GD symptoms on people’s lives fears and avoids pain, what may initially be adaptive in the (e.g. work, social activities), as demonstrated by several studies acute phase of pain may paradoxically lead to poorer out- (e.g. [6, 40]), cannot be ignored and will likely require attention comes and the development of chronic pain [48, 49]. The over and above symptom reduction. Approaches to PGAD/GD FA model has been previously applied to other forms of gen- that incorporate all elements of the biopsychosocial model will ital discomfort (e.g. vulvodynia, [50]) which, as described therefore allow for a more comprehensive conceptualization of above, share similarities with PGAD/GD. PGAD/GD, which will then likely lead to a multidisciplinary While the predictive validity of the FA model in assessment and treatment approach. The fear-avoidance model PGAD/GD has yet to be tested, research supports the discrete (FA model; [48, 49]) is one such approach that can prove useful variables within the model. Figure 1 presents the FA model, in PGAD/GD. modified for PGAD/GD. Evidence that supports each part of the model is cited beneath. For example, previous research has found high rates of PGAD/GD catastrophizing (e.g. ‘Ibecome Application of the Fear-Avoidance Model to PGAD/GD afraid that the sensations will get worse’)were positivelyas- sociated with symptom severity, distress and symptoms of An example of a model integrating both biological and psy- depression and anxiety [7�� ]. Application of the FA model to chological factors in our understanding of chronic pain is the PGAD/GD would provide information about psychosocial fear-avoidance model (FA model; [48, 49]). This model may factors that contribute to the development and maintenance also help guide future research and the development of clinical of this condition. interventions for PGAD/GD. The FA model of chronic pain PGAD/GD Aetiologies Many Different Aetiologies: Jackowich et al. 2016; Komisaruk & Goldstein, 2017 Disuse Depression Disability Anxiety with Autonomic Arousal Depression: Carvalho et al., 2015; Leiblum et al., 2007; Jackowich et al., 2020. Anxiety: Leiblum et al., 2007; Jackowich et al., 20 . 20 Disability: Jackowich et al., 2018 Recovery/Symptom Avoidance Reduction Hypervigilance Hypervigalence: Leiblum et al., 2007; Avoidance: Jackowich et al., 2018; Jackowich et al., 2020 Arousal Experience Confrontation Arousal-Related Fear Catastrophizing: Jackowich et al., 2020; Jackowich et al., 2018 Arousal Catastrophizing No Fear CBT & Mindfulness Recommende : d Facelle et al., 2013; Hiller & Hekster, Negative Affectivity Threatening Arousal/Illness Information Negative Affect During Sexual Activity: Carvalho et al., 2015 Fig. 1 The fear-avoidance model is presented here based on Vlaeyen and model are presented in grey. Dotted lines represent findings that have Linton (2000) [49] and modified theoretically for persistent genital found associations between variables within the model (e.g. association arousal disorder/genitopelvic dysesthesia (PGAD/GD) [2–8, 9� ]. The between catastrophizing and greater depression and anxiety symptoms: model is presented in white boxes with circular black arrows, while [6]) citations for past studies that have examined factors related to this Curr Sex Health Rep (2020) 12:127–135 133 Conclusions: Future Research and Clinical and wanted experience [9� ]. We may look to efforts in other Considerations domains of sexuality, and even genitopelvic pain such as the #Itsnotinyourhead campaign. Some initial efforts have been Research on PGAD/GD is beginning to emerge, but it is still in made in this regard (for example, the #PGADFacts campaign its infancy. To date, studies have examined biological and psy- from our team at the Queen’s University Sexual Health chosocial factors separately, failing to investigate or acknowl- Research Lab. See https://sexlab.ca/pgad for more edge the interrelationship between these components. In addition, information). Continued efforts to increase awareness of the majority of research on PGAD is in the form of case studies, PGAD/GD in the healthcare community and more broadly which are limited in their ability to assess the effectiveness of may facilitate increased awareness of this distressing treatments for PGAD. A literature predominantly consisting of condition. case studies may also underrepresent studies in which symptom improvement was not found. The chronic pain literature, which is Compliance with Ethical Standards more established, may provide a framework for guiding future Conflict of Interest Dr. Pukall reports grants from CIHR, grants from research and clinical efforts from a biopsychosocial perspective. ISSWSH, and grants from Queen’s University, during the conduct of the Longitudinal and/or experimental studies are needed to better study. understand and evaluate biopsychosocial models of Robyn Jackowich reports grants from ISSWSH, outside the submitted PGAD/GD—such as the fear-avoidance model. Recognizing work. the ways in which biopsychosocial factors influence one another Human and Animal Rights and Informed Consent This article does not may highlight potential areas for intervention and support. contain any studies with human or animal subjects performed by any of Clinical assessment of PGAD/GD should include care- the authors. ful consideration of contributing psychosocial factors and resulting psychosocial impairment. For example, individ- Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adap- uals with PGAD/GD should be carefully screened for sui- tation, distribution and reproduction in any medium or format, as long as cidal ideation given the high rates found in recent studies you give appropriate credit to the original author(s) and the source, pro- [7�� ] and these individuals should be connected with sup- vide a link to the Creative Commons licence, and indicate if changes were port as needed. made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a Current recommendations for interventions are multidisci- credit line to the material. If material is not included in the article's plinary, including pharmacotherapy, physiotherapy of the pel- Creative Commons licence and your intended use is not permitted by vic floor and psychotherapy [23, 51]. Given the limited em- statutory regulation or exceeds the permitted use, you will need to obtain pirical support for all treatment interventions, healthcare pro- permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. viders treating individuals with PGAD/GD must balance this limited empirical evidence with the invasive or costly nature of proposed treatments. Given the highly distressing nature of PGAD/GD, there is References great need for clinical trials evaluating the effectiveness of differ- ent medical, physiotherapy and psychosocial interventions. The development of a patient-reported outcome instrument could bet- Papers of particular interest, published recently, have been highlighted as: ter assess treatment outcomes and quality of life. As a starting point, we may look to the Initiative on Methods, Measurement � Of importance �� Of major importance and Pain Assessment in Clinical Trials (IMMPACT) guidelines, as has been done for vulvodynia [52� ]. Similar to the IMMPACT 1. Leiblum SR, Nathan SG. Persistent sexual arousal syndrome: a recommendations for vulvodynia, the inclusion of measures that newly discovered pattern of female sexuality. J Sex Marital Ther. target daily functioning, sexuality, psychosocial and relationship 2001;27(4):365–80. wellbeing would also be important for assessing outcomes in 2. Jackowich R, Pink L, Gordon A, Poirier É, Pukall CF. Symptom PGAD/GD. 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The fear-avoidance model of musculoskeletal pain: tional claims in published maps and institutional affiliations. current state of scientific evidence. J Behav Med. 2007;30(1):77– http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Current Sexual Health Reports Springer Journals

Persistent Genital Arousal Disorder: a Biopsychosocial Framework

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Springer Journals
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Copyright © The Author(s) 2020
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1548-3584
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1548-3592
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10.1007/s11930-020-00268-2
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Abstract

Purpose of Review Persistent genital arousal disorder (also referred to as genitopelvic dysesthesia or PGAD/GD) is a distressing and largely underrecognized condition characterized by persistent, unwanted genital arousal (sensations, sensitivity, vasocongestion) in the absence of subjective/cognitive arousal and sexual desire. The purpose of this review is to summarize recent findings on biological and psychosocial factors in PGAD/GD as they pertain to the assessment and treatment of this condition. These findings will be considered within a biopsychosocial framework, for the purposes of considering next steps for clinical and research efforts. Recent Findings A small number of studies have recently examined potential biological aetiologies for PGAD/GD: pharmaco- logical agents, spinal pathology and peripheral nerve involvement. Recent studies have also found that PGAD/GD is associated with a significant negative impact on psychosocial wellbeing and daily functioning as compared to symptom-free individuals. In addition, these results highlight cognitive/affective responses to symptoms (e.g. catastrophizing of symptoms) that may influence outcomes. However, biological and psychological research are rarely integrated in these studies, despite the interrelationship between these factors. Summary Although PGAD/GD was first described in the scientific literature almost two decades ago, most research on PGAD/GD is presented in the form of case studies. Prospective treatment trials that integrate biopsychosocial factors are needed in order to provide effective and efficient care to this population. This research would be facilitated by the development of a patient-reported outcome measure, as well as greater education/awareness among healthcare providers and the public about this distressing condition. . . . . . Keywords Persistent genital arousal disorder Genitopelvic dysesthesia PGAD/GD Biopsychosocial Catastrophizing Fear avoidance Introduction may last for hours to days, while for others, these symptoms may be continuously present [2]. Symptoms may be triggered First described by Sandra Leiblum and Sharon Nathan in by sexual cues, non-sexual cues or there may be no identifi- 2001, persistent genital arousal disorder (or PGAD) is a highly able trigger [2]. A subset of individuals with PGAD also re- distressing condition, characterized by genital arousal (e.g. port experiencing distressing and unwanted spontaneous or- genital sensations, sensitivity and/or vasocongestion) that oc- gasms [2]. PGAD symptoms are not relieved by any behav- curs in the absence of subjective/cognitive arousal or sexual iour (e.g. masturbation to orgasm) or over-the-counter remedy desire [1]. For some individuals, a ‘flare’ of PGAD symptoms [2, 3]. The symptoms are described as distressing, unwanted and sometimes painful [2, 4, 5] and are associated with a Topical Collection on Female Sexual Dysfunction and Disorder significant negative impact on psychosocial and daily func- tioning [6, 7 ]. * Robyn A. Jackowich Given the similarities between PGAD and other forms of robyn.jackowich@queensu.ca genitopelvic discomfort (e.g. vulvodynia: chronic, idiopathic vulvar pain [8]), it has been suggested that PGAD may be best Caroline F. Pukall considered a type of ‘genitopelvic dysesthesia’ (defined as an caroline.pukall@queensu.ca abnormal, unpleasant sensation) where the primarily unwant- ed sensation is arousal [6, 8, 9� ]. This shift in conceptualiza- Department of Psychology, Queen’s University, 62 Arch Street, Kingston, ON K7L 3N6, Canada tion may help decrease stigma associated with the condition �� 128 Curr Sex Health Rep (2020) 12:127–135 (i.e. that genital arousal is always wanted or pleasurable [9� ]), [2], with 10% reporting lifelong symptoms. While research and it represents a more accurate description of PGAD— has primarily focused on women, there are a growing number specifically that PGAD is a disorder of unpleasant sensations, of case studies of men who experience similar symptoms not high/unwanted sexual desire or high subjective/cognitive [15–20]. arousal. As such, we will refer to the condition as Despite approaching two decades since PGAD/GD was PGAD/genitopelvic dysesthesia (PGAD/GD). More recent first described in the scientific literature, there remains limited and detailed diagnostic criteria for PGAD/GD, based on ex- research on, and awareness of, PGAD/GD. Research has pri- pert opinion, were proposed by ISSWSH in 2016 [10] and are marily been in the form of individual case studies (for a recent summarized alongside other proposed diagnostic criteria in review, see [3]), and no systematic treatment outcome studies Table 1. (i.e. controlled trials) have been published. PGAD/GD was There is limited information available about the prevalence only recently listed in a diagnostic manual—the 11th edition of PGAD/GD. One study found 1% of women attending a of the International Classification of Diseases [21]. This lack sexual health clinic in the UK reported all the Leiblum and of awareness and knowledge of PGAD/GD results in difficul- Nathan (2001) criteria for PGAD/GD, while 33.3% reported ty accessing treatment for these highly distressing symptoms. at least one of the criteria [13]. Another study surveying the Indeed, individuals with PGAD/GD frequently attribute a lack first-year undergraduate cohort in psychology at a Canadian of healthcare provider knowledge of PGAD/GD to be a barrier university (N = 1641) also found approximately 1% of male to receiving timely and effective care [22]. Overall, there is and female students reported experiencing all of the Leiblum need for more research on this distressing condition as well as and Nathan (2001) PGAD/GD criteria at a moderate frequen- a theoretical framework to guide and integrate research and cy or higher [14]. PGAD/GD affects individuals of all ages clinical efforts in the biological and psychosocial domains. Table 1 Proposed diagnostic criteria for persistent genital arousal disorder/genitopelvic dysesthesia (PGAD/GD). Common features of all criteria are noted in italics Citation Proposed PGAD/GD diagnostic criteria Leiblum and Nathan 1. Physiological responses characteristic of sexual arousal (genital and breast vasocongestion and sensitivity) persist for an (2001) [1] extended period of time (hourstodays) and do not subside completely on their own. 2. The signs of physiological arousal do not resolve with ordinary orgasmic experience and may require multiple orgasms over hours or days to remit. 3. These physiological signs of arousal are usually experienced as unrelated to any subjective sense of sexual excitement or desire. 4. The persistent sexual arousal may be triggered not only by a sexual activity but also by seemingly non-sexual stimuli or by no apparent stimulus at all. 5. The physiological signs of persistent arousal are experienced as unbidden, intrusive and unwanted. When feelings of genital arousal persist for days, week or even months, they are experienced as personally distressing and worrisome. Basson et al. (2004) [11] Spontaneous intrusive and unwanted genital arousal (e.g. tingling, throbbing, pulsating) in the absence of sexual interest and desire. Any awareness of subjective arousal is typically but not in variably unpleasant. The arousal is unrelieved by one or more orgasms and the feelings of arousal persist for hours or days. Parish et al. (2016) [10] 1. Characterized by persistent or recurrent, unwanted or intrusive, distressing feelings of genital arousal or being on the verge of orgasm (genital dysesthesia), not associated with concomitant sexual interest, thoughts or fantasies for > or equal to 6 months. Could be associated with: 2. Limited resolution, no resolution or aggravation of symptoms by sexual activity with or without aversive and/or compromised orgasm 3. Aggravation of genital symptoms by certain circumstances 4. Despair, emotional lability, catastrophizing and/or suicidality 5. Inconsistent evidence of genital arousal during symptoms Waldinger et al. (2018) 1. A state of unwanted restless genital sensations that are associated with [12] 2. restless legs, and/or 3. complaints of overactive bladder, and/or 4. urethral hypersensitivity, 5. and not resolving by sexual activity PGAD/GD persistent genital arousal disorder/genitopelvic dysesthesia These criteria are based on the most recent publication by Waldinger and Schweitzer (2018). Previous publications from Waldinger and colleagues include a different set of diagnostic criteria: the combination of PGAD (defined by Leiblum & Nathan, 2001) plus the presence of restless leg syndrome, bladder syndrome and/or urethral hypersensitivity Curr Sex Health Rep (2020) 12:127–135 129 Biological Factors in PGAD and cauda equina syndrome [24]. One study found that 66.7% (n = 12/18) of spinal MRIs collected from individuals Research on biological factors associated with in an online PGAD/GD support group contained a sacral PGAD/GD is beginning to increase, but overall remains Tarlov cyst [25]. Subsequently, Feigenbaum and Boone found relatively limited. This paucity of research leaves that removal of Tarlov cysts in individuals with PGAD/GD healthcare providers who treat PGAD/GD in a position symptoms resulted in improvement in 10/11 women [26]. where they must carefully consider and balance the lack More recently, a retrospective chart review found that 4 of of empirically informed treatments and the invasiveness 10 women presenting with PGAD/GD symptoms at a neurol- or cost of treatments to their patients/clients. ogy clinic had Tarlov cysts [27]. Komisaruk and Goldstein Multiple aetiologies have been hypothesised for the symp- also suggest that irritation (via a herniated intervertebral toms of PGAD/GD including vascular factors, central and disc) of the roots of the genital sensory nerves within the peripheral nervous system factors, pharmacological factors, cauda equina, called cauda equina syndrome, may lead to dietary and psychosocial factors (for a recent summary of case the development of PGAD/GD symptoms [24]which has studies examining causes and treatments of PGAD/GD, see been examined in a series of case studies as well [28]. [3])—suggesting that the aetiology of PGAD/GD may be Although these initial studies suggest spinal pathology multifactorial. In addition, like a diagnosis of vulvodynia, may be one potential aetiology in PGAD/GD, we still re- some cases of PGAD/GD may be idiopathic. Recent research quire systematic studies that include measurement of on biological factors in PGAD/GD has focused on pharmaco- symptomology (i.e. intensity, duration, quality of sensa- logical causes and interventions, peripheral sensory neuropa- tions, associated distress) and functioning (i.e. psychoso- thy and spinal pathology (e.g. cauda equina syndrome, Tarlov cial, sexual and relationship wellbeing) following treat- cysts). Each will be briefly summarized here. ment and at long-term follow-up. Pharmacological Causes and Interventions Small-Fibre Sensory Neuropathy A small number of case reports and self-report studies have observed that pharmacological agents (either initiation or Waldinger and colleagues have hypothesized that small-fibre withdrawal) can induce PGAD/GD symptoms—primarily sensory neuropathy of the pudendal nerve could lead to the seen with antidepressant medications [2, 23]. To date, no dysesthesias that characterize PGAD/GD [12]. In addition to randomised placebo-controlled clinical trials have been under- compression at the nerve root ( [24], see above), PGAD/GD taken to investigate pharmacological treatments for symptoms may result from compression or entrapment of PGAD/GD. Current knowledge of these treatments relies ex- more distal areas of the pudendal nerve, including the dorsal clusively on case reports. Kruger and colleagues provide a nerve of the clitoris [29]. Waldinger and colleagues named recent review of different drug classes (antiandrogens, antide- PGAD/GD ‘restless genital syndrome’ when it occurs with a pressants, anticonvulsants, partial agonist at nicotinic acetyl- common constellation of comorbidities, specifically restless choline receptor subtypes for smoking cessation, dopamine leg syndrome, overactive bladder syndrome and urethral hy- agonists, dopamine antagonists, opioids, toxins and cannabi- persensitivity [12]. While these authors recommend pelvic noids) that have been used to treat PGAD/GD symptoms and and/or lumbosacral MRIs to investigate for spinal or periph- the results of the corresponding case reports [23]. Only a small eral nerve involvement in PGAD/GD, so far, surgical inter- number of cases have examined each medication (i.e. less than ventions have only been evaluated in small samples, and larg- 10 cases per medication), with most finding some improve- er prospective clinical trials are needed [29, 30]. ment in PGAD/GD symptoms (improvement found in 25 of 30 cases summarized; [23]).However,Krugerand colleagues note that there is likely an underreporting of treatment failures in the current case report format Psychosocial Factors in PGAD/GD literature, which would also provide important informa- tion about the effectiveness of pharmacological treat- A small number of recent studies have examined the ments for PGAD/GD [23]. psychosocial wellbeing of individuals with PGAD/GD using validated self-report measures and control group Spinal Pathology comparisons. By definition, PGAD/GD is associated with distress—however, psychosocial factors may poten- Komisaruk and Goldstein recently reviewed two proposed tially increase one’s risk of developing PGAD/GD, me- aetiologies for PGAD/GD resulting from spinal pathology: diate PGAD/GD symptoms or may be negatively im- Tarlov cysts (i.e. fluid-filled sacs at the spinal nerve root) pacted by PGAD/GD. 130 Curr Sex Health Rep (2020) 12:127–135 Psychosocial Risk Factors Associated With PGAD/GD women with higher PGAD symptom severity and higher sex- Symptoms and Distress ual conservatism reported significantly greater distress then women with higher PGAD symptom severity and lower sex- Although psychosocial factors alone are unlikely to cause ual conservatism [35]. Of note, more research is needed to PGAD/GD, they may contribute to the development and understand the directionality among these findings, as this maintenance of the disorder and its associated distress. was a single-timepoint study. Research in this area is minimal, and studies are limited by their cross-sectional designs. Longitudinal and/or intervention Sexual Abuse Two studies have inquired about sexual abuse studies are needed to assess the relationships among psycho- histories in samples of women with PGAD/GD. Both found social factors, PGAD/GD symptoms and PGAD-related dis- high rates of childhood sexual abuse (46.7–52.6%; [4, 36]). tress. However, the associations described below may serve as Direct comparisons are challenging due to differences in the a starting point for our understanding of the ways in which wording of questions about past sexual abuse; however, the psychosocial factors contribute to PGAD/GD symptoms. rates of childhood sexual abuse reported by individuals with PGAD/GD appear to be higher than those reported by indi- Cognitive/Affective Factors In 2007, Leiblum and Chivers viduals with other forms of genitopelvic discomfort (e.g. proposed a psychological model to explain the development vulvodynia, chronic pelvic pain [37–39]). Although more re- of PGAD/GD symptoms [31� ]. They proposed that negative search is needed in larger samples of individuals with appraisals of spontaneous genital arousal, which is a norma- PGAD/GD, sexual abuse experiences may negatively influ- tive part of female sexuality, may lead to increased anxiety ence evaluations of arousal and/or emotional responses to and sympathetic nervous system activity. This in turn could normative or dysfunctional spontaneous genital arousal. increase genital arousal sensitization and narrow one’satten- Indeed, Carvalho and colleagues found that individuals with tion to these sensations, thus creating a feedback cycle of PGAD/GD report more negative thoughts, increased negative increased genital arousal sensations. Supporting this model, affect and decreased positive affect during sexual activity than other studies have found high rates of pre-existing difficulties women without PGAD/GD, which could result from a history with mood, anxiety and stress [7�� , 31� ]inwomen with of abuse and/or from the PGAD/GD symptoms themselves PGAD/GD symptoms. Indeed, many women with [40]. However, the association between sexual abuse and PGAD/GD self-report that stress (33.98%), anxiety PGAD, as well as the timing of these associations, has yet to (29.13%) and loss (13.59%) were the initial triggers of their be investigated. A history of sexual and physical abuse is a symptoms [32]. Jackowich and colleagues found risk factor for other forms of genitopelvic discomfort, such as catastrophizing of vulvar sensations (rumination about symp- vulvodynia [39] and other forms of chronic pelvic pain [41]. toms, magnification of symptoms, hopelessness) was signifi- cantly associated with depression, anxiety, sexual distress, Psychosocial Consequences of PGAD/GD symptom distress and symptom severity [7�� ]. These results highlight the relationship between biological Depression, Anxiety and Stress Three recent studies have and psychological factors. Cognitive and affective reactions noted that individuals with PGAD/GD report significantly toward symptoms (e.g. hypervigilance, catastrophizing) may greater mental health concerns than individuals without mediate the relationship between PGAD/GD symptoms and these symptoms [7�� , 40, 42]. Compared to individuals functional outcomes (e.g. symptom severity, distress, daily without PGAD/GD, an online sample of women with functioning). In addition, these cognitive and affective factors PGAD/GD reported significantly poorer psychosocial may be potential targets of treatment. Facelle and colleagues wellbeing on all subscales of the Brief Symptom have recommended mindfulness-based treatments for Inventory scale, except interpersonal sensitivity [40]. PGAD/GD symptoms, and one case study by Hiller and Another study found that among women with Hekster found a couples-based cognitive-behavioural therapy PGAD/GD, average scores on measures of depression, effective in reducing symptoms and improving the overall anxiety and stress fell above clinical cut-offs [42]. A third relationship [33, 34]. study found that women with PGAD/GD symptoms re- ported significantly greater depression and anxiety symp- Personality Factors Certain personality characteristics have toms than age-matched women without PGAD/GD symp- also been found to be associated with greater PGAD/GD toms [7�� ]. Over half (54%) of women reported some symptom distress. Carvalho and colleagues found that neurot- degree of suicidal ideation—much higher rates than are icism, low openness and sexual conservatism significantly seen in the general population (e.g. from 2.3 to 14.6% predicted distress associated with PGAD/GD symptoms across 5 countries; [43]) and in those with chronic pain [35]. Additionally, sexual conservatism moderated the rela- conditions (prevalence rate of approximately 20% [44]). It tionship between symptom severity and distress, such that is important to note that this research is primarily cross- Curr Sex Health Rep (2020) 12:127–135 131 sectional; therefore, we cannot determine the direction of of the conceptualization of and treatment approaches to causality. These findings highlight the importance of a PGAD/GD is the assumption that PGAD/GD is a purely bio- thorough clinical assessment of psychosocial wellbeing logical condition that can only be treated by medical/surgical with individuals experiencing PGAD/GD, so that appropri- means. Though this biomedical perspective may be supported ate support can be provided. by the existing literature, it is important to stress that the symptoms of PGAD/GD will have an impact on psychosocial Daily Functioning Likely contributing to the psychosocial dif- functioning. The psychosocial experience can, in turn, influ- ficulties associated with PGAD/GD, research has found ence the experience and maintenance of the symptoms; there- PGAD/GDinterfereswithmanyactivitiesofdaily living, in- fore, psychosocial factors can also be targeted for potential cluding social activities and work [6]. PGAD/GD symptoms symptom management. that are described as ‘painful’ additionally interfere with inter- The recognition of the shared importance of the biological mediate activities of daily living (e.g. driving, housework). In and psychosocial components of a condition is well represent- an online sample of women with PGAD/GD symptoms, the ed by the biopsychosocial perspective, a model often used in most commonly reported activities affected by PGAD/GD the chronic pain literature [46]. This model has been applied to symptoms included sitting (85.2%), concentrating (83.5%), vulvodynia [47], which has some shared characteristics with social activities (69.6%), work (67.8%), wearing tight fitting PGAD/GD [8]; for example, their common comorbidities and clothing (67.8%) and sleep (67.8%; [2]). features are consistent with central sensitization. As described above and consistent with this view, Pukall and colleagues Sexual and Relationship Functioning Like other forms of proposed that all forms of genitopelvic discomfort, including genitopelvic discomfort (e.g. vulvodynia, [39]), PGAD/GD vulvodynia and PGAD/GD, be conceptualized as genitopelvic also appears to have negative consequences for sexuality more dysesthesias [9� ]. They further suggested that each form of broadly. Women with PGAD/GD report fewer erotic genitopelvic dysesthesia be classified by cause of symptoms thoughts, greater negative affect and less positive affect during (known or idiopathic), much like the most recent classification sexual activity [40]. Squibb and colleagues found an associa- of chronic vulvar pain and vulvodynia [47]. Importantly, this tion between greater numbers of other medical conditions, approach allows for the future incorporation of ‘potential as- greater depression, greater anxiety and higher levels of sexual sociated factors’ (e.g. pelvic floor musculature, psychosocial distress in women with PGAD/GD [42]. Women with factors)—which are biopsychosocial in nature—to idiopathic PGAD/GD report poorer sexual functioning than women GP/PGAD, in the same manner as they have been applied to without PGAD/GD on all domains of the Female Sexual vulvodynia [47]. Although the research on PGAD/GD is not Function Index, except desire [45]. Many women with yet sufficiently broad or well developed to delineate what PGAD/GD also report avoiding sexual activity in order to biopsychosocial factors may be potentially associated with reduce symptoms [7�� ]. This is reflected in the high levels of PGAD/GD, it is essential to adopt, at the very least, a sexual distress seen in individuals with PGAD/GD across biopsychosocial perspective of PGAD/GD as early as possible multiple studies [7�� , 42]. Little is known about the impact in order to prevent dualistic theoretical and practical ap- that PGAD/GD has on relationships, though women with proaches to PGAD/GD treatment. PGAD/GD reported significantly lower relationship satisfac- A biopsychosocial approach to PGAD/GD is supported by tion than age-matched women without PGAD/GD symptoms two main lines of evidence. First, one’s interpretation of the [7�� ]. sensations of genital arousal can influence one’s distress level. For example, as suggested by Leiblum and Chivers (2007), a negative appraisal of arousal sensations may lead to distress Integrating Mind and Body: a Biopsychosocial associated with these sensations, whereas a positive interpreta- Framework tion may result in no distress to, or even a positive experience of, the sensations [31� ]. Indeed, there is evidence of individuals Most of the research to date on PGAD/GD has focused on who have persistent sensations of genital arousal but who are possible biological underpinnings of the condition and psy- not distressed by the sensations (e.g. [13]), suggesting that one’s chosocial outcomes of having the condition. The predominant reaction to the sensations (as determined by psychosocial influ- literature on intervention has focused on medication and phys- ences, for example) can play a significant role in the distress iological treatments, ranging from transcutaneous electrical experienced. The continuum of possible reactions to the symp- nerve stimulation to surgery (e.g. removal of Tarlov cysts; toms may be primarily influenced by psychosocial factors, [3]). Very little is known about long-term treatment outcomes thereby supporting the importance of an approach that accounts and what options are, in fact, effective even in the short term. for psychosocial factors. Related to this point is the emerging In addition, many cases of PGAD/GD have unknown evidence (summarized above) of cognitive and emotional me- diators of symptoms and distress (e.g. catastrophizing, aetiology. A potential downfall of the early literature in terms 132 Curr Sex Health Rep (2020) 12:127–135 hypervigilance), which again suggest that psychosocial factors specifies that the way pain is interpreted may lead to different play a role in the experience of the symptoms and may be outcomes. If individuals interpret pain as non-threatening and potential targets for treatment (e.g. cognitive skills to decrease maintain their activities, then recovery from pain is promoted. catastrophizing and hypervigilance). Second, the significant Alternatively, if one is hypervigilant to pain, catastrophizes, psychosocial impact of PGAD/GD symptoms on people’s lives fears and avoids pain, what may initially be adaptive in the (e.g. work, social activities), as demonstrated by several studies acute phase of pain may paradoxically lead to poorer out- (e.g. [6, 40]), cannot be ignored and will likely require attention comes and the development of chronic pain [48, 49]. The over and above symptom reduction. Approaches to PGAD/GD FA model has been previously applied to other forms of gen- that incorporate all elements of the biopsychosocial model will ital discomfort (e.g. vulvodynia, [50]) which, as described therefore allow for a more comprehensive conceptualization of above, share similarities with PGAD/GD. PGAD/GD, which will then likely lead to a multidisciplinary While the predictive validity of the FA model in assessment and treatment approach. The fear-avoidance model PGAD/GD has yet to be tested, research supports the discrete (FA model; [48, 49]) is one such approach that can prove useful variables within the model. Figure 1 presents the FA model, in PGAD/GD. modified for PGAD/GD. Evidence that supports each part of the model is cited beneath. For example, previous research has found high rates of PGAD/GD catastrophizing (e.g. ‘Ibecome Application of the Fear-Avoidance Model to PGAD/GD afraid that the sensations will get worse’)were positivelyas- sociated with symptom severity, distress and symptoms of An example of a model integrating both biological and psy- depression and anxiety [7�� ]. Application of the FA model to chological factors in our understanding of chronic pain is the PGAD/GD would provide information about psychosocial fear-avoidance model (FA model; [48, 49]). This model may factors that contribute to the development and maintenance also help guide future research and the development of clinical of this condition. interventions for PGAD/GD. The FA model of chronic pain PGAD/GD Aetiologies Many Different Aetiologies: Jackowich et al. 2016; Komisaruk & Goldstein, 2017 Disuse Depression Disability Anxiety with Autonomic Arousal Depression: Carvalho et al., 2015; Leiblum et al., 2007; Jackowich et al., 2020. Anxiety: Leiblum et al., 2007; Jackowich et al., 20 . 20 Disability: Jackowich et al., 2018 Recovery/Symptom Avoidance Reduction Hypervigilance Hypervigalence: Leiblum et al., 2007; Avoidance: Jackowich et al., 2018; Jackowich et al., 2020 Arousal Experience Confrontation Arousal-Related Fear Catastrophizing: Jackowich et al., 2020; Jackowich et al., 2018 Arousal Catastrophizing No Fear CBT & Mindfulness Recommende : d Facelle et al., 2013; Hiller & Hekster, Negative Affectivity Threatening Arousal/Illness Information Negative Affect During Sexual Activity: Carvalho et al., 2015 Fig. 1 The fear-avoidance model is presented here based on Vlaeyen and model are presented in grey. Dotted lines represent findings that have Linton (2000) [49] and modified theoretically for persistent genital found associations between variables within the model (e.g. association arousal disorder/genitopelvic dysesthesia (PGAD/GD) [2–8, 9� ]. The between catastrophizing and greater depression and anxiety symptoms: model is presented in white boxes with circular black arrows, while [6]) citations for past studies that have examined factors related to this Curr Sex Health Rep (2020) 12:127–135 133 Conclusions: Future Research and Clinical and wanted experience [9� ]. We may look to efforts in other Considerations domains of sexuality, and even genitopelvic pain such as the #Itsnotinyourhead campaign. Some initial efforts have been Research on PGAD/GD is beginning to emerge, but it is still in made in this regard (for example, the #PGADFacts campaign its infancy. To date, studies have examined biological and psy- from our team at the Queen’s University Sexual Health chosocial factors separately, failing to investigate or acknowl- Research Lab. See https://sexlab.ca/pgad for more edge the interrelationship between these components. In addition, information). Continued efforts to increase awareness of the majority of research on PGAD is in the form of case studies, PGAD/GD in the healthcare community and more broadly which are limited in their ability to assess the effectiveness of may facilitate increased awareness of this distressing treatments for PGAD. A literature predominantly consisting of condition. case studies may also underrepresent studies in which symptom improvement was not found. The chronic pain literature, which is Compliance with Ethical Standards more established, may provide a framework for guiding future Conflict of Interest Dr. Pukall reports grants from CIHR, grants from research and clinical efforts from a biopsychosocial perspective. ISSWSH, and grants from Queen’s University, during the conduct of the Longitudinal and/or experimental studies are needed to better study. understand and evaluate biopsychosocial models of Robyn Jackowich reports grants from ISSWSH, outside the submitted PGAD/GD—such as the fear-avoidance model. Recognizing work. the ways in which biopsychosocial factors influence one another Human and Animal Rights and Informed Consent This article does not may highlight potential areas for intervention and support. contain any studies with human or animal subjects performed by any of Clinical assessment of PGAD/GD should include care- the authors. ful consideration of contributing psychosocial factors and resulting psychosocial impairment. For example, individ- Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adap- uals with PGAD/GD should be carefully screened for sui- tation, distribution and reproduction in any medium or format, as long as cidal ideation given the high rates found in recent studies you give appropriate credit to the original author(s) and the source, pro- [7�� ] and these individuals should be connected with sup- vide a link to the Creative Commons licence, and indicate if changes were port as needed. made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a Current recommendations for interventions are multidisci- credit line to the material. If material is not included in the article's plinary, including pharmacotherapy, physiotherapy of the pel- Creative Commons licence and your intended use is not permitted by vic floor and psychotherapy [23, 51]. Given the limited em- statutory regulation or exceeds the permitted use, you will need to obtain pirical support for all treatment interventions, healthcare pro- permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. viders treating individuals with PGAD/GD must balance this limited empirical evidence with the invasive or costly nature of proposed treatments. Given the highly distressing nature of PGAD/GD, there is References great need for clinical trials evaluating the effectiveness of differ- ent medical, physiotherapy and psychosocial interventions. The development of a patient-reported outcome instrument could bet- Papers of particular interest, published recently, have been highlighted as: ter assess treatment outcomes and quality of life. As a starting point, we may look to the Initiative on Methods, Measurement � Of importance �� Of major importance and Pain Assessment in Clinical Trials (IMMPACT) guidelines, as has been done for vulvodynia [52� ]. Similar to the IMMPACT 1. Leiblum SR, Nathan SG. Persistent sexual arousal syndrome: a recommendations for vulvodynia, the inclusion of measures that newly discovered pattern of female sexuality. J Sex Marital Ther. target daily functioning, sexuality, psychosocial and relationship 2001;27(4):365–80. wellbeing would also be important for assessing outcomes in 2. Jackowich R, Pink L, Gordon A, Poirier É, Pukall CF. Symptom PGAD/GD. 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Published: Sep 6, 2020

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