Persistent Genital Arousal in Women With Pelvic and Genital ..., Study notes of History

Download Persistent Genital Arousal in Women With Pelvic and Genital ... and more Study notes History in PDF only on Docsity! Persistent Genital Arousal in Women With Pelvic and Genital Pain 324 l APRIL JOGC AVRIL 2014 WOMEN’S HEALTH Key Words: Persistent genital arousal disorder, unprovoked genital arousal, spontaneous genital arousal, restless genital syndrome Competing Interests: None delcared. Received on August 22, 2013 Accepted on December 11, 2013 Persistent Genital Arousal in Women With Pelvic and Genital Pain leah Pink, RN, MN,1 Valérie Rancourt, MD, FRCPC,2 Allan Gordon, MD, FRCPC1 1 Wasser Pain Management Centre, Toronto ON 2 Institut universitaire de cardiologie et de pneumologie de Québec, Quebec City QC J Obstet Gynaecol Can 2014;36(4):324–330 Abstract Objective: Persistent genital arousal disorder (PGAD) has been identified as a condition of often unprovoked genital arousal associated with a significant level of distress. PGAD is not well understood, and no definitive cause has been determined. The aim of this study was to gain a better understanding of the disorder and to seek commonalities between cases of PGAD encountered in a chronic pain management clinic. Method: We reviewed a cohort of 15 women with PGAD who presented to a chronic non-cancer pain clinic in a large urban tertiary teaching hospital that provides pelvic and genital pain management. We conducted a series of interviews to examine medical history, history of presenting illness, and management. Descriptive statistics were used to examine the data. Results: Findings were largely consistent with previous research on PGAD regarding symptomatology and aggravating and alleviating factors. Symptoms of genital pain, depression, and interstitial cystitis were found in over one half of the patients in this cohort. Previous antidepressant use, restless legs syndrome, and pudendal neuralgia were found in a number of cases. Pelvic varices and Tarlov cysts have been previously identified as possible contributors to PGAD, but these were not a common finding in our cohort. Conclusion: Further research is needed to build on the current understanding of PGAD. Patients should be asked about persistent arousal as part of a sexual and reproductive history, especially in the case of common comorbidities. Résumé Objectif : Le syndrome d’excitation génitale persistante (SEGP) a été identifié comme étant une excitation génitale souvent non provoquée qui est associée à un degré de détresse considérable. Le SEGP n’est pas bien compris et aucune cause définitive ne lui a été attribuée. Cette étude avait pour but d’améliorer la compréhension de ce syndrome et de tenter d’établir des points communs entre les cas de SEGP constatés au sein d’une clinique de maîtrise de la douleur chronique. Méthode : Nous avons analysé une cohorte de 15 femmes aux prises avec le SEGP qui ont consulté une clinique de maîtrise de la douleur chronique n’étant pas attribuable au cancer, au sein d’un hôpital universitaire tertiaire urbain de grande envergure qui offre des services de maîtrise de la douleur pelvienne et génitale. Nous avons mené une série d’entrevues visant à examiner les antécédents médicaux, l’historique de la maladie en question et la prise en charge. Nous avons eu recours aux principes de la statistique descriptive pour examiner les données. Résultats : Nos constatations se sont en grande partie inscrites dans la suite logique des résultats obtenus par les efforts de recherche précédents ayant porté sur le SEGP, en ce qui a trait à la symptomatologie et aux facteurs aggravants et atténuants. Des symptômes de douleur génitale, de dépression et de cystite interstitielle ont été constatés chez plus de la moitié des patientes de cette cohorte. Des antécédents en matière d’utilisation d’antidépresseurs, de syndrome des jambes sans repos et de névralgie pudendale ont été constatés dans un certain nombre de cas. Des varices pelviennes et des kystes de Tarlov ont déjà été identifiés comme étant de possibles facteurs contribuant au SEGP; toutefois, leur présence n’a pas été fréquemment signalée au sein de notre cohorte. Conclusion : La tenue d’autres recherches s’avère requise pour enrichir notre compréhension actuelle du SEGP. Dans le cadre de l’anamnèse sexuelle et génésique, des questions portant sur l’excitation persistante devraient être posées aux patientes, particulièrement en présence de comorbidités courantes. APRIL JOGC AVRIL 2014 l 325 Persistent Genital Arousal in Women With Pelvic and Genital Pain INTRODUCTION Persistent genital arousal disorder has been identified as an unprovoked genital arousal condition associated with a significant level of distress.1–3 PGAD is often characterized by unwelcome and intrusive episodes of genital arousal without corresponding desire or stimulation.4–6 Symptoms most frequently identified include genital congestion, tingling, vaginal lubrication, throbbing, and contractions. Pelvic pain both with and without vaginal penetration as well as nipple erection have also been identified. The frequency of symptoms varies from rare to continuous occurrence; the disorder has been reported to be triggered by visual stimuli, physical stimuli (both sexual and non-sexual stimulation, such as the vibrations felt travelling in a car), stress, and anxiety.7 Factors that have been identified as reducing the degree of arousal include masturbation, orgasm, distraction, intercourse, exercise, and the application of cold compresses.7,8 This spontaneous arousal is often described as distressing, and orgasm may not diminish the sensation.9 While almost all research has focused on PGAD in women, there are at least two case studies of the condition in men.8 For a diagnosis of PGAD, patients should meet the following five diagnostic criteria: 1. genitals are persistently aroused, 2. arousal remains following orgasm or requires multiple orgasms to diminish, 3. arousal is unrelated to desire, 4. arousal is triggered by both sexual and non-sexual stimuli, and 5. symptoms are intrusive and unwelcomed.3,7,9–11 Currently, the definitive cause of PGAD continues to elude clinicians and researchers,9 although a number of potential causes have been proposed. These include central and peripheral neurological anomalies (including small fibre sensory neuropathy), vascular abnormalities causing pelvic congestion, mechanical impingement of the pudendal nerve,12–14 use or withdrawal of medications such as anti- depressants, increased dietary soy intake, and psychological factors such as anxiety.9,15–18 A recent case report described a periclitoral mass in a patient presenting with PGAD. Symptoms temporarily decreased with drainage of the mass and then eventually subsided following surgical excision. The authors proposed that the dorsal nerve was stimulated by the cyst.12 Another study of pudendal nerve entrapment identified continuous arousal in females as a possible presentation.13 Waldinger and colleagues have recently identified a potential connection between PGAD and restless legs syndrome. In patients with PGAD who also have RLS and/or overactive bladder, they classify the condition as restless genital syndrome.19 On the basis of a number of case reports, 16 scientific arguments for ReGS were developed. These included the higher prevalence of RLS in women with PGAD than the general population (67% vs. 3% to 19%), the presence of temporal links between RLS and PGAD, and the observations that RLS and PGAD are both linked to withdrawal of SSRIs, are both associated with a sensation of needing to move and rub the affected area, are both exacerbated by sitting and alleviated by moving, and are both associated with an increased prevalence of pelvic varices.19,20 There is also some evidence that PGAD may be related to pelvic congestion; in a case series describing 18 women with PGAD, the prevalence of pelvic varices was higher than expected (55%). However, the degree of varices in most cases was mild, and the authors were not able to make an argument for causation.21 Tarlov or sacral spinal cysts have also been implicated in the etiology of PGAD. These cysts have been reported to cause paraesthesia and sensory disturbances in the pelvic region. In one study, the prevalence of Tarlov cysts in a cohort of women with PGAD was unusually high.22 While the authors of this study suggested that epidural anaesthesia might temporarily suppress the arousal symptoms if Tarlov cysts were in fact the cause of PGAD symptoms, they had not conducted a prospective study to confirm this.22 There also seems to be overlap between PGAD and pelvic pain. Peripheral nerve hypersensitivity or entrapment and pelvic congestion are identified as two of the most likely etiologies; both of these can result in pelvic pain. In a survey of 103 women who identified themselves as having persistent genital arousal, one third reported having pain in the vaginal or clitoral region.7 PGAD induced by withdrawal of antidepressants has been discussed most commonly in the context of case studies.9 Leiblum et al. found that rates of antidepressant use were particularly high in women with PGAD, although no causal relationship could be inferred because there was also a high rate of depression and anxiety in the study cohort.15 Other authors reviewed cases in which discontinuation of SSRIs and SNRIs were anecdotally linked to the onset of PGAD.16,17 ABBREVIATIONS MRV magnetic resonance venography PGAD persistent genital arousal disorder ReGS restless genital syndrome RLS restless legs syndrome SNRI serotonin–norepinephrine reuptake inhibitors SSRIs selective serotonin reuptake inhibitors 328 l APRIL JOGC AVRIL 2014 WOMEN’S HEAlTH Persistent Genital Arousal in Women With Pelvic and Genital Pain no objective method of detecting or diagnosing PGAD; diagnosis relies on self-reported symptoms. The cohort of 15 women with PGAD described here adds to this growing body of literature. In a previous study, 107 women with PGAD were asked about pelvic pain with and without vaginal penetration, and the authors reported that it was present in 45.7% and 28.6%, respectively.7 No other pain conditions were noted, but it seems that there is an association between PGAD and pelvic pain. All women in our cohort presented to a chronic non-cancer pain clinic, and it was during the assessment of pelvic pain that they were asked about persistent arousal. The relationship of PGAD to pain likely extends beyond the fact that these cases were identified in a pain clinic. The diagnostic criteria for PGAD include the presence of spontaneous and persistent genital arousal that causes distress, which patients often liken to pain or describe as painful. A number of the proposed etiologies include conditions associated with pain, such as central and peripheral neurological anomalies, mechanical impingements of the pudendal nerve, overactive bladder/ ReGS, and pelvic congestion.7,12–14,19–21 Table 4. Aggravating factors n (%) n (%) No trigger 7 (46.7) Lack of sexual activity 1 (6.7) Tension/stress 4 (26.7) Driving 1 (6.7) Sitting 4 (26.7) Post-hysterectomy 1 (6.7) Contact 4 (26.7) Urination 1 (6.7) Walking 2 (13.3) Lack of sleep 1 (6.7) Lying 2 (13.3) Alcohol 1 (6.7) Standing 2 (13.3) Biking 1 (6.7) Arousal 2 (13.3) Alcohol 2 (13.3) Table 5. Alleviating factors n (%) n (%) Orgasm 8 (53.3) Relaxation 1 (6.7) Position 5 (33.3) Cold shower 1 (6.7) Ice application 3 (20.0) Heat application 1 (6.7) Medication 2 (13.3) Sleep 1 (6.7) Post-voiding 1 (6.7) Progressive relaxation 1 (6.7) Distraction 1 (6.7) Table 6. Symptoms Symptoms n (%) Associated symptoms n (%) Arousal 15 (100.0) Swelling 11 (73.3) Spontaneous orgasm 10 (66.7) Allodynia 7 (46.7) Tingling 8 (53.3) Urinary symptoms 6 (40.0) Cramps/spasm 7 (46.7) Hyperalgesia 5 (33.3) Throbbing 6 (40.0) Female “ejaculation” 5 (33.3) Numbness 3 (20.0) Vaginal secretions 4 (26.7) Burning 3 (20.0) Back pain 3 (20.0) Electric sensation 3 (20.0) Abdominal pain 2 (13.3) Itching 2 (13.3) Breast pain 1 (6.7) Pressure/fullness 1 (6.7) Urinary retention 1 (6.7) Sensation of foreign object in urethra 1 (6.7) APRIL JOGC AVRIL 2014 l 329 WOMEN’S HEAlTH Persistent Genital Arousal in Women With Pelvic and Genital Pain While most presentations of PGAD did not coincide with any precipitating event, nerve injury was identified just before the development of persistent arousal in two patients. Sexual abuse immediately before presentation was implicated by one patient. Several more reported a history of childhood sexual abuse, although they did not identify this as a precipitating factor. In our cohort, almost one half of the patients (46.7%) reported a history of sexual abuse, compared with 18% cited in a recent meta-analysis of the world-wide population conducted by Stoltenborgh et al.23 and 19.2% in a previous study at the same centre that assessed a broad scope of patients.24 In a comparative study reported by Leiblum et al., 53% of patients who met all the criteria for PGAD reported having been sexually abused, compared with 44% of patients who did not meet all the criteria.15 While our study identified rates of abuse much higher than in the meta-analysis,23 they are similar to those in the study of Leiblum et al.15 These authors made no causal connections between sexual abuse and PGAD, but speculated whether the patient’s history contributed to their perceptions of symptoms and their rates of depression and anxiety.15 Because seven women in our cohort of 15 reported a history of sexual abuse, in one case occurring immediately before the development of PGAD, a history of sexual abuse should be explored in future studies. A number of comorbidities such as depression and bladder symptoms were found in our cohort. The depression and anxiety rates we found were comparable to those in the study of Leiblum et al., in which 57.9% of patients with PGAD had a history of depression and 42.1% had anxiety.15 In another survey study, 42.7% of patients with PGAD had depression.7 While at least one group of researchers believes that use of antidepressants can be helpful in women with PGAD, especially in those with obsessive traits, anxiety, or depression,9 others have implicated use of antidepressants in the etiology. As described above, it has been proposed that antidepressant withdrawal may play a role in the development of PGAD in some cases.16 In our cohort, eight women (53.3%) reported past tricyclic antidepressants use, four (26.7%) had taken SSRIs, and four (26.7%) had taken SNRIs. It is not surprising that these medications had been so frequently prescribed because they are used in the management of both pain and depression. While a number of women in our cohort had taken and withdrawn from antidepressants, timelines are unclear; we are therefore unable to infer any associations with the development of PGAD in these cases. A better sense of how this class of drugs behaves in the development and treatment of PGAD would be helpful for clinicians. High rates of pelvic varicies and Tarlov cysts in women with PGAD have been described and discussed as possible contributors to PGAD.21,22 Women in our cohort did not all undergo MRI of the sacral spine or MRV, which may account for some of this discrepancy. It is our impression that these conditions are not uncommon in women with pelvic pain. Participants in our study were asked about their ability to have vaginal and/or clitoral orgasm. The responses (60% and 66.7%, respectively) contrast with the numbers that have been cited previously; a lay report suggests that approximately 30% of women are able to have vaginal orgasm without clitoral stimulation.25 Our findings could indicate a heightened arousal tendency. The proportion of sexually active women in our cohort was 73.3%, higher than the 64% reported in a general population sample,26 which also supports a heightened arousal tendency. A number of aggravating and alleviating factors for PGAD were identified by our study subjects. These were fairly consistent with those discussed by Leiblum et al.7 Medication was reported to provide the most relief (at least some relief in 52% of respondents and complete relief in 3.9%) followed by masturbation, orgasm, intercourse, distraction, exercise, and application of cold compresses. Medication was not identified as helpful in our cohort except in two cases (using duloxetine and topical EMLA cream). This may have been because it was not systematically explored. There are some limitations to this study. First, the study cohort was drawn from a pain clinic, so the findings in this study apply only to the chronic pain population. Second, the study’s sample size is small, comprising 15 women. In order to move beyond a descriptive study, a larger sample size is required. The lack of a control group is also a limitation of the study. We examined indicators of associated conditions rather than the prevalence of PGAD. Another limiting feature of this study was the heavy reliance on patient reports. It was difficult to understand, for example, if a medication used in the past was targeted at pelvic pain or PGAD, and in some cases patients found it challenging to differentiate between “pain” and arousal. Future studies should be prospective and should assess treatment modalities. We suspect that there are several variants of PGAD and that multiple pathophysiological factors are involved. Whether this is the case can be determined only by consistent questioning and clinical evaluation. We need to understand 330 l APRIL JOGC AVRIL 2014 WOMEN’S HEAlTH the phenotypes before we can start to consider treatments. Women should be asked routinely about persistent arousal symptoms as a component of sexual and reproductive history-taking. It is likely that more cases would be identified if this question was asked routinely. For women who present with PGAD, the history should include questions about past or current sexual abuse, heightened arousal (i.e., anorectal, urethral, and nipple-related arousal), and orgasm (including spontaneous nocturnal orgasm), in addition to asking about persistent arousal. We need to learn more about the natural history of PGAD. A review of use of antidepressants and other medications, including the indications for use, discontinuation, and timing in relation to the development of persistent arousal would also be both clinically relevant and useful in future research. Finally, women presenting with PGAD would ideally undergo MRI to identify Tarlov cysts and MRV to identify pelvic varices. While this may not always be feasible given the cost and availability of these tools, it would provide subsidiary information to bolster further research. CONClUSION The number of women presenting to a chronic non- cancer pain clinic who had persistent genital arousal symptoms was surprising. All women should be asked about these symptoms, as they are often hesitant to discuss them despite the significant distress and disability they cause. Specifically, women who present with pelvic pain, interstitial cystitis and other urinary pain, restless legs syndrome, and pudendal neuralgia should be asked about persistent genital arousal symptoms. Further study is required to understand the etiology of PGAD and how widespread and disabling this problem is, and to investigate approaches to management. REFERENCES 1. Garvey LJ, West C, Latch N, Leiblum S, Goldmeier D. Report of spontaneous and persistent genital arousal in women attending a sexual health clinic. Int J STD AIDS 2008;20:519–21. 2. Goldmeier D, Leiblum S. Persistent genital arousal in women—a new syndrome entity. Int J STD AIDS 2006;17:215–21. 3. Goldmeier D, Mears A, Hiller J, Crowley T. Persistent genital arousal disorder: a review of the literature and recommendations for management. Int J STD AIDS 2009;20: 373–7. 4. Basson R, Althof S, Davis S, Fugl-Meyer K, Goldstein I, Leiblum S, et al. Summary of the recommendations on sexual dysfunctions in women. 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