Pudendal nerve blockade for persistent genital arousal disorder (PGAD): A clinical review and case report.

BACKGROUND: Persistent genital arousal disorder (PGAD) is a condition characterized by unwanted and potentially painful genital sensations or spontaneous orgasms without stimulation. We present a case of a 55-year-old woman with refractory genital arousal disorder that was treated with serial pudendal nerve blocks. CASE: RW is a 55-year-old woman with chronic pelvic pain, pudendal neuralgia, MDD, SI, GAD, CRPS, and persistent genital arousal disorder for 11 years. Her PGAD was refractory to conservative management, physical therapy, and bilateral clitoral artery embolization. We performed bilateral pudendal nerve blocks with Kenalog and Bupivacaine, which provided almost complete relief for 2-3 months. We performed a bilateral pudendal nerve radiofrequency ablation; however, there was minimal benefit. RW continues to have significant relief with serial pudendal nerve blocks. SUMMARY AND CONCLUSION: Persistent genital arousal disorder is often refractory to medication and physical therapy requiring significant intervention such as entrapment surgery or artery embolization. Our case demonstrates pudendal nerve blocks as a potential treatment modality with minimal side effects.

Lumbar endoscopic spine surgery for persistent genital arousal disorder/genitopelvic dysesthesia resulting from lumbosacral annular tear-induced sacral radiculopathy.

BACKGROUND: Persistent genital arousal disorder/genitopelvic dysesthesia (PGAD/GPD) is characterized by distressing, abnormal genitopelvic sensations, especially unwanted arousal. In a subgroup of patients with PGAD/GPD, cauda equina Tarlov cyst-induced sacral radiculopathy has been reported to trigger the disorder. In our evaluation of lumbosacral magnetic resonance images in patients with PGAD/GPD and suspected sacral radiculopathy, some had no Tarlov cysts but showed lumbosacral disc annular tear pathology. AIM: The aims were 2-fold: (1) to utilize a novel multidisciplinary step-care management algorithm designed to identify a subgroup of patients with PGAD/GPD and lumbosacral annular tear-induced sacral radiculopathy who could benefit from lumbar endoscopic spine surgery (LESS) and (2) to evaluate long-term safety and efficacy of LESS. METHODS: Clinical data were collected on patients with PGAD/GPD who underwent LESS between 2016 and 2020 with at least 1-year follow-up. LESS was indicated because all had lumbosacral annular tear-induced sacral radiculopathy confirmed by our multidisciplinary management algorithm that included the following: step A, a detailed psychosocial and medical history; step B, noninvasive assessments for sacral radiculopathy; step C, targeted diagnostic transforaminal epidural spinal injections resulting in a temporary, clinically significant reduction of PGAD/GPD symptoms; and step D, surgical intervention with LESS and postoperative follow-up. OUTCOMES: Treatment outcome was based on the validated Patient Global Impression of Improvement, measured at postoperative intervals. RESULTS: Our cohort included 15 cisgendered women and 5 cisgendered men (mean ± SD age, 40.3 ± 16.8 years) with PGAD/GPD who fulfilled the criteria of lumbosacral annular tear-induced sacral radiculopathy based on our multidisciplinary management algorithm. Patients were followed for an average of 20 months (range, 12-37) post-LESS. Lumbosacral annular tear pathology was identified at multiple levels, the most common being L4-L5 and L5-S1. Twenty-two LESS procedures were performed in 20 patients. Overall, 80% (16/20) reported improvement on the Patient Global Impression of Improvement; 65% (13/20) reported improvement as much better or very much better. All patients were discharged the same day. There were no surgical complications. CLINICAL IMPLICATIONS: Among the many recognized triggers for PGAD/GPD, this subgroup exhibited lumbosacral annular tear-induced sacral radiculopathy and experienced long-term alleviation of symptoms by LESS. STRENGTHS AND LIMITATIONS: Strengths include long-term post-surgical follow-up and demonstration that LESS effectively treats patients with PGAD/GPD who have lumbosacral annular tear-induced sacral radiculopathy, as established by a multidisciplinary step-care management algorithm. Limitations include the small study cohort and the unavailability of a clinical measure specific for PGAD/GPD. CONCLUSION: LESS is safe and effective in treating patients with PGAD/GPD who are diagnosed with lumbosacral annular tear-induced sacral radiculopathy.

Psychological Treatment of Persistent Genital Arousal Disorder/Genitopelvic Dysesthesia Using an Integrative Approach.

Persistent genital arousal disorder/genitopelvic dysesthesia (PGAD/GPD) is characterized by persistent, unwanted physiological genital arousal (i.e., sensitivity, fullness, and/or swelling) in the absence of sexual excitement or desire which can persist for hours to days and causes significant impairment in psychosocial well-being (e.g., distress) and daily functioning. The etiology and course of PGAD/GPD is still relatively unknown and, unsurprisingly, there are not yet clear evidence-based treatment recommendations for those suffering from PGAD/GPD. We present the case of a 58-year-old woman with acquired persistent genital arousal disorder, which began in March 2020; she believed she developed PGAD/GPD due to a period of significant distress and anxiety related to the COVID-19 pandemic. After seeking medical diagnosis and treatment from multiple healthcare providers and trying a combination of pharmacological and medical treatment modalities, she presented for psychological treatment. An integrative therapy approach (3 assessment sessions, 11 treatment sessions), which included cognitive behavior therapy, distress tolerance and emotion regulation skills from dialectical behavior therapy, and mindfulness practice, was utilized. The patient reported improvements anecdotally (e.g., decreased impact on occupational and social functioning, greater self-compassion, less frequent and shorter duration of PGAD/GPD flare-ups, improved ability to cope with PGAD/GPD symptoms, and decreased need for sleeping medication) and on self-report measures (e.g., lower PGAD/GPD catastrophizing, lower anxiety and depression, and greater overall quality of life).We report the use of an integrative (i.e., psychoeducational, cognitive behavioral, dialectical behavioral, and mindfulness-based) intervention, which may be an effective psychological treatment for PGAD/GPD.

Treatments for Persistent Genital Arousal Disorder in Women: A Scoping Review.

BACKGROUND: Persistent genital arousal disorder (PGAD) is characterized by elevated discomfort associated with persistent genital arousal in the absence of sexual desire. AIM: To perform a scoping review of the proposed treatments for PGAD and their efficacy. METHODS: A scoping review was carried out (PRISMA-Scr) that included articles on PGAD as the main disorder, only in women, which explained, in detail, the treatment and its efficacy, was empirical, was written in English and Spanish. No prior filtering by years was performed. OUTCOMES: Three different effective treatments were found (Physical therapies, pharmacological therapies, and psychotherapeutics in combination with other therapies). RESULTS: Thirty-eight articles were selected. From physical therapies, treatments using neuromodulation, transcutaneous electrical stimulation, Botox, surgery, electroconvulsive therapy, manual therapy, pelvic floor therapy, dietary changes, and transcranial magnetic stimulation showed effectiveness. Using the pharmacological approach, paroxetine, duloxetine, pramipexole, ropinirole, and clonazepam treatments were effective. Psychotherapy treatments showed effectiveness only in combination with other types of treatments, specifically a combination of cognitive-behavioral strategies with pharmacological treatment. CLINICAL IMPLICATIONS: Pharmacological treatment, specifically SSRIs, have proven to be the therapy of choice for different subtypes of patients. STRENGTHS AND LIMITATIONS: This study analyzed treatment effectiveness with different approaches and took into consideration those articles where psychotherapy was used as a combination treatment with pharmacological and physical therapy. The main limitation is that it was focused exclusively on women, and the results cannot be generalized to include men. CONCLUSIONS: To date, a combination of pharmacological interventions with physical therapy and, in some occasions, with psychological therapy is main strategy followed to accomplish effective treatment of PGAD. Martín-Vivar M, Villena-Moya A, Mestre-Bach G, et al. Treatments for Persistent Genital Arousal Disorder in Women: A Scoping Review. J Sex Med 2022;19:961-974.

Persistent Genital Arousal Disorder: A Study on an Italian Group of Female University Students.

There are very few studies in literature about Persistent Genital Arousal Disorder (PGAD) prevalence to date, and no one has ever been done in Italy. This study replicated the earlier Canadian research in order to investigate the prevalence of PGAD diagnostic criteria in an Italian group of 679 female university students and to compare the results with those obtained in the Canadian group. In addition, the research aimed at verifying if the type or number of PGAD criteria met and the intensity of symptoms are associated with more negative emotions, and if they can be related to age and sexual orientation. The results confirm that some women can experience spontaneous genital sensations that can vary in intensity and duration, and can be associated with a wide range of emotions. However, the results seem to suggest the importance of other factors in the evaluation of the symptoms, rather than factors related to their nature and intensity or age and sexual orientation. Moreover, 2 participants (0.29%) met all five criteria and reported high distress, confirming that there is a potential clinical condition called PGAD, in which the hypertrophic, intense and intrusive spontaneous genital arousal might determine the presence of extremely negative emotions.

Prevalence of Persistent Genital Arousal Disorder in 2 North American Samples.

BACKGROUND: Persistent genital arousal disorder (PGAD) is a highly distressing, understudied condition characterized by persistent genital arousal (eg, genital sensations, sensitivity) in the absence of sexual desire. Currently, there is limited information about the prevalence of PGAD based on its proposed diagnostic criteria ("PGAD criteria"). AIM: This study sought to assess the prevalence of PGAD criteria in 2 North American samples: a large, non-clinical sample of Canadian undergraduate students (Study 1), and a nationally representative sample from the U.S. (Study 2). METHODS: The incoming class of undergraduate students (N = 1,634) enrolled in the Introduction to Psychology course at a Canadian university and a nationally representative sample of U.S. participants (N = 1,026) responded to questions about each PGAD criterion, and distress associated with these experiences. OUTCOMES: 5 self-report questions were developed based on each of the Leiblum and Nathan 2001 PGAD criteria, and a measure of associated distress was included. The U.S. sample (Study 2) also responded to questions about medical comorbidities and their knowledge of the term "PGAD." RESULTS: 1.1% (n = 4; Study 1) to 4.3% (n = 22; Study 2) of men and 0.6% (n = 7; Study 1) to 2.7% (n = 14; Study 2) of women reported experiencing all 5 PGAD criteria at a moderate to high frequency. Even greater proportions of participants reported experiencing all 5 criteria at any frequency (6.8-18.8%). Although ratings of associated distress varied, participants who were distressed by these symptoms most frequently endorsed the first PGAD criterion: physiological genital arousal in the absence of sexual excitement or desire. These results are similar to previously reported rates of PGAD. CLINICAL IMPLICATIONS: A non-trivial number of individuals may experience PGAD, and it should be screened for by healthcare practitioners. STRENGTHS & LIMITATIONS: This study is the first to use 2 large, non-clinical samples to assess the prevalence of PGAD symptoms. However, barriers to reporting symptoms, such as shame or embarrassment, may have resulted in underestimates of prevalence in the present sample. CONCLUSION: The prevalence of the 5 PGAD criteria in 2 large non-clinical samples ranged from similar to higher than rates reported in previous research. However, distress ratings associated with each of the 5 criteria varied, with most respondents describing them primarily as neutral or non-distressing. Jackowich RA, Pukall CF. Prevalence of Persistent Genital Arousal Disorder in 2 North American Samples. J Sex Med 2020;17:2408-2416.

Female Sexual Dysfunction-Medical and Psychological Treatments, Committee 14.

INTRODUCTION: Since the millennium we have witnessed significant strides in the science and treatment of female sexual dysfunction (FSD). This forward progress has included (i) the development of new theoretical models to describe healthy and dysfunctional sexual responses in women; (ii) alternative classification strategies of female sexual disorders; (iii) major advances in brain, hormonal, psychological, and interpersonal research focusing on etiologic factors and treatment approaches; (iv) strong and effective public advocacy for FSD; and (v) greater educational awareness of the impact of FSD on the woman and her partner. AIMS: To review the literature and describe the best practices for assessing and treating women with hypoactive sexual desire disorder, female sexual arousal disorder, and female orgasmic disorders. METHODS: The committee undertook a comprehensive review of the literature and discussion among themselves to determine the best assessment and treatment methods. RESULTS: Using a biopsychosocial lens, the committee presents recommendations (with levels of evidence) for assessment and treatment of hypoactive sexual desire disorder, female sexual arousal disorder, and female orgasmic disorders. CONCLUSION: The numerous significant strides in FSD that have occurred since the previous International Consultation of Sexual Medicine publications are reviewed in this article. Although evidence supports an integrated biopsychosocial approach to assessment and treatment of these disorders, the biological and psychological factors are artificially separated for review purposes. We recognize that best outcomes are achieved when all relevant factors are identified and addressed by the clinician and patient working together in concert (the sum is greater than the whole of its parts). Kingsberg SA, Althof S, Simon JA, et al. Female Sexual Dysfunction-Medical and Psychological Treatments, Committee 14. J Sex Med 2017;14:1463-1491.

Toward a More Evidence-Based Nosology and Nomenclature for Female Sexual Dysfunctions-Part II.

INTRODUCTION: Current Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5) definitions of sexual dysfunction do not identify all sexual problems experienced clinically by women and are not necessarily applicable for biologic or biopsychosocial management of female sexual dysfunction. A unified nomenclature system enables clinicians, researchers, and regulatory agencies to use the same language and criteria for determining clinical end points, assessing research results, and managing patients. AIM: To develop nomenclature with classification systems for female sexual desire, arousal, and orgasm disorders with definitions pertinent to clinicians and researchers from multiple specialties who contribute to the field of sexual medicine. METHODS: Key national and international opinion leaders diverse in gender, geography, and areas of expertise met for 2 days to discuss and agree to definitions of female sexual desire, arousal, and orgasm disorders and persistent genital arousal disorder. The attendees consisted of 10 psychiatrists and psychologists; 12 health care providers in specialties such as gynecology, internal medicine, and sexual medicine; three basic scientists; and one sexuality educator, representing an array of societies working within the various areas of sexual function and dysfunction. MAIN OUTCOME MEASURE: A unified set of definitions was developed and accepted for use by the International Society for the Study of Women's Sexual Health (ISSWSH) and members of other stakeholder societies participating in the consensus meeting. RESULTS: Current DSM-5 definitions, in particular elimination of desire and arousal disorders as separate diagnoses and lack of definitions of other specific disorders, were adapted to create ISSWSH consensus nomenclature for distressing sexual dysfunctions. The ISSWSH definitions include hypoactive sexual desire disorder, female genital arousal disorder, persistent genital arousal disorder, female orgasmic disorder, pleasure dissociative orgasm disorder, and female orgasmic illness syndrome. CONCLUSION: Definitions for female sexual dysfunctions that reflect current science provide useful nomenclature for current and future management of women with sexual disorders and development of new therapies.

A periclitoral mass as a cause of persistent genital arousal disorder.

INTRODUCTION: Persistent genital arousal disorder (PGAD) is an intrusive and unremitting disorder for which several possible etiologies and treatments have been suggested. AIM: To describe a woman who developed PGAD in association with a periclitoral mass, a potential physical cause of the disorder that has not been previously described in the medical literature. METHODS: A postmenopausal woman presented with 6 months of persistent, unrelenting genital arousal and clitoral pain that was unrelated to sexual stimuli. Careful examination revealed a tender, firm, mobile, left-sided mass that appeared to compress the dorsal nerve of the clitoris. RESULTS: Complete excision of the mass resulted in full resolution of her symptoms over several weeks. CONCLUSION: Localized causes of persistent genital arousal, though rare, should be included in the differential diagnosis PGAD as detection and treatment can lead to a complete recovery.

Persistent genital arousal in women with pelvic and genital pain.

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.

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.

Treatment in persistent genital arousal disorder: a scoping review.

Background: Persistent genital arousal disorder (PGAD) is a rare condition characterized by unwanted and distressing symptoms of arousal and dysesthesia. The aim of this scoping review was to map the current state of PGAD management, identify gaps in the literature, and understand patient perspectives. Methods: We completed a scoping review following guidelines from the Joanna Briggs Institute and the Preferred Reporting Items for Systematic Reviews and Meta Analyses Scoping Reviews extension. A systematic literature search for articles pertaining to PGAD/genito-pelvic dysesthesia (GPD) was conducted in August 2023 via Medline, Embase, Scopus, and Web of Science. The search returns were deduplicated and the remaining titles and abstracts were screened for inclusion. General publication characteristics and treatment data were extracted from the included publications via a pilot-tested Google form. All screening and extraction were completed in a masked, duplicate fashion. Results: Findings from our scoping review revealed a scarcity of systematic research, limited evidence-based data, and the importance of addressing both physical and psychiatric concerns. Our sample included 46 publications from an initial pool of 636 returns. Case studies were the most common study design. Thirty-three studies examined medication, either alone or as part of a treatment regimen. Selective serotonin reuptake inhibitors were the most used medication, followed by pramipexole and carbamazepine. Seven studies used a surgical or procedural intervention. Treatment with pelvic floor Botox was the most common procedure. Patient perspectives in the included case studies highlighted themes of shame, suicidal ideation, social isolation, decreased sleep, and overall decline in quality of life. Conclusion: The findings from our study emphasize patients' distressing and psychiatric symptoms, indicating a need to improve treatment regimens, using both evidence-based research outcomes and patient-reported outcomes. Management for PGAD/GPD lacks a standardized framework, indicating a need for further research and the development of clinical practice guidelines to improve patient care.

Predictors of Psychosocial and Functional Outcomes in Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia: Application of the Fear-Avoidance Model.

Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia (PGAD/GPD), which affects up to 4.3% of individuals, is a distressing and poorly understood condition characterized by persistent, unwanted, and often painful sensations of genito-pelvic arousal (eg, throbbing) in the absence of sexual desire. PGAD/GPD is associated with significant negative impacts on psychosocial well-being and daily functioning. Recent research has indicated that PGAD/GPD shares many similarities with other forms of chronic genito-pelvic pain. This study applied the fear-avoidance model of chronic pain to PGAD/GPD to identify cognitive and behavioral factors associated with psychosocial and functional outcomes. A total of 263 individuals with PGAD/GPD symptoms completed a cross-sectional online survey of symptom intensity, cognitive and behavioral predictors (symptom catastrophizing, hypervigilance to symptoms, symptom fear and avoidance, self-efficacy), depression symptoms, and role functioning. Symptom catastrophizing, fear of symptoms, avoidance of symptoms, and hypervigilance to PGAD/GPD symptoms were significantly correlated with poorer psychosocial and functional outcomes, whereas higher self-efficacy was significantly associated with lower depression and better role functioning. Two serial parallel mediation models examined the fear-avoidance pathway from PGAD/GPD symptom intensity to depression symptoms and role functioning. In both models, the pathway through symptom catastrophizing, fear of symptoms, and symptom avoidance was significant, but the pathway through symptom catastrophizing, fear of symptoms, and symptom hypervigilance was not. The results of this study provide support for the applicability of the fear-avoidance model to PGAD/GPD. Interventions targeting fear-avoidance factors may help to reduce PGAD/GPD symptom intensity, distress, and increase psychological well-being and daily functioning. PERSPECTIVE: This article provides support for the applicability of the fear-avoidance model of chronic pain to Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia (PGAD/GPD). These results suggest that interventions targeting fear-avoidance cognitions and behaviors (catastrophizing, fear, avoidance, hypervigilance) may help to reduce PGAD/GPD symptom intensity and improve psychological well-being and daily functioning.

Pharmacotherapy of persistent genital arousal disorder/genito-pelvic dysesthesia: an updated review and data from a registry.

INTRODUCTION: Persistent genital arousal disorder/genitopelvic dysesthesia (PGAD/GPD) is a rare, yet debilitating disease, which has been included in ICD-11. Pharmacotherapy in PGAD/GPD is a mixed blessing - drugs may either alleviate symptoms or worsen/induce them. Therefore, we aim at offering both an overview of pharmacological approaches to treat this disorder, including treatment failures, and to highlight drugs that may induce or worsen symptoms. We include all available data published so far as well as data from an own registry. AREAS COVERED: The international registries have not recorded any completed or ongoing trials on pharmacotherapy of PGAD/GPD. We refer to case reports, case series, online surveys, and data from our own registry that includes 90 subjects with PGAD/GPD. EXPERT OPINION: There is weak evidence (level 4) that some drugs such as SSRIs, SNRIs, cannabinoids, and anticonvulsants (pregabalin and gapabentin) may alleviate symptoms in PGAD/GPD. However, serotonergic drugs may also induce or worsen PGAD/GPD during administration or termination, as observed in 28% of cases. In conclusion, the pharmacotherapy of PGAD/GPD is still in its infancy just like the etiopathological understanding of the disease. Clinicians should be aware of PGAD/GPD, conduct careful diagnostics, and discuss an individual treatment plan with the patient.

Restless Genital Syndrome: A Case Report of the Challenging Diagnosis.

Restless genital syndrome (RGS) is a rare disorder marked by paresthesia and discomfort in the genital area, akin to restless legs syndrome (RLS). While RLS typically affects the lower limbs, its impact on areas such as the bladder has been noted. RGS individuals exhibit sensory symptoms akin to RLS, including difficulty expressing sensations and a compulsion for genital rubbing. Thus, RGS is viewed as an atypical RLS presentation, characterized by genital sensory symptoms. Despite the rarity, this report details a successfully managed case using conventional RLS treatments. Numerous RGS aspects need clarification, including prevalence and treatment. Due to its distressing nature, sustained investigation is vital. Though lacking a standard treatment, our patient benefited from traditional RLS medication, hinting at shared mechanisms. Further research is vital for understanding and treating RGS effectively.

Two case presentations of profound labial edema as a presenting symptom of hypermobility-type Ehlers-Danlos syndrome.

INTRODUCTION: Hypermobility-type Ehlers-Danlos syndrome (EDS), an often-missed diagnosis with the potential for serious sequelae, may have a variety of uncommon presentations, some of which may be gynecologic. AIM: The aim of this case report is to present two cases of profound labial edema associated with intercourse as a presenting symptom of hypermobility-type EDS. METHODS: A 25-year-old female presented with severe labia minora swelling and bladder pressure associated with intercourse, in addition to persistent genital arousal. History revealed easy bruising, joint pain, and family history of aneurysm. A 22-year-old female presented with intermittent profound labial swelling for 6 years, associated with sensitivity and pain with intercourse. The patient has a history of joint pain and easy bruising, as well a sister with joint hypermobility and unexplained lymphedema. The presenting symptom of profound labial edema led to the diagnosis of hypermobility-type EDS. RESULTS: Patients with hypermobility syndrome exhibit an increased ratio of type III collagen to type I collagen, causing tissue laxity and venous insufficiency. Abnormal collagen may lead to gynecologic manifestations, including unexplained profound labial edema, pelvic organ prolapse in the absence of risk factors, and possibly persistent genital arousal. CONCLUSIONS: This case report highlights the need for further research to determine incidence of labial edema in hypermobility-type EDS and to further elucidate a potential correlation between profound labial edema and collagen disorders.

Persistent Genital Arousal Disorder: Two Case Studies and Exploration of a Novel Treatment Modality.

Persistent genital arousal disorder (PGAD) is characterized by persistent unwanted feelings of sexual arousal that can be debilitating. Despite first being defined >20 years ago, the precise etiology and treatment of this disorder remain elusive. Mechanical disruption of nerves, neurotransmitter changes, and cyst formation have all been considered as etiologies involved with the development of PGAD. With limited and ineffective treatment modalities, many women live with their symptoms untreated or undertreated. To broaden the literature, we present two cases of PGAD and present a novel treatment modality of the disorder with the use of a pessary. Although there was subjective success in dampening the symptoms, they were not completely resolved. These findings open the door for the potential of similar treatments in the future.

Effectiveness of Brexpiprazole in a Patient With Bipolar Disorder and Comorbid Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia: A Case Report.

Although the symptoms of persistent genital arousal disorder/genito-pelvic dysesthesia (PGAD/GPD) can have negative impacts on patients' lives, it is an under-recognized clinical entity. We describe the case of a 61-year-old Japanese female who suffered simultaneously from bipolar disorder and PGAD/GPD. She developed PGAD/GPD approx. 10 years after being diagnosed with bipolar disorder. Despite 20 years of various drug treatments, her bipolar disorder and PGAD/GPD symptoms showed little improvement. She had also undergone multiple sessions of cognitive behavioral therapy (CBT) and mindfulness, nerve block, botulinum toxin injections, and laser treatment for PGAD/GPD. Her PGAD/GPD symptoms remained with no significant improvement, and her bipolar disorder symptoms had also not responded well to medication. With the administration of brexpiprazole, she achieved remission of her bipolar disorder. Her PGAD/GPD symptoms also eventually improved. When PGAD/GPD is comorbid with bipolar disorder, the improvement of bipolar disorder may also lead to relief of PGAD/GPD symptoms. This case reveals that brexpiprazole, which has a unique profile, may be effective for PGAD/GPD.

Persistent genital arousal disorder - the present knowledge. / Zespól przetrwalego pobudzenia genitalnego ­ aktualny stan wiedzy.

Persistent genital arousal disorder (PGAD) is a relatively recently described sexual disorder, characterized by symptoms of spontaneous genital arousal which persist in the absence of sexual desire and may affect women and men. Epidemiological studies conducted so far indicate that the prevalence of PGAD in the population may reach 1-4%. The etiology of PGAD remains unclear and complex, hypothesized causes include vascular, neurological, hormonal, psychological, pharmacologic, dietary, mechanical factors or a combination of these factors. Proposed methods of treatment include pharmacotherapy, psychotherapy, electroconvulsive therapy, hypnotherapy, injection of botulinum toxin, pelvic floor physical therapy, application of anesthetizing agents, reduction of identifiable factors exacerbating the symptoms, and transcutaneous electrical nerve stimulation. There is no standardized treatment algorithm for PGAD due to lack of clinical trials (evidence-based medicine). The classification of PGAD is under discussion: it could be classified as a separate sexual disorder, a subtype of vulvodynia or a disorder with pathogenesis similar to overactive bladder (OAB) and restless legs syndrome (RLS). Due to specificity of symptoms, patients may feel shame and discomfort during the examination or even delay reporting symptoms to the specialist. Thus, it is crucial to spread knowledge about this disorder, which would allow doctors to diagnose and help PGAD patients sooner.

Exploring the link between eating disorders and persistent genital arousal disorder/genito-pelvic dysesthesia: first description and a systematic review of the literature.

BACKGROUND: Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia (PGAD/GPD) characterized by recurrent physiological genital without corresponding psychological arousal is a poorly understood and researched condition. Based on the first two case descriptions of eating disorders directly linked to PGAD/GPD the aim of this paper was to systematically review the literature on possible associations between eating disorders and PGAD/GPD. METHOD: A systematic literature search on eating disorders and PGAD/GPD was conducted in PubMed, PsycINFO, and Scopus, complemented by Google Scholar. We included case reports, case series, cross-sectional studies and review articles published in peer-reviewed journals written in English or German-language. RESULTS: The included original papers described a total of 2078 cases with PGAD/GPD symptomatology. Of these, 892 participants fulfilled all five PGAD/GPD core criteria. The aetiology of PGAD/GPD is unknown. Multifactorial genesis of PGAD/GPD is presumed including neurological, pharmacological, hormonal, vascular and psychological causes. A high degree of psychological comorbidity is reported. No study was found that drew a direct link between eating disorders and PGAD/GPD. Although PGAD/GPD symptoms also occur in adolescents, there are no findings in this regard. However, we found a gap in data collection: eating disorders as potential psychiatric comorbidities were systematically recorded in only a few studies. CONCLUSION: The existing literature have not yet considered a possible link between eating disorders and PGAD/GPD so far. According to the authors' knowledge, this work is the first review to systematically explore the associations. We suspect underreporting of PGAD/GPD cases in eating disorders and particularly during adolescence. We argue that there are several common factors that appear to be important in the etiology, course, and treatment of both disorders (e.g. hormonal dysregulation or sensory sensitivity and avoidance), warranting future research on the possible comorbidity of these disorders.

Persistent genital arousal disorder/genito-pelvic dysesthesia (PGAD/GPD) is characterized by recurrent physiological genital arousal without corresponding psychological arousal. PGAD/GPD is a poorly understood and researched disorder. It is often associated with significant psychological impairment and distress.Eating disorders are severe psychological illnesses that predominantly affect the female sex. They have a high psychological comorbidity and are often associated with severe physical consequences.We treated two female patients in whom there was a direct association between PGAD/GPD and eating disorder. In our systematic review of the literature, we found no study that further investigated the co-occurrence between PGADPGAD/GPD and eating disorders. This paper is the first to describe and investigate a relationship between PGAD/GPD and eating disorders.In previous literature, several factors have been hypothesized for the development of PGAD/GPD but also eating disorders. We argue that there are several common factors that appear to be important in the etiology, course, and treatment of both disorders (e.g. hormonal dysregulation or sensory sensitivity and avoidance), warranting future research on the possible comorbidity of these disorders.

Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia caused by Sacroiliac Joint Dysfunction.

INTRODUCTION: Persistent genital arousal disorder/genito-pelvic dysesthesia (PGAD/GPD) consists of persistent or recurrent unwanted sensations of genital arousal that may include other types of genito-pelvic dysesthesia, which occur without concomitant sexual interest or thoughts. There are multiple triggering factors for PGAD/GPD. AIM: To report the case of a 38-year-old woman with low back pain and PGAD/GPD triggered by sacroiliac joint dysfunction. METHODS: The medical data of the female patient with low back pain and PGAD/GPD were reviewed and analyzed. RESULTS: Resetting of the subluxated sacroiliac joint resulted in complete remission of the patient's symptoms. CONCLUSION: Sacroiliac joint dysfunction may be a trigger for PGAD/GPD in some cases. Lack of relevant knowledge among patients and healthcare providers is the biggest challenge of the proper diagnosis and treatment of PGAD/GPD at present. Zhang Y, Su L, Ge H, et al. Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia caused by Sacroiliac Joint Dysfunction. Sex Med 2022;10:100544.