Persistent Genital Arousal Disorder in Perimenopausal Woman: A Case Report

Introduction

Persistent genital arousal disorder (PGAD) is a newly recognized sexual dysfunction with poor understanding and few case reports. There are concerns as to the validity of the results because of little research and the prevalence is yet to be identified [1-5]. It is characterized by physiological signs of genital arousal that persist despite the absence of sexual desire and diagnosed in female [6]. The symptoms of PGAD are as follows: [7]

• Symptoms characteristic of sexual arousal (genital fullness/ swelling and sensitivity with or without nipple fullness/ swelling) which persist for an extended period of time (hours to days) and do not subside completely on their own.

• Symptoms of physiological arousal which do not resolve with ordinary orgasmic experience and may require multiple orgasms over hours or days to remit.

• Symptoms of arousal which are usually experienced as unrelated to any subjective sense of sexual excitement or desire.

• The persistent genital arousal which may be triggered not only by a sexual activity but seemingly also by nonsexual stimuli or by no apparent stimulus at all.

• Symptoms which are experienced as unbidden, intrusive, and unwanted.

• The symptoms which cause the patient at least a moderate degree of distress.

It was previously called persistent sexual arousal syndrome (PSAS) and was first described by psychiatrists Leiblum and Nathan in 2001. Subsequently renamed as PGAD by Goldmeier et al. in 2009 and also named as Restless Genital Syndrome [1,7,8]. Though PGAD is a newly recognized condition as well as scanty published data regarding etiology, diagnosis and treatment, it has significant impact on social life as it is very distressful of being irritated by genitalia. It was aimed to report the new diagnosis as well as significant improvement in symptom profile and social functioning.

Case

A 49-year-old Asian perimenopausal female, sent from the Psychiatry out-patient department (OPD) of a tertiary care hospital presented to the Psychiatric Sex Clinic (PSC) in January 2015 with the complaints of intense sexual urge which is uncomfortable; demands release; and interferes with life pleasure and activities for last two years. She becomes bound to masturbate to relief tension, but it does not always go away with orgasm. She also experiences of hot flashes and her menstruation became irregular for last 1 year. The desire is quite different from her normal sexual desire and she experiences it without any sexual cues. Sometimes it persists continuously and aggravated with the touch of her thigh while she sitting with closed thigh position. She feels better if she masturbates and she gets more pleasure in women on top position instead of lithotomy position. She feels guilty as masturbation is sinful to her judgment as well as less enjoyable. She remarriages a 40 years old man secretly to be comfortable for sex for last 6 months; which is not acceptable to her family members as well as not also culturally praiseworthy as her husband died 3 years back and she stays with her two adult sons. She used to meet her new husband secretly and faces legal problems based on social grounds that make her socially very disgraceful and so distressful that she intends to commit suicide. As a person, premorbidly she was sociable, open minded and had a great enthusiasm for sex. She had boyfriends but no extramarital relationship as well as no premarital sex but she used to talk about sex with them.

Her methodical physical examination revealed nothing contributory regarding her present complaints. Mental state examination revealed a perimenopausal woman with average body build wearing socially and culturally appropriate dress up. She looked tense, embarrassed and anxious. Her mood was depressed. She had pessimistic thought about her physical symptom and had suicidal ideation. Delusion and hallucination was not found and she was in normal cognitive state. All routine investigations, Pelvic ultra sonography, MRI of lumbo sacral spine, possible detailed hormonal analysis revealed nothing abnormal. She was diagnosed as a case of PGAD according to the criteria proposed by Leiblum; as her arousal was intrusive in nature, not associated with sexual context and psychological desire for sex, her urge does not go away with orgasm as well as her style of masturbation more related with clitoral stimulation. The diagnosis was assigned as a diagnosis of exclusion of other possible differentials.

Treatment

After diagnosis; psychoeducation, explanation and counseling provided. She was advised tablet Risperidone 4mg per day (after initial up titration), and tablet Carbamazepine 200mg (Controlled Release formulation) per day as pharmacotherapy based on evidence in previous reports and advised to follow up after 2 weeks in the OPD set up. In the first follow up her symptoms decrease both in frequency and intensity more than fifty percent. She is followed up for last 1 year with the above mentioned medications and she became symptom free after 6 months of continuous treatment. At present she is with medications and without symptoms and attempts to taper the medications is not tried yet.

Discussion

It is the first case of PSC documented detailed description of symptoms, associated with PGAD. There is little and not reliable data on the prevalence of PGAD. It’s possible that the disorder is more common than researches’ assumption [8,9]. On one hand the disorder is being very rare, on the other- the condition is frequently unreported by sufferers who may consider it embarrassing. Leiblum (2006) postulated that there may be two subtypes of PGAD: one more closely related to physiological factors (e.g, neurovascular or neurochemical), and the other to psychological factors. Several hypotheses have been summarized as: central neurological changes (e.g, post-surgical, post-injury brain lesion, seizuredisorder), peripheral neurological changes (e.g, pelvic nerve hypersensitivity or entrapment), vascular changes (e.g, pelvic congestion or dilatation, or vascular pathology associated with chronic fatigue syndrome, mechanical pressure against genital structures, medication-induced changes (e.g. upon either initiation or cessation of SSRI or mood stabilizer therapy), psychological factors, (e.g. severe stress, the perception and persistence of symptoms, anxiety and obsessive vigilance about physical symptoms, overall lower levels of sexual satisfaction, lower desire and greater pain), beginning menopause, physical inactivity [12-14].

There are no recommended treatment guidelines to treat the disorder due to the lack of clear understanding of the etiology and mechanisms. Pharmacologic strategies have included use of antidepressants, olanzapine, risperidone, anti-seizure medications (e.g. carbamazepine), use of the opioid agonist tramadol, and use of varenicline (a partial agonist at the nicotinic receptor subtype that decreases the ability of nicotine to stimulate the release of mesolimbic dopamine) [15,16]. Although the proposed treatments do not completely eliminate the condition, they may help reduce pain, stress, and discomfort. It was hypothesized that our reported PGAD may be associated with stress as precipitating as well as perpetuating factor. The risperidone was applied to block dopamine receptor in infundibular pathway as the prolactin has a role in maintaining the refractory and relief phase after orgasm. The carbamazepine was also advised because it was assumed that carbamazepine would be beneficial for modulation of overexcited neurons. The psychological approach was psychoeducation regarding symptom and reassurance, improving stress coping ability and relaxation training.

Conclusion

Lack of recommended guidelines and evidences regarding epidemiology, etiology, symptom profile, diagnostic criteria, control trials, management guidelines and prognosis makes difficulties for the clinicians. Holistic approach comprised of Psychological support, Pharmacological acceptance as well as environmental addressing is needed to handle the clients. These observations warrant further investigation of risperidone and carbamazepine in PGAD as well as larger scale control trials needed to establish the strong evidence, which will make comfortable the clinicians as well as the patients.

Abbreviations

PGAD: Persistent Genital Arousal Disorder; MRI: Magnetic Resonance Imaging; PSAS: Persistent Sexual Arousal Syndrome; OPD: Out Patient Department; PSC: Psychiatric Sex Clinic; BSMMU: Bangabandhu Sheikh Mujib Medical University.

Acknowledgements

Authors acknowledges Psychiatric Sex Clinic (PSC) of BSMMU and the Department of Psychiatry BSMMU.

References

1. Simone Eibye, Hans Mørch Jensen (2014) Persistent Genital Arousal Disorder: Confluent Patient History of Agitated Depression, Paroxetine Cessation, and a Tarlov Cyst. Case Reports in Psychiatry 2014: 4.
2. Amsterdam A, Abu-Rustum N, Carter J, Krychman M (2005) “Persistent sexual arousal syndrome associated with increased soy intake. Journal of Sexual Medicine 2: 338-340.
3. Goldmeier D, Leiblum S (2008) Interaction of organic and psychological factors in persistent genital arousal disorder in women: a report of six cases. International Journal of STD & AIDS 19: 488-490.
4. Korda JB, Pfaus JG, Kellner CH, Goldstein I (2009) Persistent Genital Arousal Disorder (PGAD): case report of long-term symptomatic management with electroconvulsive therapy. Journal of Sexual Medicine 6: 2901–2909.
5. Yero SA, McKinney T, Petrides G, Goldstein I, Kellner CH (2006) Successful use of electroconvulsive therapy in 2 cases of persistent sexual arousal syndrome and bipolar disorder. Journal of ECT 22: 274-275.
6. Sadok BJ, Sadok VA, Ruiz P (2015) Synopsis of Psychiatry. (11th edn), Wolters Kluwer.
7. Goldmeier D, Mears A, Hiller J, Crowley T (2009) Persistent genital arousal disorder: a review of the literature and recommendations for management. International Journal of STD & AIDS 20: 373-377.
8. Leiblum SR, Nathan SG (2001) Persistent sexual arousal syndrome: a newly discovered pattern of female sexuality. Journal of Sex and Marital Therapy 27: 365-380.
9. Leiblum SR, Nathan S (2002) Persistent sexual arousal syndrome in women: A not uncommon but little recognized complaint. Sex Rel Ther 17: 191-198.
10. Leiblum SR (2003) Arousal disorders in women: complaints and complexities. Med J Aust 178: 638-640.
11. Leiblum SR (2006) Persistent genital arousal disorder: Whatit is and what it is not. Contemporary Sexuality.
12. Khan F, Spence V, Kennedy G, Belch JJ (2003) Prolonged acetylcholine-induced vasodilatation in the peripheral microcirculation of patients with chronic fatigue syndrome. Clin Physiol Funct Imaging 23: 282-285.
13. Clayton A, Kornstein S, Prakash A, Mallinckrodt C, Wohlreich M (2007) Changes in sexual functioning associated with duloxetine, escitalopram, and placebo in the treatment of patients with major depressive disorder. J Sex Med 4: 917-929
14. Giuliano F, Rampin O, Allard J (2002) Neurophysiology and pharmacology of female genital sexual response. J SexMarital Ther 28 Suppl 1: 101-121.
15. Philippsohn S, Kruger TH (2012) Persistent genital arousal disorder: successful treatment with duloxetine and pregabalin in two cases. J Sex Med 9: 213-217.
16. Korda JB, Pfaus JG, Goldstein I (2009) Persistent Genital Arousal Disorder: ACase Report in a Woman with Lifelong PGAD Where Serendipitous Administration of Varenicline Tartrate Resulted in Symptomatic Improvement. J Sex Med 6: 1479-1486.