Pelvic floor physical therapy in the treatment of a patient with persistent genital arousal disorder/genito-pelvic dysesthesia: a case report

Abstract

Introduction

Persistent genital arousal disorder/genito-pelvic dysesthesia (PGAD/GPD) is a condition of persistent or recurrent, unwanted or intrusive sensation of genital arousal that is usually associated with a distressing feeling and has a great impact on patients’ daily life. Pelvic floor physical therapy is one of the effective conservative treatment options that deserves increased attention.

Aims

This case report aims to provide a comprehensive pelvic floor physical therapy evaluation and treatment plans for a patient with PGAD/GPD that resulted in a complete resolution of symptoms.

Methods

The patient is a 63-year-old female who suffered from persistent and uncontrolled sexual arousal over 3 months. Despite her efforts, the symptoms gradually worsened and never completely resolved, resulting in significant distress and despair. Her examination findings include myofascial restrictions on superficial pelvic floor structures, hypertonic pelvic floor muscles with trigger points, radiating pain along with genitofemoral nerve innervated areas, and lack of pelvic floor muscle strength and coordination. Physical therapy treatment plan included patient education, manual therapy, muscle strengthening exercises, and home exercise programs.

Results

The patient’s symptoms were completely resolved after a total of four pelvic floor physical therapy sessions. Home exercise program was able to maintain satisfactory treatment outcomes 3 months post treatment. No complaints were reported at the 6-month and 1-year follow-ups.

Conclusion

Genitofemoral nerve pathology may be a potential etiology for PGAD/GPD. Pelvic floor physical therapy can an effective medical treatment for PGAD/GPD originating from the pelvic and perineum region. A comprehensive pelvic floor examination and an evidence-based treatment plan will be able to improve symptoms and potentially resolve them completely.

Introduction

Persistent genital arousal disorder/genito-pelvic dysesthesia (PGAD/GPD) is a medical condition presenting with persistent or recurrent genital arousal in the clitoris or other genito-pelvic regions.¹ The symptoms are not associated with concomitant sexual interest, thoughts or fantasies. PGAD/GPD may highly impact patients’ daily social life and mental health.² Previous studies reported that patients may feel frustrated, miserable, embarrassed with suicidal ideation estimated up to 54%.³ International Society for the Study of Women’s Sexual Health (ISSWSH) presented five pathological regions to introduce a new diagnostic algorithm to identify PGAD/GPD including vaginal end organs, pelvis/perineum, cauda equina, spinal cord pathology, and brain.¹

Owing to the complexity of pathophysiology of PGAD/GPD, the ISSWSH expert panels highly recommended a multidisciplinary management of this medical condition. Pelvic floor physical therapy is considered a standard treatment for PGAD/GPD patients with pelvis/perinium origin.¹ A thorough assessment and a success to identify pelvic floor dysfunction will guide pelvic floor treatment interventions. A comprehensive pelvic floor physical therapy evaluation include both intra- and extra-pelvic assessments.⁴ The symptom-related pelvic floor dysfunction includes but not limited to overactive/hypertonic pelvic floor muscles, soft tissue trigger points, pudendal nerve neuropathy, and abdominal wall afferent nerve neuropathy. The treatment consists of patient education, manual therapy, therapeutic exercises, and neuromuscular re-education targeted at specific pelvic floor dysfunctions.⁵ The purpose of this case report is to present a comprehensive pelvic floor physical therapy evaluation of PGAD/GPD and provide detailed pelvic floor physical therapy intervention plans to achieve a complete resolution of symptoms.

Method

Case description

The patient was a 63-year-old female who was seen at the Department of Obstetrics and Gynecology on September 25, 2023. She had a total vaginal hysterectomy in 2005 due to uterine prolapse and a tension-free vaginal tape procedure in 2010 due to urinary incontinence. No symptoms of prolapse and incontinence were reported after surgeries. Her main complaint was “persistent and uncontrolled sexual arousal in the past 3 months”. Following prolonged periods of sitting or standing, she experienced sensations akin to being on the verge of orgasm, accompanied by increased clitoral temperature and heaviness in her vulvar region. Despite attempting masturbation twice to alleviate her symptoms, she found no relief. Instead, she predominantly chose to lie down but her symptoms persisted without resolution. She achieved the highest score in the desire domain of the Female Sexual Function Index (FSFI), indicating “almost always or always” and “very high” levels of sexual desire or interest over the past 4 weeks. She scored zero points in other domains of FSFI as she has not engaged in sexual activities for a couple of years after her first pelvic surgery. Symptoms, coupled with significant emotional distress, had a detrimental effect on her mental health and substantially restricted her social interactions with family and friends, which were a vital part of her retirement life. Although the patient reported adequate emotional and financial support from her family, she became visibly upset during subjective intake, expressing embarrassment about discussing her condition. She denied experiencing symptoms associated with restless legs or urogenital issues. The patient applied vaginal estrogen twice weekly post hysterectomy. She denied having attempted any other treatments prior to or following this medical visit.

Examination

A complete pelvic floor physical therapy assessment was performed in accordance with published guidelines for pelvic floor physical therapy assessment.⁴ Several special tests were performed to rule out the possibility of lumbar neurological disease, including the straight leg raise test and slump test. The cauda equina syndrome was ruled out with sensory assessments in the saddle area, L1-S1 myotome tests, anal/Achilles reflex tests, and a subjective intake of bladder and bowel functions. Additionally, a magnetic resonance imaging scan of the pelvis was performed to rule out a sacral Tarlov cyst. All tests revealed no red flags or other positive results, with all findings within normal limits. Details of musculoskeletal physical therapy assessments were shown in Supplementary Table S1.

The pelvic floor physical therapy examination included assessments of external perineum and internal pelvic floor structures. Skin integrity, erythema, edema of the vulvar area was observed in lithotomy position. By palpating the vulvar, clitoris, and vestibular areas, a sensation test was conducted. Tenderness and myofascial restrictions were noted during palpation of the superficial external pelvic floor muscles.

A comprehensive internal vaginal assessment includes the assessment of deep pelvic floor muscle tone, the evaluation of myofascial tenderness along with trigger points, and the evaluation of pelvic floor muscle strength using the Modified Oxford Grading Scale (MOGS).⁶ The patient was in lithotomy position and asked to perform Valsalva maneuver as the therapist placed 2 digits to observe for anterior or posterior vaginal wall bulging.⁷ Pelvic organ prolapse quantification system (POP-Q) as a clinical standardized measurement was utilized for POP stage grading. Pelvic floor physical therapy examination results at each visit were presented in Table 1.

Table 1.

Pelvic floor physical therapy examination results.

Pelvic Floor Examination Findings	Initial Visit	Second Visit	Third Visit	Final Visit
Observation	Dry skin with slight erythema, especially on left majora labia was noted. Slight engorgement on bilateral labia majora areas. The glans clitoris was visible and firm.	Improved skin integrity with decreased erythema noted. Improved engorgement and mobility of glans clitoris.	No dryness and erythema noted. No engorgement with improved mobility of glans clitoris.	No dryness and erythema noted. No engorgement with improved mobility of glans clitoris.
External Sensationa	Burning sensation around clitoris and vestibule area (2/10). Unconscious sexual arousal usually begins within vestibular areas (2/10) and second layer of pelvic floor muscle regions (5-6/10); however, that was not reproduced by palpation. Radiation sensation along genital-femoral nerve innervated region (1/3 anterior inner side of thigh) aroused by palpation on either side of second-layer pelvic floor muscles. Left discomfort is stronger than right.	Continued abnormal sensation noted around second-layer pelvic floor muscle (2-3/10); however, no radiation sensation reproduced during palpation on anterior inner thigh regions. Ipsilateral pain (2/10) noted on lower abdominal regions between two anterior superior iliac spines.	Improved soft tissue movement noted around clitoris with decreased abnormal sensation rating (1-2/10) during palpation on second-layer pelvic floor muscles. No radiation noted on either lower abdominal or anterior inner thigh regions.	No abnormal sensation, discomfort or muscle tenderness noted during palpation. No radiation noted on lower abdominal or anterior inner thigh regions.
Internal Sensationa	Bilateral deep pelvic floor muscle pain noted during internal palpation (Left: pubococcygeus 1/10, iliococcygeus 4-5/10, obturator internus 4-5/10; Right: iliococcygeus 3-4/10)	Improved bilateral deep pelvic floor muscle pain (Left: iliococcygeus 3-4/10, obturator internus 3-4/10). No pain reported on right deep pelvic floor muscles.	Only slight discomfort noted on left pelvic floor muscles (iliococcygeus 1-2/10, obturator internus 1-2/10)	Only slight discomfort on left obturator internus (1-2/10)
Superficial Pelvic Floor Muscle Tone	Bilateral myofascial restriction around clitoris. Palpable tenderness noted on left bulbocavernosus	Decreased myofascial restriction around clitoris. Improvement noted as tenderness relieved on left bulbocavernosus; however, trigger points palpated around perineal body area	Improved soft tissue movement with no myofascial restrictions noted around clitoris. Continued minimal tenderness and trigger points palpated on left bulbocavernosus around perineal body area	Normal muscle tone palpated with no discomfort reported on superficial pelvic floor muscles
Deep Pelvic Floor Muscle Tone	Increased muscle tone with moderate spasm noted	Slightly increased muscle tone with minimal to moderate muscle spasm	Improved muscle tone with no muscle spasm	Improved muscle tone with no muscle spasm
Pelvic Floor Muscle Strength/Relaxation/Coordination	Muscle power: 3/5 with evident bilateral shoulder shrugs as compensation strategies. Muscle endurance: 5 s with holding breaths. Decreased pelvic floor proprioception with incomplete relaxation noted after each contraction.	Muscle power: 1/5 with no visible compensation from other muscles. Muscle endurance: 2 s with no breath holding. Improved muscle proprioception and relaxation.	Muscle power: 1/5 with no compensation. Muscle endurance: 2-3 s with no breath holding. Complete muscle relaxation noted after each contraction.	Muscle power: 2/5 with no compensation. Muscle endurance: 4-5 s. Complete muscle relaxation with improved pelvic floor proprioception.

^aNumerical Rating Scale of Pelvic Floor Muscle Pain during Palpation at a rating from 0 to 10.

Assessment

Clinical impression 1—abnormal sensation with nerve entrapment

The patient’s impairments could be attributed to a possible entrapment of the pudendal nerve, which presented burning sensations on the clitoris and majora labia, palpable deep pelvic floor muscle tenderness and pain, and a lack of strength and coordination in the pelvic floor muscles.

Another hypothesis was the patient also had genitofemoral nerve impairment, which aligns with her radiating pain on the 1/3 anterior inner side of her thigh during the palpation test on superficial pelvic floor muscles.

Clinical impression 2—unconscious sexual arousal with hyperactive anatomic structures

During palpation tests on superficial pelvic floor structures, bilateral myofascial restriction around the clitoris and palpable tenderness on the bulbocavernosus were noted. As a result of restricted soft tissue structures, in particular a hyperactive bulbocavernosus, the clitoris is continuously irritated and may contribute to the unconscious sexual arousal experienced by the patient. A hypertonic bulbocavernosus may compress the deep dorsal vein of the clitoris, preventing or retarding vein outflow, leading to sexual dysfunction symptoms.

Intervention

Physical therapy interventions included patient education, manual therapy, pelvic floor muscle strengthening, abdominal strengthening exercises, and individualized home exercise programs.

At the evaluation and throughout the intervention period, patient education on the following was provided: (1) pelvic floor anatomy; (2) pelvic floor neurology and physiology; (3) pain education and self-management strategies; (4) application of coconut oil for vaginal tissue moisture and dryness prevention; (5) correct sitting and standing posture education; (6) use of footstool to increase hip flexion ≥90° during seated toileting, allowing anal-rectal angle straight to reduce straining during bowel movement.

Interventions were determined based on the findings of the pelvic floor evaluation. Mobilization was applied to the second layer of pelvic floor muscles behind the mons pubic area to centralize the symptoms aroused by irritate genito-femoral nerve. To release pudendal nerve entrapment, mobilization of the inguinal canal and internal muscle release on the obturator internus were performed. Furthermore, soft tissue release was applied to the clitoris and bilateral bulbocavernosus to relieve superficial restrictions. Pelvic floor muscle strengthening exercises began with diaphragmatic breathing techniques with internal tactile cues to improve the patient’s proprioception, decrease muscle tone, and facilitate complete muscle relaxation. Pelvic floor muscle power and endurance exercises with the control of compensation strategies were prescribed with progression through intervention visits as shown in Table 2. Additional strengthening exercises included supine abdominal exercises to increase the stability of the pelvic girdle and maintain correct posture. Detailed home exercise programs with progressions are outlined in Table 2.

Table 2.

Pelvic floor physical therapy intervention program.

Week	Education	Manual Therapy	Pelvic Floor Muscle (PFM) Exercises	Other Strengthening Exercise	Home Exercise Program
1	Pelvic floor anatomy education. Sitting/Standing posture. Pain education. Coconut oil to Moisture vaginal tissue. Superficial self-pelvic floor release.	Soft tissue release on clitoris and bilateral bulbocavernosus. Internal deep pelvic floor muscle release techniques. Tactile PFM facilitation for full relaxation	Diaphragmatic breathing exercises on PFM proprioception facilitation: 10 mins	None	Meditation: 10 mins per day. Diaphragmatic Breathing Exercises: 10 mins per day. Self-muscle release on superficial pelvic floor muscles: 5-10 mins per day
2	Pain education. Posture education. Discussed internal self-pelvic floor release.	Trigger points release on left bulbocavernosus and perineal body. Mobilization on inguinal canal Internal deep pelvic floor muscle release. Tactile PFM cues and facilitations for contraction and relaxation	Diaphragmatic breathing: 10 mins. Supine PFM quick contractions: 2 sets of 10 reps. Supine PFM contractions holding for 2 s: 2 sets of 10 reps.	Transverse abdominis activation exercise: 2 sets of 10 reps	Meditation: 10 mins per day. Diaphragmatic Breathing Exercise: 10 mins per day. Self-muscle release on superficial pelvic floor muscles: 5-10 mins per day. Supine PFM quick contractions: 2 sets of 10 reps, three times per day. Supine PFM contractions holding for 2 s: 2 sets of 10 reps, three times per day.
3	Internal Self-pelvic floor release	Trigger points release on bulbocavernosus. Internal Deep pelvic floor muscle release. Tactile PFM cues and facilitations for full “closure and lift”.	Supine PFM quick contractions: 2 sets of 10 reps. Supine PFM contractions holding for 3 s: 2 sets of 10 reps.	Transverse abdominis activation exercise: 2 sets of 10 reps. Transverse abdominis with alternation 90-90 leg raise: 2 sets of 10 reps	Diaphragmatic Breathing Exercise: 10 mins per day. Supine PFM quick contractions: 2 sets of 10 reps, 3 times per day. Supine PFM contractions holding for 3 s: 2 sets of 10 reps, 3 times per day. Self-superficial and Internal pelvic floor muscle release on obturator internus: 10 mins per day.
4	Discussed continued home exercise program for symptom improvement maintenance and regular follow-ups	Internal Deep pelvic floor muscle release on obturator internus. Tactile PFM cues and facilitations for full “closure and lift”	Supine PFM quick contractions: 2 sets of 10 reps. Supine PFM contractions holding for 3 s: 2 sets of 10 reps.	Transverse abdominis with alternation 90-90 leg raise: 2 sets of 10 reps. Dead bugs holding for 10s: 1 set of 10 reps	Diaphragmatic Breathing Exercise: 10 mins per day. Supine PFM quick contractions: 2 sets of 10 reps, 3 times per day. Supine PFM contractions holding for 3 s: 2 sets of 10 reps, 3 times per day. Self-Superficial and Internal pelvic floor muscle release on obturator internus: 10 mins per day. Core exercises: 2 sets of 10 reps each exercise

Results

The patient attended a total of 4 pelvic floor physical therapy sessions once a week. Her feelings of sexual desire or interest have dropped from “almost always or always” to “almost never or never”. Her rating of sexual desire or interest decreased from “high” to “very low or not at all”. At the second visit after manual therapy, the radiating pain along the genito-femoral nerve innervated region was centralized from 1/3 of the anterior inner side of the thigh to lower abdominal area between two anterior superior iliac spines. The use of manual techniques on both superficial and deep pelvic floor structures led to a reduction in muscle pain and discomfort, an improvement in soft tissue mobility, and a reduction in the engorgement of the glans clitoris during the treatment period. The pelvic floor muscle demonstrated improved proprioception, complete relaxation, and enhanced strength and endurance after physical therapy sessions, although further muscle strengthening is recommended. After four sessions, the patient reported a complete resolution of her symptoms, and the intervention has been terminated at her request. Follow-ups continued at 3 months, 6 months, and 1 year following treatment. At the 3-month follow-up, the patient reported two minor episodes of symptom recurrence, which were both resolved after performing the home exercise program. The patient did not report any complaints at the 6-month and 1-year follow-ups.

Discussion

In this case, we presented a 63-year-old female who experienced uncontrolled and persistent sexual arousal over 3 months. The symptoms met the diagnostic criteria of PGAD/GPD and caused her physical discomfort as well as significant mental distress, severely affecting her quality of life. Complete resolution of her symptoms was achieved through a comprehensive pelvic floor physical therapy evaluation and detailed treatment plans.

There is no doubt that psychosocial factors play a significant role throughout physical therapy sessions. It is important to recognize that the symptoms are possibly influenced to some extent by mental responses; thus, reducing distress and anxiety may be an alleviating factor for symptom relief. An essential component of pelvic floor physical therapy treatment plans is patient education, which starts with a thorough understanding of pelvic anatomical structures, potential pathologies, and introduction to sexual arousal cycles. Self-management strategies have been proven to be effective in reducing her anxiety, such as diaphragmatic breathing and meditation.⁸ It is highly recommended to explain to the patient explicitly that this condition is primarily genital in nature and to divert her hypothesis that she is sexually addicted.

It has been hypothesized that pudendal nerve entrapment is one possible cause of synptoms in this case. Pudendal nerve is a mixed somatic sensory and motor nerve that can cause pelvic dysesthesia and pelvic floor dysfunction. Pudendal nerve entrapment at Alcock’s canal under obturator internus fascia is a well-established reason for pudendal neuralgia and pelvic floor muscle dysfunction.⁹ In this current case, we also hypothesized that the entrapment of one of the abdominal wall nerve occurred, as the patient experienced radiating pain to the anteromedial skin of the ipsilateral thigh. The resolve of her symptoms is accompanied with the centralization of radiating sensation to her suprapubic region near the skin of mons pubis, consistent with our hypothesis of genitofemoral nerve entrapment.¹⁰ This case provides further evidence and supports the hypothesis generated by the ISSWSH panels that the entrapment of abdominal wall nerves has the possibility being one of the etiologies of PGAD/GPD. In order to appropriately evaluate and identify this neurological dysfunction, the health care provider is required to be familiar with neural anatomy and function in the pelvis and perineum region.

Conclusions

The case report substantiates a hypothesis from prior research suggesting that the pathology of abdominal wall nerves, particularly the genitofemoral nerve, may be a potential etiology. This case directly demonstrates the integral role of PFPT within the current multidisciplinary standard of care for PGAD/GPD. It is important to note that the comprehensive PFPT approach offers a unique non-invasive, patient-centered approach that addresses neuro-musculoskeletal drivers of symptoms without involving surgery or pharmaceuticals, thereby causing minimal side effects to patients. This case is critical for validating PFPT’s efficacy in the management of PGAD/GPD, providing sufficient clinical guidelines for fellow practitioners, and advocating for timely PFPT access within PGAD/GPD treatment pathways.

Author contributions

Conceptualization: Lan Zhu, Wenjia Lou.

Data curation: Tangdi Lin.

Formal analysis: Tangdi Lin, Guorong Fan, Lina Niu.

Funding acquisition: Lan Zhu, Wenjia Lou.

Investigation: Tangdi Lin, Guorong Fan, Lina Niu.

Methodology: Tangdi Lin, Wenjia Lou.

Project administration: Wenjia Lou.

Resources: Lan Zhu, Wenjia Lou.

Software: Tangdi Lin.

Supervision: Lan Zhu, Wenjia Lou.

Validation: Tangdi Lin, Guorong Fan, Lina Niu.

Visualization: Tangdi Lin, Guorong Fan, Lina Niu.

Writing—original draft: Tangdi Lin, Guorong Fan, Lina Niu.

Writing—review & editing: Lan Zhu, Wenjia Lou, Tangdi Lin, Guorong Fan, Lina Niu.

Funding

This work was supported by the National Key R&D Program of China (Grant Numbers: 2021YFC2701300, 2021YFC2701303) and National High-Level Hospital Clinical Research Funding (Grant Number: 2022-PUMCH-A-112).

Conflicts of interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Details of ethics approval

The study protocol was approved by the institutional review board of Peking Union Medical College Hospital (Number: K4999). The study protocol was approved and written informed consent was obtained from the patient.