Therapeutic Efficacy of Ultrasound-Guided High-Voltage Long-Duration Pulsed Radiofrequency for Pudendal Neuralgia.

Pudendal neuralgia (PN) is a complex disease with various clinical characteristics, and there is no treatment showing definite effectiveness. This study is aimed at evaluating the clinical efficacy of ultrasound-guided high-voltage long-duration pulsed radiofrequency (PRF) for PN. Two cadavers (one male, one female) were dissected to provide evidence for localization of the pudendal nerve. Patients diagnosed as PN who failed or were intolerant in regular medication were screened for diagnostic local anesthesia block of the pudendal nerve before recruitment. Twenty PN patients were enrolled in this study. In the PRF procedure, the needle tip was inserted medially into the internal pudendal artery under ultrasound guidance. The position of the PRF needle tip was then adjusted by the response of the pudendal nerve to the electrical stimulation within the pudendal area (42°C, a series of 2 Hz, and 20 ms width pulses that lasted for 900 s). Alleviation of pain was assessed by the visual analogue scale (VAS) and sitting time pretreatment and on 7 d, 14 d, 1 m, 2 m, 3 m, and 6 m posttreatment in outpatient follow-up or by telephone interview. Two patients were lost due to intervention-irrelevant reasons. Patients showed significantly decreased VAS scores on 7 d after RFP, compared with pretreatment status (7.0 ± 0.9 vs. 3.2 ± 1.7, P < 0.001). The efficacy remained steady till the end of 6 months, with a final remission rate of 88.9%. Sitting time also significantly lengthened following PRF (7 d, 14 d, 1 m, 2 m, 3 m, and 6 m vs. pretreatment, all P < 0.05). Only short-term ipsilateral involuntary convulsion of the lower extremity was reported in one patient, who recovered within 12 h. Six patients were treated with nonsteroidal drugs for a short time. All patients stopped taking medication finally. In conclusion, the ultrasound-guided high-voltage long-duration PRF approach not only reduced the pelvic pain caused by PN but also improved the quality of life by extending sitting time without nerve injury.

Post-traumatic double crush pudendal nerve entrapment syndrome after fracture of the pelvis: A case report.

Pudendal syndrome is primarily characterized by stress urinary incontinence, dysuria, sexual arousal syndrome, painful erections, and anal incontinence. The syndrome occurs when the pudendal nerve or one of its branches is compressed, stretched, or injured. Double crush is the compression of a peripheral nerve at two or more separate areas with various signs and symptoms. We, herein, aimed to introduce the case of a 42-year-old male who underwent the distal release procedure due to the diagnosis of "double crush pudendal syndrome" following a proximal release surgery previously performed elsewhere. The patient's history revealed a pelvic fracture with urethral injury 27 years ago. Throughout the years, the patient had been evaluated by various medical disciplines and undergone several treatments. In 2017, an orthopedic surgeon performed proximal pudendal nerve release using transgluteal approach, and then rectal pain and defecation complaints relieved. However, in 2019, the patient was referred to our clinic because of the persistence of erection and perineal complaints after the proximal pudendal nerve release. Based on a detailed clinical and laboratory assessment, the diagnosis of double crush neuropathy was established, and distal release of the pudendal nerve using transperineal approach was performed. To determine the efficiency of the surgical treatment, International Index of Erectile Function (IIEF) and Quality of Erection Questionnaire (QAQ) tests were used preoperatively and at the first postoperative year. Furthermore, to assess the perineal pain, erection pain, and pain during intercourse Visual Analog Scale (VAS) was used. The erectile dysfunction improved from the severe degree (9 points) to the mild degree (22 points) postoperatively. The patient's general and sexual satisfaction scores, and erection quality score improved compared to the preoperative baseline. According to VAS, the perineal pain, erectile pain,and pain during intercourse decreased postoperatively. (from 7 to 2 out of 10, from 8 to 3 out of 10, from 7 to 2 out of 10, respectively). When perineal and sexual complaints are encountered following pelvic trauma, the pudendal nerve-related problems, especially double crush syndrome, should be kept in mind in differential diagnosis. A multidisciplinary approach must be established in order to avoid any delay in diagnosis and treatment. Surgical intervention may provide a significant improvement in clinical and functional status.

Estimate of the proportion of uncertain diagnoses of pudendal neuralgia in women with chronic pelvic-perineal pain: A systematic review with a descriptive data synthesis.

BACKGROUND: There is a gap between pudendal neuralgia (PN) due to pudendal entrapment syndrome and PN without pudendal entrapment syndrome. The latter could have atypical symptoms. AIM: Defining a rate of atypical PN from a clinical series of female patients with chronic pelvic-perineal pain. METHODS: The atypical PN was defined as a pain not meeting clinical criteria for pudendal entrapment syndrome. The effect size was the rate of atypical PN. Such a rate was expected to be found among patients screened for enrollment in clinical series on pudendal neuropathic pain. A systematic search was performed looking for clinical series on PN. Studies must report information on female patients, pelvic-perineal pain, at least a clinical criterion for diagnosing the pudendal neurogenic origin of pain, the proportion of patients with pain not meeting the clinical criterion/a for diagnosing the pudendal entrapment pain. RESULTS: From 2637 references, nine studies were included for qualitative analysis. Three of them were not suitable for data synthesis: one assessed the rate of PN after hip arthroscopy; second enrolled miscellaneous patients, a third investigated patients with gynecological diseases. Six studies involved patients with suspicion of pudendal entrapment symptoms (205 patients observed), allowing data synthesis. One of these series was judged as being of good quality. The overall rate of atypical PN is 0.013 (95% confidence interval, 0.008-0.021), I2 0%. Further analysis suggests the risk of bias for all studies. CONCLUSIONS: Atypical PN in females is low when clinical criteria for pudendal entrapment syndrome are applied.

A minimally invasive, endoscopic transgluteal procedure for pudendal nerve and inferior cluneal nerve neurolysis in case of entrapment: 3- and 6-month results. The ENTRAMI technique for neurolysis.

BACKGROUND: Pudendal and cluneal nerve entrapment can cause a neuropathic pain syndrome in the sensitive areas innervated by these nerves. Recently, a new endoscopic minimal invasive approach for pudendal and inferior cluneal nerve neurolysis has been published in a cadaver study. The aim of our study was to describe the feasibility of this new approach and to evaluate the clinical outcome. METHODS: Fifteen patients underwent the ENTRAMI technique. The Numeric Pain Rating Scale (NPRS) and Patient Global Impression of Change (PGIC) were recorded at baseline and at 3 and 6 months after surgery. RESULT: The average duration of intervention (skin to skin) was 139 min (range 50-270 min) for bilateral pudendal neurolysis and/or cluneal neurolysis and 113 min (range 100-130 min) for unilateral pudendal and/or cluneal neurolysis. No perioperative blood loss occurred. At 3 months, 50% of patients declared a more than 30% improvement of their PGIC, increasing to 57% at 6 months; 31% reported more than 90% improvement of PGIC at 6 months. Overall reduction of the average maximal NPRS score was from 9 (range 7-10) to 6 at 3 months (range 0-10; p value < 0.05) and to 5 at 6 months (range 0-10; p value < 0.05). There were no postoperative complications. CONCLUSIONS: The ENTRAMI technique is feasibly in patients suffering from pudendal and/or cluneal neuralgia and preliminary results are promising. CLINICAL TRIAL NUMBER: NCT03883178.

Neurophysiologic study in idiopathic overactive bladder.

AIM: Idiopathic overactive bladder (OAB) is a prevalent, mystifying disorder with a questionable neurogenic background. We aimed to investigate the possible subtle neuropathic affection underlying its pathogenesis. METHODS: A cross-sectional cut off study was carried out on a series of 38 females with idiopathic OAB and 22 healthy matched female volunteers. The following was performed: symptom score questionnaire, determination of pudendal nerve terminal motor latency (PNTML), sacral reflexes' latencies, pudendal somatosensory evoked potentials, and needle electromyography of the external anal and urethral sphincters. RESULTS: A highly significant prolongation of PNTMLs and sacral reflexes latencies among the patients group was detected (P ≥ 0.001). Pudendal somatosensory evoked potentials showed non- significance among the two studied groups (P ≥ 0.05). External anal sphincter neuropathic affection was detected in 27 patients (71%) and external urethral sphincter neuropathic affection was detected in 30 patients (78.9%). The clitoral anal reflex showed the highest sensitivity and specificity among the neurophysiologic tests used in assessing the neuropathic affection (86.7 and 83%, respectively), followed by PNTML (83.3 and 80%, respectively). CONCLUSION: Pudendal neuropathy is the dominating possible attributing factor in the pathogenesis underlying idiopathic OAB. An integrated clinical, urodynamic, and electro-physiological assessment is recommended for evaluation of any overactive bladder patients.

Neuroactive steroid levels and psychiatric and andrological features in post-finasteride patients.

Recent reports show that, in patients treated with finasteride for male pattern hair loss, persistent side effects including sexual side effects, depression, anxiety and cognitive complaints may occur. We here explored the psychiatric and andrological features of patients affected by post-finasteride syndrome (PFS) and verified whether the cerebrospinal fluid (CSF) and plasma levels of neuroactive steroids (i.e., important regulators of nervous function) are modified. We found that eight out of sixteen PFS male patients considered suffered from a DSM-IV major depressive disorder (MDD). In addition, all PFS patients showed erectile dysfunction (ED); in particular, ten patients showed a severe and six a mild-moderate ED. We also reported abnormal somatosensory evoked potentials of the pudendal nerve in PFS patients with severe ED, the first objective evidence of a neuropathy involving peripheral neurogenic control of erection. Testicular volume by ultrasonography was normal in PFS patients. Data obtained on neuroactive steroid levels also indicate interesting features. Indeed, decreased levels of pregnenolone, progesterone and its metabolite (i.e., dihydroprogesterone), dihydrotestosterone and 17beta-estradiol and increased levels of dehydroepiandrosterone, testosterone and 5alpha-androstane-3alpha,17beta-diol were observed in CSF of PFS patients. Neuroactive steroid levels were also altered in plasma of PFS patients, however these changes did not reflect exactly what occurs in CSF. Finally, finasteride did not only affect, as expected, the levels of 5alpha-reduced metabolites of progesterone and testosterone, but also the further metabolites and precursors suggesting that this drug has broad consequence on neuroactive steroid levels of PFS patients.

Altered defaecatory behaviour and faecal incontinence in a video-tracked animal model of pudendal neuropathy.

AIM: The aim was to develop a behavioural animal model of faecal continence and assess the effect of retro-uterine balloon inflation (RBI) injury. RBI in the rat causes pudendal neuropathy, a risk factor for obstetric related faecal incontinence in humans. METHOD: Video-tracking of healthy rats (n = 12) in a cage containing a latrine box was used to monitor their defaecatory behaviour index (DBI) over 2 weeks. The DBI (range 0-1) was devised by dividing the defaecation rate (pellets per hour) outside the latrine by that of the whole cage. A score of 0 indicates all pellets were deposited in the latrine. Subsequently, the effects of RBI (n = 19), sham surgery (n = 4) and colostomy (n = 2) were determined by monitoring the DBI for 2 weeks preoperatively and 3 weeks postoperatively. RESULTS: The DBI for healthy rats was 0.1 ± 0.03 with no significant change over 2 weeks (P = 0.71). In the RBI group, 13 of 19 rats (68%) showed no significant change in DBI postoperatively (0.08 ±  -0.05 vs 0.11 ±  -0.07) while in six rats the DBI increased from 0.16 ±  -0.09 to 0.46 ± 0.23. The negative control, sham surgery, did not significantly affect the DBI (0.09 ± 0.06 vs 0.08 ± 0.04, P = 0.14). The positive control, colostomy, increased the DBI from 0.26 ± 0.03 to 0.86 ± 0.08. CONCLUSIONS: This is the first study showing a quantifiable change in defaecatory behaviour following injury in an animal model. This model of pudendal neuropathy affects continence in 32% of rats and provides a basis for research on interventions for incontinence.

Injury to Perineal Branch of Pudendal Nerve in Women: Outcome from Resection of the Perineal Branches.

Background This study describes outcomes from a new surgical approach to treat "anterior" pudendal nerve symptoms in women by resecting the perineal branches of the pudendal nerve (PBPN). Methods Sixteen consecutive female patients with pain in the labia, vestibule, and perineum, who had positive diagnostic pudendal nerve blocks from 2012 through 2015, are included. The PBPN were resected and implanted into the obturator internus muscle through a paralabial incision. The mean age at surgery was 49.5 years (standard deviation [SD] = 11.6 years) and the mean body mass index was 25.7 (SD = 5.8). Out of the 16 patients, mechanisms of injury were episiotomy in 5 (31%), athletic injury in 4 (25%), vulvar vestibulectomy in 5 (31%), and falls in 2 (13%). Of these 16 patients, 4 (25%) experienced urethral symptoms. Outcome measures included Female Sexual Function Index (FSFI), Vulvar Pain Functional Questionnaire (VQ), and Numeric Pain Rating Scale (NPRS). Results Fourteen patients reported their condition pre- and postoperatively. Mean postoperative follow-up was 15 months. The overall FSFI, and arousal, lubrication, orgasm, satisfaction, and pain domains significantly improved (p < 0.05). The VQ also significantly improved (p < 0.001) in 13 (93%) of 14 patients. The NPRS score decreased on average from 8 to 3 (p < 0.0001). All four patients with urethral symptoms were relieved of these symptoms. Conclusion Resection of the PBPN with implantation of the nerve into the obturator internus muscle significantly reduced pain and improved sexual function in women who sustained injury to the PBPN.

Entrapment of the posterior femoral cutaneous nerve and its inferior cluneal branches: anatomical basis of surgery for inferior cluneal neuralgia.

PURPOSE: The apparent failure of pudendal nerve surgery in some patients has led us to suggest the possibility of entrapment of other adjacent nerve structures, leading to the concept of inferior cluneal neuralgia. Via its numerous collateral branches, the posterior femoral cutaneous nerve innervates a very extensive territory including the posterior surface of the thigh, the infragluteal fold, the skin over the ischial tuberosity, but also the lateral anal region, scrotum or labium majus via its perineal branch. METHODS: We described the pathophysiological features of cluneal neuralgia, the surgical technique and our preliminary results. RESULTS: We performed a transmuscular approach leading to the fat of the deep gluteal region. Exploration was continued cranially underneath the piriformis, looking for potential entrapments affecting the posterior femoral cutaneous nerve and the sciatic nerve. Nerve decompression on the lateral surface of the ischial tuberosity was then performed. A constant anatomical finding must be highlighted: the presence of a lateral fibrous expansion from the ischium passing behind the nerves and vessels, especially the posterior femoral cutaneous nerve and its perineal branches. In our patients, release of this expansion allowed decompression of the nerve trapped by this expansion. CONCLUSION: Cluneal neuralgia constitutes a distinct entity of perineal pain, which must be identified and distinguished from pudendal neuralgia. Surgery should be performed via a transgluteal approach. A lateral ischial obstacle must be investigated, in the form of a constant fibrous expansion, which, like a retinaculum, can cause nerve entrapment.

A Novel Electrophysiological Method in the Diagnosis of Pudendal Neuropathy: Position-related Changes in Pudendal Sensory Evoked Potentials.

OBJECTIVE: To examine the diagnostic value of pudendal somatosensory evoked potentials (SEPs) in pudendal nerve entrapment (PNE) neuropathy by stimulating the 2 sides separately after provocation by a standard sitting position. Routine pudendal SEPs performed in the supine position with bilateral simultaneous stimulation may fail to show the abnormality because the complaints of PNE appear or worsen in the sitting position. METHODS: Forty-nine patients with PNE and 16 controls were included. SEP recordings were performed by stimulating the dorsal nerve of penis or clitoris on either side. The recordings were performed at the initial supine position, at the beginning and end of the provocation by sitting position, and at the second supine position. RESULTS: Amplitude loss in the SEP responses after prolonged sitting was significantly more pronounced on the symptomatic sides of the patients. Approximately 45% decrease in the SEP amplitude or an amplitude value less than 1.5 µV at the end of sitting are the parameters to be used with high selectivity. CONCLUSION: The dynamic pudendal SEP study described herein seems to be more helpful in PNE diagnosis than in conventional SEPs.

Pudendal Neuropathy Alone Results in Urge Incontinence Rather Than in Complete Fecal Incontinence.

BACKGROUND: Conscious external anal sphincter contraction is mediated by the pudendal nerve. Pudendal neuropathy is, therefore, believed to result in fecal incontinence. Until urge sensation is experienced, fecal continence is maintained by unconscious external anal sphincter contraction, which is regulated by the anal-external sphincter continence reflex. The innervation of unconscious contraction is yet unknown. OBJECTIVE: We aimed to determine whether unconscious contraction is mediated by the pudendal nerve and whether age influences unconscious contraction. DESIGN: This was a retrospective comparative study. SETTINGS: The study was conducted in a tertiary care center. PATIENTS: Seventy adult patients experiencing defecation problems who underwent anorectal function tests were included in this study. MAIN OUTCOME MEASURES: Conscious and unconscious contractions were compared between patients with and without pudendal neuropathy. Conscious contraction was defined by maximum anal sphincter contractility, unconscious contraction by pressure in the anal canal at maximum tolerable or retainable sensation during the balloon retention test. RESULTS: Unconscious contraction did not differ significantly between patients with pudendal neuropathy and non-pudendal neuropathy patients, whereas conscious contraction was significantly lower in patients with pudendal neuropathy. Multiple linear regression analyses demonstrated that unconscious contraction, in contrast to conscious contraction, was not predicted significantly by age and anal electrosensitivity at 2 cm, which represents pudendal neuropathy. Patients with pudendal neuropathy were significantly older than patients with nonpudendal neuropathy. LIMITATIONS: The pudendal nerve motor latency and EMG tests were not performed. CONCLUSIONS: The pudendal nerve does not mediate unconscious external anal sphincter contraction. Pudendal neuropathy alone, therefore, results in urge incontinence rather than in complete fecal incontinence. Unconscious contraction appears not to be influenced by age. Therefore, most of the elderly patients experience urge incontinence rather than complete fecal incontinence.

Spinal cord stimulation of the conus medullaris for refractory pudendal neuralgia: a prospective study of 27 consecutive cases.

AIMS: Thirty percent of patients with pudendal neuralgia due to pudendal nerve entrapment obtain little or no relief from nerve decompression surgery. The objective was to describe the efficacy of spinal cord stimulation of the conus medullaris in patients with refractory pudendal neuralgia. METHODS: This prospective study, conducted by two centers in the same university city, described the results obtained on perineal pain and functional disability in all patients with an implanted conus medullaris stimulation electrode for the treatment of refractory pudendal neuralgia. Twenty-seven consecutive patients were included by a multidisciplinary pelvis and perineal pain clinic between May 2011 and July 2012. Mean follow-up was 15 months. The intervention was an insertion of a stimulation electrode was followed by a test period (lasting an average of 13 days) before deciding on permanent electrode implantation. Maximum and average perineal pain scores and the pain-free sitting time were initially compared during the test and in the long-term (paired t-test). The estimated percent improvement (EPI) was evaluated in the long-term. RESULTS: Twenty of the 27 patients were considered to be responders to spinal cord stimulation and 100% of implanted patients remained long-term responders (mean tripling of sitting time, and mean EPI of 55.5%). CONCLUSIONS: Spinal cord stimulation of the conus medullaris is a safe and effective technique for long-term treatment of refractory pudendal neuralgia. Routine use of this technique, which has never been previously reported in the literature in this type of patient, must now be validated by a larger scale study.

Pudendal neuralgia.

Pudendal neuralgia is a painful condition affecting the nerve distribution of the pudendal nerve. The Nantes criteria give some structure for making this diagnosis. A step-ladder approach to therapy, as described, is suggested when treating these patients.

Chronic perineal pain: analyses of prognostic factors in pudendal neuralgia.

OBJECTIVES: To establish the prognostic factors and outcomes of patients with pudendal neuralgia (PN). MATERIALS AND METHODS: A total of 51 patients with PN treated at the Puerta de Hierro University Hospital of Madrid between January 2011 and June 2012 were included in this study. Patients were compared in relation to pain intensity and response to the first-line treatment of neuropathic pain. Univariate and multivariable Cox regression analyses were used. RESULTS: The median for patients' age, duration, and intensity of pain evaluated by visual analogue scale were 40.9, 3.6, and 7.6 years, respectively. Among 45 patients, there were 19 good responders to first-line treatment for neuropathic pain and 26 nonresponders. The 19 responders measured their improvement at 47%. Tramadol was used for nonresponding patients, 30.8% of whom expressed a 35% improvement. Analysis of 45 patients with PN demonstrated that the pain intensity was associated with dorsal clitoris nerve damage (15.4% vs. 52.2%; P=0.035; odds ratio 4.5; 95% confidence interval [CI], 1.11-18.1) and with sensory deficit at the S2-S4 dermatome map (57.7% vs. 87%; P=0.05; odds ratio 3.7; 95% CI, 0.80-16.8). The pain located at the dorsal clitoris nerve was a significant prognostic factor for having no response to the first-line treatment of neuropathic pain (28% vs. 53%; P=0.033; odds ratio 4.5; 95% CI, 1.06-19.6). DISCUSSION: A mixed analgesic ladder for chronic pain showed improvement in 73% of the patients with PN. Pain restricted to the dorsal clitoris nerve and small fibers in the S2-S4 dermatome were classified as bad prognostic factors. A longer duration of pain was also correlated with a worse prognosis.

Response to pudendal nerve block in women with pudendal neuralgia.

OBJECTIVE: To examine the evolution of pain and the duration of numbness after neural blockade of the pudendal nerve in women with pudendal neuralgia and correlate with clinical and historical data. DESIGN: Prospective, single arm, open label study. SETTING: University hospital and outpatient clinic. SUBJECTS: Eighty-two adult female patients were recruited from November 8, 2008 to February 14, 2010. Patients were selected based on the presence of spontaneous or provoked pain in the distribution of the pudendal nerve. INTERVENTIONS: Subjects underwent a standardized pudendal nerve block. OUTCOME MEASURES: Visual analog pain scores and the presence of numbness were recorded before and for 64 hours after the pudendal nerve block. A complete clinical history and examination were documented. RESULTS: Sixty-six patients completed the study. About 86.9% had a reduction in one or more pain symptom, while 44.3% found that more than one of their pain symptoms did not return. About 69.7% of patients reported numbness lasting up to 16 hours or longer. Previous gynecological surgery was recorded in 75.8%, previous traumatic obstetric events in 47.0% of cases. Prolonged history of pain correlated with a reduced chance of positive outcome of the pudendal nerve block. CONCLUSION: In patients with pudendal neuralgia, the pudendal nerve block has a variable response, but may have a beneficial effect in a subset of women. Surgical and obstetrical trauma are common historical antecedents.

Chronic proctalgia and chronic pelvic pain syndromes: new etiologic insights and treatment options.

This systematic review addresses the pathophysiology, diagnostic evaluation, and treatment of several chronic pain syndromes affecting the pelvic organs: chronic proctalgia, coccygodynia, pudendal neuralgia, and chronic pelvic pain. Chronic or recurrent pain in the anal canal, rectum, or other pelvic organs occurs in 7% to 24% of the population and is associated with impaired quality of life and high health care costs. However, these pain syndromes are poorly understood, with little research evidence available to guide their diagnosis and treatment. This situation appears to be changing: a recently published large randomized, controlled trial by our group comparing biofeedback, electrogalvanic stimulation, and massage for the treatment of chronic proctalgia has shown success rates of 85% for biofeedback when patients are selected based on physical examination evidence of tenderness in response to traction on the levator ani muscle--a physical sign suggestive of striated muscle tension. Excessive tension (spasm) in the striated muscles of the pelvic floor appears to be common to most of the pelvic pain syndromes. This suggests the possibility that similar approaches to diagnostic assessment and treatment may improve outcomes in other pelvic pain disorders.