Pudendal nerve entrapment

Pudendal nerve entrapment
Other names
  • Pudendal neuralgia.
  • Alcock canal syndrome.[1][2]
  • Pudendal nerve entrapment syndrome.[3]
  • Pudendal canal syndrome.[4]
  • Pudendal syndrome.[5]
  • Pudendal neuropathy.[5]
  • Cyclist syndrome.[6]
SpecialtyNeurology Edit this on Wikidata

Pudendal nerve entrapment (PNE) is an uncommon chronic pelvic pain condition in which the pudendal nerve (located in the pelvis) is entrapped and compressed. There are several different anatomic locations of potential entrapment (see Anatomy). Pudendal nerve entrapment is an example of nerve compression syndrome.

Pudendal neuralgia (PN) refers to pain along the course of the pudendal nerve and in its distribution. This term is often used interchangeably with pudendal nerve entrapment. However, it has been suggested that the presence of symptoms of pudendal neuralgia alone should not be used to diagnose pudendal nerve entrapment. That is because it is possible to have all the symptoms of pudendal nerve entrapment, as per the diagnostic criteria specified at Nantes in 2006, without actually having an entrapped pudendal nerve.[7] Another study of 13 normal female cadavers found that the pudendal nerve was attached or fixed to the sacrospinous ligament (therefore "entrapped") in all cadavers studied, suggesting that the diagnosis of pudendal nerve entrapment may be overestimated.[8]

The pain is usually located in the perineum, and is worsened by sitting. Other potential symptoms include genital numbness, sexual dysfunction, bladder dysfunction or bowel dysfunction. Pudendal neuralgia can be caused by many factors including nerve compression or stretching of the nerve. Injuries during childbirth, sports such as cycling, chronic constipation and pelvic surgery have all been reported to cause pudendal neuralgia.

Management options include lifestyle adaptations, physical therapy, medications, long acting local anesthetic injections and others. Nerve decompression surgery is usually considered as a last resort. Pudendal neuralgia and pudendal nerve entrapment are generally not well-known by health care providers. This often results misdiagnosis or delayed diagnosis. If the pain is chronic and poorly controlled, pudendal neuralgia can greatly affect a person's quality of life, causing depression.

Terminology

In pain research, there are different types of pain, although these types may be combined in some conditions and in some cases.

  • Nociceptive pain is pain that occurs with activation of nociceptors and arises from actual (or threatened) damage to non-neural tissue. In contrast with neuropathic pain, nociceptive pain involves a normally functioning somatosensory nervous system.[9]
  • Neuropathic pain is due to nerve damage. It is pain caused by a lesion or disease of the somatosensory nervous system.[9] In this type of pain, something is causing the nerve(s) to function abnormally.[9]
  • Nociplastic pain is pain that arises from altered nociception (perception of pain) even though there is no detectable tissue damage causing the activation of peripheral nociceptors, and no evidence for any disease process or lesion affecting the somatosensory nervous system which could explain the pain.[9] Nociplastic pain is a feature of central sensitization, in which peripheral neurons are functioning normally but there is distorted processing of pain signals in the central nervous system. In central sensitization, nociceptive neurons in the central nervous system have increased responsiveness to normal or subthreshold afferent (incoming) input signals.[9]

Several related terms are used in the context of pain research and the literature surrounding this pain condition:

  • Peripheral neuropathy (or simply neuropathy) is a disturbance of function or pathological change in a nerve outside the brain and spinal cord (the peripheral nervous system).[9]
  • Pudendal neuropathy is any damage or disease process affecting the pudendal nerve, regardless of whether said disease process involves nerve entrapment and manifests as pain or not. It is an example of mononeuropathy (neuropathy affecting one peripheral nerve).
  • Neuralgia is pain in the distribution of a nerve.[9] Usually, such conditions involve paroxsysmal pain (sudden, severe, electric shock like pain). Neuralgia can be considered as a subtype of neuropathic pain.
  • Pudendal neuralgia is neuropathic pain which is perceived along the course of and in the distribution of the pudendal nerve or its branches (anus, perineum, vulva, clitoris, glans penis, posterior aspect of scrotum).[10] Pudendal neuralgia is caused by some pathology affecting the pudendal nerve or its branches.[10] The pain in pudendal neuralgia may or may not be of similar character to other medical conditions which are classified as neuralgia. One potential cause of pudendal neuralgia is pudendal nerve entrapment.[11] However, symptoms of pudendal neuralgia are also possible without any detectable entrapment of the pudendal nerve.[7]
  • Neuritis is inflammation of a nerve.[9] It is a subtype of neuropathy.[9] However, not all neuropathies involve inflammation.
  • Neuritic pain is pain related to neuritis. Neuritic pain could be considered as a subtype of neuropathic pain.

Symptoms

There are no specific clinical signs or complementary test results for this condition.[12]

Pelvic pain

Pain is located in the distribution of the pudendal nerve.[5] In over 50% of cases, the pain is in the perineum, but may be located in the genital areas (vulva, vagina, clitoris in females; glans penis, scrotum in males).[13] Pain may also be perceived in the rectum in males and females (described as "sitting on a golf ball" or a "hot poker in the rectum").[13] Pain may also involve the supra-pubic region and the sacrum.[5] The pain is usually on both sides.[13] Another possible site of pain is the coccyx. The pain symptoms usually begin gradually, although sometimes the condition may appear suddenly after trauma.[13] The character of the pain may be burning, tingling, aching, stabbing, or like an electric shock.[13] Additionally, there may be referred as sciatic pain, or pain in the medial thigh which may indicate involvement of the obturator nerve.[13] Pain may also be referred to the calf, foot and toes.[13] Sometimes, pain is perceived in the region of the lower abdomen, posterior (back) and inner thigh, or lower back.[13] Hyperesthesia may be present.[4] The pain typically gets slowly worse over the course of the day.[13] The pain is positional and typically provoked or aggravated by sitting and relieved by standing, lying down or sitting on a toilet seat.[14] If the perineal pain is positional (i.e. changes with a person's position, for example sitting or standing), this suggests a tunnel syndrome.[clarification needed][15] According to one opinion, pain while sitting which is relieved by standing or sitting on a toilet seat is the most reliable diagnostic parameter.[16] The pain may be intense, chronic, and debilitating.[13]

Urinary

There may be bladder dysfunction such as urinary incontinence,[17] urinary frequency, dysuria, urinary urgency, or dyspareunia. There may be symptoms which are similar to interstitial cystitis.

Rectal

Bowel dysfunction may be present such as fecal incontinence[18] There may be numbness of the anal region.

Sexual

A systematic review found that PN may be implicated in various sexual dysfunctions such as persistent genital arousal disorder (PGAD), erectile dysfunction / impotence, premature ejaculation, and vestibulodynia.[19] There may be pain after ejaculation and pain after sex.[13] Additionally, another review that looked at cycling-related sexual dysfunction suggested that cycling may indirectly cause sexual dysfunction by disturbing the testosterone signaling aspect of the hypothalamic-pituitary-gonadal axis of the body.[20] There may be numbness of the genital area.

In cyclists

In male competitive cyclists, it is often called "cyclist syndrome".[6] This is a rare condition in which recurrent numbness of the penis and scrotum develops after prolonged cycling. There may be altered sensation of ejaculation, disturbance of micturition (urination), and reduced awareness of defecation.[21][22] Nerve entrapment syndromes, presenting as genital numbness, are amongst the most common bicycling associated urogenital problems.[23]

Anatomy

Anatomy of the pudendal nerve from the spinal nerve roots to the terminal branches in an adult male.
Main article: Pudendal nerve

The pudendal nerve carries both motor (controlling muscles) and sensory axons. It stems from the spinal nerves S2–S4 of the sacral plexus.[24][25] The nerve progresses between the piriformis and coccygeus muscles and exits the pelvis by passing through the greater sciatic foramen.[24] The pudendal nerve then re-enters the pelvic cavity by passing through the lesser sciatic foramen. After re-entering the pelvis, it breaks off into three branches known as the inferior rectal nerve, the perineal nerve, and the dorsal sensory nerve of the penis or clitoris.[24] These three nerves are also referred to as the terminal branches, and they are more susceptible to injuries due to their locations.[medical citation needed]

There are also four levels of pudendal nerve entrapment compressions:[26]

Although there has been no evidence for a direct functional connection between the pudendal nerve and sacrotuberous ligament, many clinical studies have pointed at the sacrotuberous ligament as a potential cause of PNE.[27] Around the ischial level of the spine, the pudendal nerve runs between the sacrotuberous ligament and the sacrospinous ligament (posteriorly and anteriorly, respectively), allowing potential compression of the pudendal nerve.[28]

Causes

Trauma from childbirth

Vaginal birth may lead to pudendal nerve damage. Childbirth causes stretching of the pelvic muscles[13] and the pudendal nerve. Such stretch-induced pudendal neuropathy may occur with a 12% stretch of the nerve.[29] The nerve is especially vulnerable to stretch damage during childbirth because of the course of the nerve,[25] as it runs in close proximity to pelvic muscles (piriformis and coccygeus) and ligaments, before exiting and then re-entering the pelvic cavity.[24] Stretching occurs during delivery, especially from the child's head.[13] The risk increases when delivering larger-than-average babies or with prolonged or difficult labour.[13]

Surgical trauma

Surgical procedures in the pelvic region may cause damage to the pudendal nerve.[25] Pudendal nerve injury has been reported in obstetric, perineal, and colorectal procedures.[25] Individuals with atypical pelvic anatomy are at higher risk of development of pudendal neuralgia after pevlic surgery.[25][30] Specific examples of procedures which have been reported to cause pudendal nerve injury include:

Surgical procedures which aim to correct prolapse of pelvic organs is reported to be the most common cause of pudendal neuralgia.[24] The risk is higher if mesh is used.[24] In some cases, subsequent removal of the mesh resulted in improvement in pain symptoms.

Cycling

PNE can develop in cyclists; likely due to both the compression and stretching of the pudendal nerve for prolonged time.[31] Heavy and prolonged cycling, especially if an inappropriately shaped or incorrectly positioned bicycle seat is used, may eventually thicken the sacrotuberous and/or sacrospinous ligaments and trap the nerve between them, resulting in PNE.

Other causes

Anatomic abnormalities can result in PNE due to the pudendal nerve being fused to different parts of the anatomy, or trapped between the sacrotuberous and sacrospinalis ligaments. Pelvic trauma can also cause pudendal neuralgia.

Pathophysiology

Main article: Pathophysiology of nerve entrapment

Prolonged pressure on the pudendal nerve and chronic traction injuries interrupt the normal microvasculature (blood supply via small blood vessels) of the pudendal nerve, triggering a cascade of physiological changes. Firstly, there is a breakdown of the blood-nerve barrier. Secondly, edema and connective tissue changes occur. This is followed by diffuse demyelination, and finally by Wallerian degeneration. In the acute form, a metabolic block by an impaired blood supply will interrupt normal function of the pudendal nerve. In the chronic form, neuropraxia and axonmetesis (Sunderland type 1 and 2) injuries will create positive symptoms (e.g. pain and paresthesias) and negative symptoms (loss of sensation).[32][33][34]

Diagnosis

Labat et al state that "there are no specific clinical signs or complementary test results of this disease".[12] Kaur et al confirm that there are no specific and consistent radiological findings in patients with PNE.[24]

Diagnostic tests that can be performed to suggest PNE are:

  • Pudendal nerve blocks to confirm the pudendal nerve is the source of pain through relief from the procedure.[24] These diagnostic blocks can also be used in place of spinal anesthesia during delivery.[25]
  • Quantitative sensory threshold testing to detect the inability to sense temperature changes.
  • High-frequency ultrasonography to identify the location of pudendal nerve compression.
  • Doppler ultra sound to detect vein compression, a result of nerve compression.[24]
  • Pudendal nerve terminal motor latency test, an invasive diagnostic test that involves a rectal or vaginal exam.[35]

Diagnoses are made through neurophysiological testing rather than imaging. However, MRI and CT imaging may be used to exclude other diagnoses.[24]

Similar to a Tinel's sign digital palpation of the ischial spine may produce pain. In contrast, people may report temporary relief with a diagnostic pudendal nerve block (see Injections), typically infiltrated near the ischial spine.[12] It is important to note that the duration of pain relief from pudendal nerve block is different per person.[36]

Imaging studies using MR neurography may be useful. In people with unilateral pudendal entrapment in the Alcock's canal, it is typical to see asymmetric swelling and hyperintensity affecting the pudendal neurovascular bundle.[37]

Nantes Criteria

Pudendal nerve entrapment is difficult to diagnose and there are no specific examinations that can clearly confirm the diagnosis. A multidisciplinary group in Nantes, France developed a set of diagnostic criteria (the "Nantes Criteria") to serve as a guide to physicians in diagnosing PNE.[29] It consists of inclusions, exclusions, and complementary characteristics of the syndrome.[24] Some sources discourage the use of this guide due to errors found in the criteria.[citation needed]

Inclusion criteria are:[24]

  1. The involved area corresponds to the area of supplied by the pudendal nerve (anus to the clitoris or penis).[29]
  2. Pain worsened by sitting, because of increased pressure on the nerve.[29]
  3. The patient is not awoken by pain during sleep.[29]
  4. No objective loss of sensation on clinical examination. Loss of superficial sensation in the perineal area is more indicative of a lesion at the root of the sacral nerves.[29]
  5. Pain relieved by an anesthetic block of the pudendal nerve.[29]

Exclusion criteria are:[24]

  • Imaging results that identify the cause of the pain
  • Pain is exclusively paroxysmal (it comes and goes in short bursts)
  • Pruritus (itching), which suggests a skin lesion
  • Pain not in the area innervated by the pudendal nerve

Complementary criteria are:[24]

  • Nerve pain associated with extreme sensitivity to touch (allodynia)
  • Described as burning/shooting/stabbing pain
  • Posterior pain following defecation
  • Predominantly unilateral pain (pain on one side only)
  • Foreign body sensation in the rectum or vagina
  • Tenderness around the ischial spine during rectal or vaginal examination
  • Abnormal neurophysiological tests

A systematic review by Indraccolo et al analyzed PN due to pudendal entrapment and PN without pudendal entrapment in women with chronic pelvic-perianal pain. The review classified the Nantes' criteria as the gold standard for diagnosing PN secondary to PNE.[38] Because of this, the authors of the systematic review additionally suggest that the criteria may be useful in assessing the efficacy and effectiveness of the pudendal nerve entrapment treatments that people may undergo.

Differential diagnosis

Differential diagnosis should consider the far commoner conditions chronic prostatitis/chronic pelvic pain syndrome and interstitial cystitis.[14] Other causes for similar symptoms of pudendal nerve entrapment include compression from a tumor, prostatitis in males, uterine diseases in females, complex regional pain syndrome (CRPS), superficial skin infections, and other neuropathies that share the same region as the pudendal nerve.[24]

Management

Treatments include behavioral modifications, physical therapy, analgesics and other medications, pudendal nerve block, and surgical nerve decompression.[7] A newer form of treatment is pulsed radiofrequency.[39] Most medical treatments are intended for symptomatic relief, such as pain. If symptoms are not managed through this standard of care, surgery is considered.[17]

Nerve protection

This is a form of self treatment to keep pressure off the pudendal nerve. It involves avoiding any activities that may increase pain in the pelvic area.[24] A seat cushion with the center area removed may be used to provide relief and prevent further pain.[35] A 2021 systematic review of preventative and therapeutic strategies found that cyclists who take precautions in maintaining proper posture may prevent the development of a more severe disorder.[40] It is also suggested that using a wider seat when cycling could prevent damage to the nerve, but more evidence is necessary to show long-term benefit.[31]

Ergonomics

Various ergonomic devices can be used to allow an individual to sit while helping to take pressure off of the nerve. A few recommendations to decrease nerve compression while cycling include having soft, wide seat in a horizontal position and setting the handlebar height lower than the seat.[40] There are also bicycle seats designed to prevent pudendal nerve compression, these seats usually have a narrow channel in the middle of them. Additionally, other recommendations include wearing padded bike shorts, standing on pedals periodically, shifting to higher gears, and taking frequent breaks.[40] For sitting on hard surfaces, a cushion or coccyx cushion can be used to take pressure off the nerves.

Physical therapy

Mobilization of the nerves and muscles in the pelvic region is a proposed way to treat symptoms associated with a nerve entrapment. An example of this is neural mobilization. The goal of neural mobilization is to restore the functionality of the nerve and muscles through a variety of exercises involving the lower extremities. Exercises to specifically target the pudendal nerve would be determined based on the anatomical layout of the nerve. It is important to note that evidence is limited to show support for this therapy.[41]

Another possible treatment for nerve entrapments in the pelvic region would be stretching and strengthening exercises. A treatment plan would be determined by a physical therapist to specifically manipulate the pudendal nerve through a variety of stretches. Strengthening exercises may also be recommended to relieve the excessive pressure caused by the entrapment, but there is currently limited evidence to support this choice of therapy.[41]

Medications

There are numerous pharmaceutical treatments for neuropathic pain associated with pudendal neuralgia. Drugs used include anti-epileptics (like gabapentin[39]), antidepressants (like amitriptyline[14]), and palmitoylethanolamide.[42] Often times polypharmacy is used with consideration of medication history and side effects.[35]

Injections

Illustration of imaging from a CT-guided injection of the pudendal nerve at the pudendal canal.

One way to identify and alleviate pain associated with the pudendal nerve is a "CT-guided nerve block."[43] During this procedure, "a long-acting local anesthetic (bupivacaine hydrochloride) and a corticosteroid (e.g. methylprednisolone) are injected to provide immediate pudendal anesthesia."[14] A pudendal nerve block can be inserted from several different anatomical locations including: transvaginal, transperitoneal, and perirectal. A reduction in pain following this injection is typically felt quickly. The most common side effect of a pudendal nerve block is injection site irritation.[36] Relief from chronic pain may be achieved through this procedure due to the reduced inflammation from the steroid medication, and "steroid-induced fat necrosis" which "can reduce inflammation in the region around the nerve" to lessen strain on the pudendal nerve. This treatment may alleviate symptoms for up to 73% of people.[14] Treatment of pudendal nerve entrapment by nerve block is not often prescribed due to "discomfort associated with the local injections as well as the risk of injuring critical structures."[36]

Pulsed radiofrequency

This can be used instead of pudendal nerve perineural injections.[35] In recent years, Pulsed radiofrequency (PRF) is starting to become more common for managing chronic pain, and has shown to have long-term benefits and low problem occurrences.[44] Pulsed radiofrequency has also been successful in treating a refractory case of pudendal neuralgia, but additional research is needed to study the effectiveness of pulsed radiofrequency on treating pudendal nerve entrapment.[39] Pudendal Nerve Stimulation (PNS) was found to significantly decrease subjective pain levels in people with pudendal neuralgia. A majority of people who underwent PNS reported "significant" or "remarkable" pain relief at 2 weeks after treatment.[45]

Surgical

Decompression surgery is a "last resort", according to surgeons who perform the operation.[15] It is highly controversial.

According to supporters of the theory of PNE, surgery is indicated when severe symptoms are present after exhausting all other forms of treatment. The surgery is also another option to confirm the diagnosis of pudendal nerve entrapment.[35]

The surgery is performed by a small number of surgeons in a limited number of countries. The validity of decompression surgery as a treatment and the existence of entrapment as a cause of pelvic pain are highly controversial.[46][47] While a few doctors will prescribe decompression surgery, most will not.

There are several different approaches in order to perform a decompression surgery on the pudendal nerve. The different access areas include: superior transgluteal, superior retrosciatic, inferior retrosciatic, medial transgluteal, inferior transgluteal and transischial entry.[26] The transgluteal entry involves "neurolysis of the PN at the infrapiriform canal and transection of the sacrospinal ligament." Another point of entry which is described as a "perineal para-anal pathway", "follows the inferior rectal nerve to the Alcock's canal."[17]

If nerve damage is discovered, other surgery options may be considered like a "neurectomy" or "neuromodulation".[17]

Prognosis

Pudendal neuralgia is not well-known. As a result, there may be misdiagnosis and inappropriate treatments, or it may take a long time before a correct diagnosis is achieved. Affected individuals may undergo various tests and investigations, and over time may seek treatment with multiple different medical specialists such as gynecologists, colorectal surgeons, and urologists. Attempts at treatment may be ineffective at resolving pain. As a result, the long-term, poorly-controlled pain may dramatically reduce quality of life. In some cases, opioid addiction or depression develops. There have been confirmed suicides because of delays in diagnosis and treatment. However, if the condition is quickly identified and properly managed, long-term control of symptoms should be possible.[13]

Epidemiology

The exact prevalence is unknown, but pudendal nerve entrapment and pudendal neuralgia are thought to be uncommon[1][48][6] or rare.[7]

Pudendal neuropathy may occur in males and females, and at any age from toddlers to 90-year-olds.[5]

History

Pudendal neuralgia was first described in cyclists in 1987.[49]

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