Rafał Przybylski, *Paulina Malinowska
Pudendal neuralgia – a challenging diagnostic pathway
Neuralgia nerwu sromowego ? trudna droga diagnostyczna
Warsaw Proctology Centre, St. Elizabeth’s Hospital in Warsaw
Streszczenie
Autorzy przedstawiają przypadek kliniczny dotyczący pacjenta z neuralgią nerwu sromowego (zwanej również zespołem kanału Alcocka), którego droga diagnostyczna i lecznicza trwała ponad 6 lat. Podejrzenie neuralgii zostało postawione po ok. 6 miesiącach od początku wystąpienia objawów, lecz na skutek braku skuteczności podejmowanego przez pacjenta leczenia poszukiwał on innych przyczyn dolegliwości. Przebył leczenie urologiczne w związku z podejrzeniem zapalenia prostaty, leczenie proktologiczne w związku z bólem okolicy odbytu oraz bólem przy defekacji, podejmował terapię w wielu ośrodkach leczenia bólu, poddał się również psychoterapii w związku z podejrzeniem psychogennego charakteru dolegliwości. Żadne działania terapeutycznie nie przyniosły pacjentowi oczekiwanej poprawy, co znacząco obniżyło jego jakość życia. Autorzy w podsumowaniu podkreślają, ze leczenie pacjentów z neuralgią nerwu sromowego jest trudne, najczęściej wieloletnie, obejmuje zabiegi fizjoterapeutyczne i w sporadycznych sytuacjach leczenie operacyjne.
Dolegliwość jest rzadka i niewiele jest ośrodków zajmujących się diagnostyką i leczeniem pacjentów z neuralgią nerwu sromowego, dlatego też pacjenci z tą chorobą często przez wiele lat oczekują na właściwą diagnozę i leczenie.
Summary
We present a clinical case of a male patient with pudendal neuralgia (also known as Alcock’s canal syndrome), whose diagnostic and therapeutic path lasted more than 6 years. Although the suspicion of neuralgia was raised about 6 months after symptom onset, the failure of the treatment undertaken by the patient prompted him to seek other causes underlying the manifestations. He underwent urological treatment for suspected prostatitis, proctological treatment for perianal pain and pain on bowel movement, therapy at a number of pain management centers, and psychotherapy for suspected psychogenic nature of symptoms. None of these therapeutic approaches led to the expected improvement, which significantly reduced the patient’s quality of life. In conclusion, we emphasise that the treatment of patients with pudendal neuralgia is difficult, usually takes many years, as well as includes physiotherapy and, occasionally, surgical treatment. Pudendal neuralgia is rare and there are few centers for the diagnosis and treatment of patients with this disorder. Therefore, the affected patients often wait many years for proper diagnosis and treatment.
Słowa kluczowe: proktalgia, nerw sromowy, neuralgia nerwu sromowego
Introduction
Pudendal neuralgia (PN) is a rare condition, and its actual incidence is unknown. The International Pudendal Neuropathy Association estimates an incidence of 1 in 100,000 people, which is however believed to be underestimated (1).
Pudendal nerve entrapment (PNE) may affect approximately 1% of the general population and accounts for approximately 4% of all patients consulted for perineal pain, with women affected more than twice as often as men (2).
Case report
A 37-year-old man with no history of chronic diseases had been experiencing urethral pain during voiding and ejaculation, as well as perineal pain since adolescence (13-15 years of age).
In his 30s, he developed perineal pain, more severe on the left side, increasing in a sitting position, especially on a hard seat. The pain was greater during the day. The patient also complained of pain on voiding and urinary frequency (only during the day), difficulty maintaining an erection and pain during ejaculation, occasional anal pain and pain during bowel movement, but only on the left side. No symptoms were experienced during sleep and in the morning, but they increased during the day and after sexual intercourse. He had negative history of perineal trauma and anorectal surgery.
The patient experienced three types of perineal and anal pain:
? stress-induced,
? sitting-related (especially on a hard surface),
? hyperesthesia.
The man was consulted by a urologist for the above symptoms and was diagnosed with chronic prostatitis. Dissatisfied with the consultation and concerned about the diagnosis, he began to search the Internet for information on disorders that could produce similar symptoms to those he was experiencing. This is how he found out about the possibility of pudendal EMG, which he had performed “on his own” in a private office. The EMG showed damage to the left pudendal nerve. Since the availability of this test is very limited, the test itself has a high risk of error (the patient has to meet a number of requirements in order for the results to be reliable), its clinical usefulness is questioned and it is rarely employed.
EMG-based suspicion of PN was made relatively quickly, about six months after the onset of chronic pain. The patient reported to the Outpatient Pain Management Clinic at one of hospitals in Warsaw.
His medical history further included:
? At 21 years, left knee arthroscopy – suturing of the meniscus after a torsional injury while playing football.
? At that time, he received an orthopaedic diagnosis of shortening of the right lower limb of about 7 mm based on an X-ray in the standing position. The patient used orthotic insoles, which he discontinued due to hip pain.
? At 24 years, he was diagnosed with a history of Scheuermann’s disease confirmed by thoracic MRI of the spine.
? At 26 years, he was admitted to the department of neurology for thoracic and shoulder muscle pain; MS was ruled out, among other things, and Tietze’s syndrome was diagnosed (so far asymptomatic).
? At 27 years, he was diagnosed with left knee arthroscopy due to traumatic damage to the meniscus (meniscus suturing) and posterior cruciate ligament (ligament reconstruction).
? At 29 years, Lyme disease was ruled out at the Hospital for Infectious Diseases in Warsaw.
? At 29 years, latent tetany was diagnosed; managed with diet and supplementation of magnesium, vitamin D3 and B complex.
? At 35 years, surgery due to a trimalleolar fracture of the right ankle; the fusion material was removed.
In the first year after symptom onset, the patient underwent:
1. non-US-guided left pudendal nerve block performed by an anaesthesiologist at a pain management clinic, with no postoperative improvement, but instead even a periodic worsening of symptoms,
2. US-guided RF ablation of the left pudendal nerve (not reimbursed by the National Health Fund), with no improvement,
3. rehabilitation of the pelvic floor and the levator ani (physical therapy, manual therapy, electrostimulation), with no improvement,
4. group and individual psychotherapy.
In the second year after symptom onset, the patient:
1. underwent Botulinum toxin (BTX) injection in the area of the anal sphincter muscles, with no improvement,
2. underwent US-guided cryoneurolysis of the left pudendal nerve (not reimbursed by the National Health Fund), with no improvement,
3. started pregabalin at 2 x 150 mg, which caused a slight reduction in the symptoms,
4. received psychiatric pharmacotherapy, which, however, caused no improvement and had no effect on the perineal or anal symptoms,
5. started to avoid sedentary position for longer periods of time and used a donut seat cushion.
Due to perianal pain and pain on bowel movement, the patient was consulted several times by proctologists.
He had been experiencing the above symptoms for about 6 years. After 6 years, he reported for repeated pudendal EMG in the same laboratory as before (not reimbursed by the National Health Fund), which showed greater damage to the pudendal nerve compared to baseline. After another proctological consultation and another consultation by a rehabilitation specialist in the field of pelvic floor disorders, the patient was qualified for surgical treatment.
He underwent the following three procedures at several-month intervals, at the European Centre for Chronic Pelvic Pain (EUCCPP) in 2023:
1. Left pudendal block with bupivacaine and epinephrine + BTX injection of the pelvic floor. A short-term symptom reduction by approx. 10% was observed postoperatively.
2. Bilateral pudendal block with bupivacaine and epinephrine + BTX injection to the pelvic floor and the left piriformis muscle. There was a permanent symptom reduction by further 20%, i.e. a total of approx. 30% after 2 procedures, and the stress-induced pain resolved completely.
3. Bilateral pudendal block with bupivacaine and epinephrine + BTX injection to the pelvic floor following neurostimulator implantation. Although this procedure did not reduce the pain, it had an impact on maintaining the effect of relieving excessive pelvic floor tension, as reported by the patient.
Due to persistent pain despite BTX injections, the patient reported for neurological and neurosurgical consultation in 2023 at the Dr Jan Biziel University Hospital No. 2 in Bydgoszcz.
Powyżej zamieściliśmy fragment artykułu, do którego możesz uzyskać pełny dostęp.
Mam kod dostępu
- Aby uzyskać płatny dostęp do pełnej treści powyższego artykułu albo wszystkich artykułów (w zależności od wybranej opcji), należy wprowadzić kod.
- Wprowadzając kod, akceptują Państwo treść Regulaminu oraz potwierdzają zapoznanie się z nim.
- Aby kupić kod proszę skorzystać z jednej z poniższych opcji.
Opcja #1
29 zł
Wybieram
- dostęp do tego artykułu
- dostęp na 7 dni
uzyskany kod musi być wprowadzony na stronie artykułu, do którego został wykupiony
Opcja #2
69 zł
Wybieram
- dostęp do tego i pozostałych ponad 7000 artykułów
- dostęp na 30 dni
- najpopularniejsza opcja
Opcja #3
129 zł
Wybieram
- dostęp do tego i pozostałych ponad 7000 artykułów
- dostęp na 90 dni
- oszczędzasz 78 zł
Piśmiennictwo
1. Hibner M, Desai N, Robertson LJ, Nour M: Pudendal neuralgia. J Minim Invasive Gynecol 2010; 17(2): 148-153.
2. Kaur J, Leslie SW, Singh P: Pudendal Nerve Entrapment Syndrome. 2023 Aug 21. [In:] StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2024 Jan?. PMID: 31334992.
3. Bochenek A, Reicher M: Anatomia człowieka. Tom II, V. Wyd. V. Wydawnictwo Lekarskie PZWL, Warszawa 2010.
4. Labat JJ, Riant T, Robert R et al.: Diagnostic criteria for pudendal neuralgia by pudendal nerve entrapment (Nantes criteria). Neurourol Urodyn 2008; 27(4): 306-310.
5. Marcus-Braun N, Bourret A, von Theobald P: Persistent pelvic pain following transvaginal mesh surgery: a cause for mesh removal. Eur J Obstet Gynecol Reprod Biol 2012; 162(2): 224-228.
6. Ramsden CE, McDaniel MC, Harmon RL et al.: Pudendal nerve entrapment as source of intractable perineal pain. Am J Phys Med Rehabil 2003; 82(6): 479-484.
7. Lien KC, Morgan DM, Delancey JO, Ashton-Miller JA: Pudendal nerve stretch during vaginal birth: a 3D computer simulation. Am J Obstet Gynecol 2005; 192(5): 1669-1676.
8. Howard EJ: Postherpetic pudendal neuralgia. JAMA 1985; 253(15): 2196.
9. Elahi F, Callahan D, Greenlee J, Dann TL: Pudendal entrapment neuropathy: a rare complication of pelvic radiation therapy. Pain Physician 2013; 16(6): E793-797.
10. Pinna Pintor M, Zara GP, Falletto E et al.: Pudendal neuropathy in diabetic patients with faecal incontinence. Int J Colorectal Dis 1994; 9(2): 105-109.
11. Czekaj A: Szkolenie u dr. Renauda Bollensa w Tournai, w Belgii. Operacja laparoskopowego uwolnienia uwięźniętego nerwu sromowego – czyżby wielkimi krokami zbliżała się rewolucja w urologii, jaką do tej pory znaliśmy? Przegl Urolog 2023; 2: 138.
