Ömer AYIK, Erol KOZANOĞLU, Yavuz ÖNOL, Hayati DURMAZ

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, painfulerections, 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 signsand symptoms. We, herein, aimed to introduce the case of a 42-year-old male who underwent the distal release procedure due tothe diagnosis of “double crush pudendal syndrome” following a proximal release surgery previously performed elsewhere. Thepatient's history revealed a pelvic fracture with urethral injury 27 years ago. Throughout the years, the patient had beenevaluated by various medical disciplines and undergone several treatments. In 2017, an orthopedic surgeon performedproximal 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 afterthe proximal pudendal nerve release. Based on a detailed clinical and laboratory assessment, the diagnosis of double crushneuropathy was established, and distal release of the pudendal nerve using transperineal approach was performed. To determinethe 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 (9points) to the mild degree (22 points) postoperatively. The patient’s general and sexual satisfaction scores, and erection qualityscore improved compared to the preoperative baseline. According to VAS, the perineal pain, erectile pain,and pain duringintercourse decreased postoperatively. (from 7 to 2 out of 10, from 8 to 3 out of 10, from 7 to 2 out of 10, respectively). Whenperineal and sexual complaints are encountered following pelvic trauma, the pudendal nerve-related problems, especiallydouble crush syndrome, should be kept in mind in differential diagnosis. A multidisciplinary approach must be established inorder to avoid any delay in diagnosis and treatment. Surgical intervention may provide a significant improvement in clinical andfunctional status.

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1. Hibner M, Desai N, Robertson LJ, Nour M. Pudendal neuralgia. J Minim Invasive Gynecol. 2010;17(2):148-153. 10.1016/j.jmig.2009.11.003

2. Kane PM, Daniels AH, Akelman E. Double crush syndrome. J Am Acad Orthop Surg. 2015;23(9):558-562. 10.5435/JAAOS-D-14-00176

3. Labat JJ, Riant T, Robert R, Amarenco G, Lefaucheur JP, Rigaud J. Diagnostic criteria for pudendal neuralgia by pudendal nerve entrapment (Nantes criteria). Neurourol Urodyn. 2008;27(4):306-310. 10.1002/nau.20505

4. Pavelka T, Džupa V, Štulík J, Grill R, Báča V, Skála-Rosenbaum J. Výsledky operační léčby nestabilního poranění pánevního kruhu. ACHOT. 2007;74(1):19-28.

5. Metze M, Tiemann AH, Josten C. Male sexual dysfunction after pelvic fracture. J Trauma Acute Care Surg. 2007;63(2):394-401. 10.1097/01.ta.0000241145. 02748.df

6. Erdogru T, Avci E, Akand M. Laparoscopic pudendal nerve decompression and transposition combined with omental flap protection of the nerve (İstanbul technique): Technical description and feasibility analysis. Surg Endosc. 2014;28(3):925-932. 10.1007/s00464-013-3248-1

7. Beco J, Climov D, Bex M. Pudendal nerve decompression in perineology: A case series. BMC Surg. 2004;4(1):15. 10.1186/1471-2482-4-15

8. Shafik A, El OS, Shafik IA, Shafik AA. Role of sacral ligament clamp in the pudendal neuropathy (pudendal canal syndrome): Results of clamp release. Int Surg. 2007;92(1):54-59.

9. Loukas M, Louis RG, Hallner B, Gupta AA, White D. Anatomical and surgical considerations of the sacrotuberous ligament and its relevance in pudendal nerve entrapment syndrome. Surg Radiol Anat. 2006;28(2):163-169. 10.1007/ s00276-006-0082-3

10. Robert R, Prat-Pradal D, Labat J, et al. Anatomic basis of chronic perineal pain: Role of the pudendal nerve. Surg Radiol Anat. 1998;20(2):93-98. 10.1007/ BF01628908

11. Báča V, Báčová T, Grill R, et al.. Pudendal nerve in pelvic bone fractures. Injury. 2013;44(7):952-956. 10.1016/j.injury.2012.12.004

12. Tang DT, Barbour JR, Davidge KM, Yee A, Mackinnon SE. Nerve entrapment: Update. Plast Reconstr Surg. 2015;135(1):199e-215e. 10.1097/PRS. 0000000000000828

13. Copeland CE, Bosse MJ, McCarthy ML, et al. Effect of trauma and pelvic fracture on female genitourinary, sexual, and reproductive function. J Orthop Trauma. 1997;11(2):73-81. 10.1097/00005131-199702000-00001

14. Polyzois I, Tsitskaris K, Oussedik S. Pudendal nerve palsy in trauma and elective orthopaedic surgery. Injury. 2013;44(12):1721-1724. 10.1016/j.injury.2013.09.001

15. Sheng J, Liu S, Wang Y, Cui R, Zhang X. The link between depression and chronic pain: Neural mechanisms in the brain. Neural Plast. 2017;2017. 10.1155/2017/ 9724371