An unusual presentation of cryptorchidism: A case report of Spigelian-cryptorchidism syndrome

• Published in: International journal of surgery case reports 2023-06, Vol.107, p.108350, Article 108350
• Main Authors: Adil, Maham Leeza, Alvi, Mahrukh, Shabbir, Arzoo, Osman, Muhammad, Bashir, Muhammad Nauman
• Format: Article
• Language: English
• Subjects: Abdominal pain; Case Report; Spigelian hernia; Spigelian-Cryptorchidism syndrome; Testicular descent; Trans-peritoneal primary repair
• ISSN: 2210-2612; 2210-2612
• DOI: 10.1016/j.ijscr.2023.108350

Spigelian hernia is an uncommon hernia presenting as a protrusion of abdominal contents through the spigelian fascia, lateral to the rectus abdominis. In some rare cases, Spigelian hernia can occur alongside cryptorchidism, which forms a recognized syndrome found in male infants with Spigelian hernia. This is a relatively unreported syndrome with very limited literature available regarding it, none of which is reported in Pakistan in adults. We report a case of a 65-year-old male with right sided obstructed spigelian hernia along with the rare finding of testis in the hernial sac. The patient was successfully managed by transperitoneal primary repair (herniotomy) with orchiectomy. The patient recovered uneventfully and was discharged 5 days after the surgery. The exact pathophysiology of this syndrome remains unclear. Three theories have been proposed to explain this syndrome, including the primary defect being Spigelian hernia leading to undescended testes (Al-Salem), testicular maldescent preceding the formation of the hernia (Raveenthiran), or the absence of the inguinal canal leading to the development of a rescue canal due to the undescended testes (Rushfeldt et al.). In this case, the absence of gubernaculum was confirmed suggesting the findings to be consistent with Rushfeldt's theory. The surgical team proceeded with hernial repair and orchiectomy. In conclusion, Spigelian-Cryptorchidism syndrome is a rare syndrome in adult male, with an unclear pathophysiology. Management of this condition involves repair of the hernia along with either orchiopexy or orchiectomy, depending upon the risk factors involved. •We reporta patient who presented with a right sided obstructed spigelian hernia along with undescended testes, forming the rare ‘Spigelian-Cryptorchidism Syndrome’•Patient was successfully managed by transperitoneal primary repair along with orchiectomy.•This report discusses the management and discusses this rare syndrome in detail.•Very limited literature is available on this syndrome, with none previously reported in Pakistan. Hence, the authors felt a need to compile this case report.