Congenital Bilateral Spigelian-Cryptorchidism Syndrome? A Case Report
Background: There is ongoing debate on nomenclature of congenital Spigelian hernia (SH) associated with undescended testis (UDT) in infants due to its frequent reporting in last two decades despite rarity of the condition. It is commonly reported as unilateral entity. To the best of our knowledge, only 4 bilateral cases have been reported.
Case report: We report a case of bilateral congenital Spigelian cryptorchidism syndrome in a small infant who was dealt surgically with good outcome.
Conclusion: The main differentiating point from inguinal hernia is direction of herniation in SH is laterally and upwards while it is downwards towards scrotum in inguinal hernia.
Skandalakis PN, Zoras O, Skandalakis JE, Mirilas P. Spigelianhernia: surgical anatomy, embryology, and technique of repair. Am Surg. 2006;72:42–8.
Jones BC, Hudson JM. The syndrome of spigelian hernia and cryptorchidism: a review of pediatric literature. J Pediatr Surg. 2015;50:325–30.
Bilici S, Güneş M, Göksu M, Melek M, Pirinçci N. Undescended testis accompanying congenital Spigelian hernia: is it a reason, a result, or a new syndrome? Eur J Pediatr Surg. 2012;22:157–61.
Al-Salem AH. Congenital spigelian hernia and cryptorchidism: cause or coincidence? Pediatr Surg Int. 2000;16:433–6.
Raveenthiran V. Congenital spigelian hernia and cryptorchidism: a distinct clinical syndrome. J Pediatr Surg. 2007;42:1794–95
Rushfeldt C, Oltmanns G, Vonen B. Spigelian-cryptorchidism syndrome: a case report and discussion of the basic elements in a possibly new congenital syndrome. Pediatr Surg Int. 2010;26:939–42.
Biasutto SN, Repetto E, Aliendo MM, Borghino VN. Inguinal canal development: the muscular wall and the role of the gubernaculum. Clin Anat. 2009;22:614–8.
O’Rahilly R, Muller F. Human Embryology and Teratology. 3rd ed. NewYork: Wiley-Liss;2001:127–8.
Levy G, Nagar H, Blachar A, Blachar A, Ben-Sira L, Kessler A. Pre-operative sonographic diagnosis of incarcerated neonatal Spigelian hernia containing the testis. Pediatr Radiol. 2003;33:407–9.
Durham MM, Ricketts RR. Congenital Spigelian hernias and cryptorchidism. J Pediatr Surg. 2006;41:1814–7.
Fascetti-Leon F, Gobbi D, Gamba P, G Cecchetto. Neonatal bilateral Spigelian hernia associated with undescended testes and scalp aplasia cutis. Eur J Pediatr Surg. 2010;20:123–5.
Copyright (c) 2019 Syed Waqas Ali, Gulfam Bibi
This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.
- Authors retain copyright and grant the journal right of first publication with the work simultaneously licensed under a Creative Commons Attribution, Non commercial, Share alike License 4.0 that allows others to share the work with an acknowledgement of the work's authorship and journal.
- Authors are permitted and encouraged to post their work online (e.g., in institutional repositories or on their website) after publication as it can lead to productive exchanges, as well as earlier and greater citation of published work (See The Effect of Open Access).
- Authors also confirmed that they have taken permission/consent for publication of this manuscript and of copyrighted material (if it is used in the manuscript).