Spigelian-Cryptorchidism Syndrome: A Literature Review and Case Report of a Rare Clinical Entity
Spigelian-Cryptorchidism Syndrome: A Literature Review and Case Report of a Rare Clinical Entity

Spigelian-Cryptorchidism Syndrome: A Literature Review and Case Report of a Rare Clinical Entity

Cureus. 2025 Jul 5;17(7):e87324. doi: 10.7759/cureus.87324. eCollection 2025 Jul.

ABSTRACT

Spigelian-cryptorchidism syndrome is a rare and diagnostically challenging condition in neonatology that is characterized by the association of an undescended testis with a Spigelian hernia, the latter usually without radiologic evidence. We report the case of a full-term male neonate delivered at 39 weeks and two days of gestation by kiwi-assisted vaginal delivery after a spell of fetal bradycardia. Antenatal imaging had shown persistent bilateral pelviectasis of the kidneys and an undescended right testis. Postnatally, the initial course was normal; however, clinical evaluation showed partially reducible sausage-shaped swelling in the right lower quadrant and an impalpable right testis. Ultrasonographic examination confirmed intra-abdominal location of the right testis and showed bilateral mild dilatation of the renal pelvis but no abdominal wall defect or hernia sac. Surgical assessment revealed a partially reducible mass, consistent with Spigelian hernia, and hence established the setting of Spigelian-cryptorchidism syndrome. The testis’s anatomical separation from the inguinal canal and the lack of a discernible processus vaginalis favored a syndromic diagnosis. The case brings to light the inherent limitations of imaging in the detection of neonatal Spigelian hernia, the value of physical examination by skilled clinicians, and the imperative of a multidisciplinary diagnostic and therapeutic approach. Conservative monitoring was chosen, with surgical repair scheduled at 12 months of age, consistent with best practices at present. This case supports that in neonatal presentations with radiological uncertainty combined with strong clinical evidence, the diagnosis can be pursued critically and not delayed based on imaging. It also stresses the requirement to consider unexplained abdominal distension and non-palpable testes on a differential which includes this uncommon but clinically important syndrome.

PMID:40765591 | PMC:PMC12322183 | DOI:10.7759/cureus.87324