Key clinical messageSeveral animal experiment studies have shown that insufficient testicular descent to the scrotum can be caused by persistence of cranial suspensory ligament (CSL). We report a case of right cryptorchidism in a male toddler surgically treated with an orchidopexy possibly associated with CSL persistence based on intraoperative and pathological findings. This case would be a precious source to further investigate the etiopathogenesis of cryptorchidism.AbstractThe CSL anchors embryonic gonads to the dorsal abdominal wall during antenatal mammalian development. Although its persistence appears to cause cryptorchidism in animal models, it has never been proven in humans. A 1‐year‐old boy with right cryptorchidism underwent right orchidopexy. Intraoperatively, a band‐like structure running from the right testis into the retroperitoneum and up to the right side of the liver was noticed and resected. The pathological findings of the specimen showed fibrous connective tissues, smooth muscles, and blood vessels but no tissues suggestive of a testis, a spermatic cord, an epididymis, or liver. Immunohistochemical analysis for an androgen receptor antibody did not detect any signal in the specimen. The right cryptorchidism in this case was possibly caused by CSL persistence, which is the first such human case, to our knowledge.