Introduction and importance: Crossed testicular ectopia is an extremely rare congenital anomaly in which both testes migrate to the same hemiscrotum through one inguinal canal. It is frequently associated with other developmental defects but rarely coexists with scrotal hypospadias. Given its infrequent presentation, standardized management protocols are limited. Case presentation: We report a case of 1-year-old male infant with abnormal genital configuration and left-sided crossed testicular ectopia. Both testes were located within the left inguinal canal without Müllerian remnants, accompanied by an inguinal hernia, a common vas deferens, and scrotal hypospadias with significant chordee. The patient received preoperative hormonal therapy (human chorionic gonadotropin and testosterone) to promote testicular descent and penile growth, followed by trans-septal orchiopexy with single-stage tubularized preputial island flap urethroplasty. Clinical discussion: Crossed testicular ectopia associated with hypospadias represents a complex form of urogenital malformation that demands individualized surgical planning. Hormonal stimulation may facilitate operative repair and optimize genital development. Thorough evaluation – including hormonal and genetic testing – is recommended to rule out disorders of sex development and guide management. Conclusion: Crossed testicular ectopia with scrotal hypospadias is an exceptionally rare entity requiring multidisciplinary assessment and tailored surgical correction. Early recognition and individualized operative strategy are key to achieving favorable functional and cosmetic outcomes.
Turyalai Hakimi (Mon,) studied this question.