Hemiscrotal agenesis is an extremely rare congenital anomaly characterized by unilateral absence of scrotal skin with preserved midline raphe. Optimal management, particularly conservative approaches in isolated cases without cryptorchidism, remains poorly defined. We report a 5-year-old boy with normal 46,XY karyotype presenting for circumcision revision, who had isolated right hemiscrotal agenesis, penile torsion, and excess preputial skin. The right testis was palpable subcutaneously with adequate perineal space. After penile detorsion and circumcision revision, sufficient skin elasticity allowed comfortable testicular positioning without reconstruction. Pubertal follow-up showed excellent cosmetic and functional outcomes, highlighting the value of individualized conservative management in selected cases. • One of the few reported cases of isolated hemiscrotal agenesis successfully managed conservatively • Adequate tissue elasticity allowed testicular positioning without scrotal reconstruction. • Excellent cosmetic and functional outcome at pubertal follow-up, avoiding surgical complications. • Emphasizes individualized approach for rare congenital scrotal anomalies with normal contralateral side. • Normal karyotype and no associated malformations in this isolated case.
Marzouki et al. (Sun,) studied this question.