Hydrocele is a common cause of scrotal enlargement inneonates; however, its assessment becomes more complexin critically ill preterm infants because scrotal swelling mayalso indicate inguinal hernia, testicular torsion, hematoma,infection, or generalized edema. This case report describesa preterm neonate with very low birth weight, severeneonatal sepsis, respiratory distress, hypoalbuminemia,and ultrasonography-confirmed right scrotal hydrocele. Amale infant was delivered by cesarean section at 31 weeksand one day of gestation because of severe maternalpreeclampsia. His birth weight was 1,435 g. The infantexperienced perinatal asphyxia, respiratory distressrequiring continuous positive airway pressure,bronchopneumonia, severe sepsis, anemia,thrombocytopenia, hypoalbuminemia, and systemicedema. Right scrotal edema was observed duringhospitalization, and ultrasonography subsequentlyconfirmed right hydrocele. The infant received intensivesupportive care, respiratory support, antimicrobial therapy,nutritional support, and albumin transfusion but diedfollowing cardiorespiratory arrest. This case highlights theneed for systematic assessment of scrotal swelling incritically ill preterm neonates and careful differentiationbetween hydrocele and urgent inguinoscrotal disorders.