Abstract Introduction/Background Peripherally inserted central catheters (PICC) are an essential and common procedure in neonatal intensive care units (NICUs). Peripheral nerve injuries secondary to PICC extravasation are a rare occurrence in neonates, who commonly recover with no sequalae. Rarely, extravasation results in compression and ischemia involving adjacent nerves, muscles or joints. Peripheral nerve injury may be induced by compression from tissue infiltration or irritation by the infiltrate. Timely diagnosis and treatment are essential to avoid complex or irreversible nerve damage, such as compartment syndrome, requiring extensive interventions. Isolated radial nerve injury in infants and neonates is limited to a few cases reported in the literature, which have been commonly related to in-utero compression or birth injury due to peripheral positioning. We report a late presentation of an isolated radial nerve injury secondary to PICC extravasation in a preterm infant, leading to neuropraxia and long-term impairment of hand function. Case Description An extreme preterm female infant (245/7 weeks’ gestation, 640 grams) was admitted to the NICU with a complex neonatal course. At 6 days after birth, a right antecubital PICC was inserted for vascular access and to provide parenteral nutrition. It was removed after 18 days following extravasation. Two months later, she developed a right wrist drop, flexed fingers, and edema of the right hand. Her right wrist flexion, elbow flexion, shoulder joint range of motion, and sensation remained intact. A Doppler study showed a small non-occlusive clot in the right brachial vein, attributed to the previous PICC. She was unable to voluntarily open her right hand or extend her metacarpophalangeal (MCP) or interphalangeal (IP) joints (see Figure A, below). Clinical diagnosis of radial nerve palsy was made, and she was referred to physiotherapy (PT) and occupational therapy (OT). Splinting, passive exercise and left-hand constraint resulted in some improvement of the right hand’s strength. At 12 months of age, she had left hand preference with limited active extension of right MCPs and IPs (Figure B). She received a Botox injection to the right-hand flexors to allow strengthening of the extensor muscles. Follow-up at 18 months showed full extension of the index and middle finger MCPs and IPs, but residual lag in ring and small finger IPs. Discussion To our knowledge, this is the first case report of isolated radial nerve injury in an infant following PICC extravasation and soft tissue infiltration. Interestingly, the clinical signs of radial nerve injury in this case presented almost two months after PICC extravasation. Additionally, the left-hand preference and limitation of the active extension of the fingers of the right hand remained at one year of corrected age. Given that this has exceeded the expected recovery time of radial nerve palsy, Botox injection was performed with good response and near age-appropriate development of fine motor skills at 18 months of corrected age. Paediatricians should be aware that the manifestation of potential nerve injury in neonates may have a delayed presentation. Neurology consultation to confirm the diagnosis and rule out other causes of muscle weakness, and referral to PT/OT, are crucial for managing infants and neonates with peripheral nerve injury because treatment is largely focused on splinting and passive motion exercise. Generally, radial nerve palsy carries a relatively good prognosis, with complete functional recovery within 1-6 months. The fact that this case developed long-term complications with impaired right-hand function, despite nerve recovery, raises concerns about the prognosis. Conclusion Peripherally inserted central venous catheters are an everyday procedure in the NICU. Radial nerve palsy secondary to venous catheter extravasation is rare in infants and neonates. Clinicians should be aware of the clinical signs of radial nerve palsy and initiate consultation and referral to physiotherapy and occupational therapy early. Families can be reassured of the good prognosis and functional recovery.