Patients with isolated peripheral branch neuralgia of trigeminal nerve usually receive traditional treatment such as medical therapy and interventional procedures targeting the entire trigeminal nerve or related ganglions. However, if the intractable pain is limited to a certain branch, the patient may also benefit from a peripheral and nerve-targeted interventional approach. Here, we report the management of a patient with isolated infraorbital neuralgia by ultrasound-guided infraorbital nerve block with steroid and local anesthetic combination. 48 years-old male patient diagnosed with trigeminal neuralgia was resistant to medical therapy for 3 years. The pain site was isolated to the area of the right nasal wing, right lateral incisor, the upper right canine and the first premolar teeth. His pain was an electric shock-like, throbbing and stabbing with a pain score of 8-9 according to numeric rating scale (NRS) and 18 according to the Leeds Assessment of Neuropathic Symptoms and Signs Pain Scale (LANSS). Following a diagnostic ultrasound-guided infraorbital nerve block with 1% lidocaine, the block was repeated twice with 15 mg lidocaine and 1.5 mg dexamethasone in a total volume of 1.5 mL in a month. The patient's NRS and LANSS scores decreased to 2 and 8, for approximately 21 months until this report was written. We suggest that ultrasound-guided infraorbital nerve block with dexamethasone and lidocaine combination may present as an initial interventional treatment option in patients with isolated infraorbital neuralgia. (C) 2016 Elsevier Inc. All rights reserved.