BACKGROUND: Neuropathic head and facial pain are relatively common and difficult to treat pain syndromes encountered in pain clinics throughout the world. Atypical syndromes with overlapping nerve distributions can be particularly frustrating for patients and providers. In recent years, as providers search for alternatives to medications, advances in neuromodulation technology, including spinal cord stimulation and peripheral nerve stimulation (PNS), have shown promise in treating neuropathic pain confined to specific nerve distributions, such as the greater and lesser occipital nerves. However, there is limited literature describing the use of PNS for atypical facial pain. We report the first known case of PNS to the superficial cervical plexus in a 45-year-old woman with bilateral ear and postauricular pain. CASE REPORT: A 45-year-old woman presented with over 6 years of bilateral ear and periauricular pain in the auriculotemporal and lesser occipital nerve distribution. Conservative management, including neuropathic pain agents, ketamine infusions, nasal sprays, opioids, nonsteroidal anti-inflammatory medications, ice, heat, and transcutaneous neurostimulation, provided limited relief. She had excellent pain control with a series of nerve blocks to the superficial cervical plexus; however, relief was short term. In an attempt for a more long-term solution, she underwent implantation of the SPRINT PNS (MicroLead, SPR Therapeutics, Cleveland, OH), which resulted in 100% pain relief, improved function at work, and improved sleep tolerance throughout the 60-day treatment. Unfortunately, her symptoms returned shortly after treatment course completion. CONCLUSIONS: To our knowledge, this is the first case utilizing PNS of the superficial cervical plexus to treat atypical facial pain. The 60-day trial resulted in 100% pain relief of previously debilitating and functionally limiting pain symptoms during the treatment phase suggesting that the superficial cervical plexus may be a reasonable target for permanent PNS implants. KEY WORDS: Peripheral nerve stimulator, peripheral nerve stimulation, superficial cervical plexus, atypical facial pain, anesthetic blockade, pain relief, neuropathic pain, neuromodulation, SPRINT