Combined Occipital Nerve and Sphenopalatine Ganglion Neuromodulation for Refractory Craniofacial Pain with Trigeminal Autonomic Features: A 12-Year Follow-Up (Case Report)

Vadim Tashlykov,1 Ruth Gur,2 Gabriel Ricardo Lichtenstein,2 Itay Goor-Aryeh,2 Evgeni Brotfain3 1Department of Anesthesiology, Pain Clinic, Samson University Medical Center, Public Hospital Assuta, Ashdod, Israel; 2Department of Anesthesiology, Pain Institute, Sheba Medical Center, Tel-HaShomer, Ramat Gan, Israel; 3Division of Anesthesiology and Critical Care, Soroka University Medical Center, Beer Sheva, IsraelCorrespondence: Evgeni Brotfain, Email [email protected]: Craniofacial complex regional pain syndrome (CRPS) is a rare, diagnostically challenging condition characterized by sympathetically maintained pain, allodynia, and autonomic features in the craniofacial region. Its overlap with trigeminal autonomic cephalalgias (TACs) creates significant diagnostic complexity. We report the case of a 41-year-old male with a 12-year history of severe right-sided hemicranial and facial pain with prominent autonomic features (facial edema, ptosis, lacrimation, allodynia) following plastic surgery in occipital area, in a patient with a prior history of CRPS. After failing extensive pharmacological management (including indomethacin, verapamil, topiramate, pregabalin, carbamazepine, lamotrigine, lithium and lidocaine infusions) and numerous interventional procedures (peripheral nerve blocks, Sphenopalatine ganglion (SPG) blocks, pulsed radiofrequency of SPG and Gasserian ganglion), the patient underwent sequential neuromodulation: occipital nerve stimulation (ONS) followed by sphenopalatine ganglion (SPG) stimulation seven years later. This combined approach resulted in sustained pain reduction from NRS 9– 10/10 to 1– 2/10, attack frequency reduction from 1– 2 daily to rare exacerbations, and complete discontinuation of all analgesic medications over a 12-year follow-up. This case demonstrates that combined ONS and SPG stimulation may provide effective long-term pain relief in refractory craniofacial pain irrespective of whether the underlying diagnosis is craniofacial CRPS or secondary TAC, and highlights the value of sequential neuromodulation when single-modality approaches are insufficient.Keywords: trigeminal autonomic cephalalgia, cluster headache, neuromodulation, occipital nerve stimulation, sphenopalatine ganglion stimulation, facial pain