The patient is a 49-year-old female shop assistant who has been under treatment at our center for about 10 years for progressive, difficult-to-treat migraine and tension-type headache localized to the occipital, left frontal, and left periorbital regions, with no coexisting painful diseases in anamnesis. During these years she has been treated prophylactically with anticonvulsants, tricyclic antidepressants, calcium channel blockers, and onabotulinumtoxin A.

She also has a history of uncontrolled use of drugs for acute migraine ranging throughout all of the pharmacological classes, such as acetaminophen, nonsteroidal anti-inflammatories, triptans, ergot derivatives, and drugs containing barbiturate combinations. Moreover, via “doctor shopping” she obtained opioids on prescription, which she tried without therapeutic success. She has since stopped using this wrong and harmful therapeutic approach.

For this reason she has fallen into a state of medication-overuse headache (MOH), which has been treated with seven repeated detoxification procedures, unresponsive to corticosteroids.

Visits to the emergency department became more frequent, with an increasing number of absences from work, and with a psychopathological profile clearly more oriented toward suicidal intent.

The absence of secondary pathology underlying the clinical diagnosis of chronic migraine followed by refractory chronic migraine was excluded twice, using magnetic resonance imaging, in November 2009 and then in December 2011.

Considering the refractoriness to pharmacological therapy, it was decided in February 2012 to refer the patient for peripheral neurostimulation treatment.

Both ONS and supraorbital nerve stimulation (SONS) were the object of discussion regarding the therapeutic decision. As the current evidence base at the time reported the ONS to be the best interventional treatment for refractory chronic migraine with or without MOH, we decided to proceed with ONS, reserving the possibility to use SONS later.

In the 4 weeks before implant, the average self-reporting pain intensity was 5/6 on the Numeric Rating 11–point Scale (NRS), with 56 crises lasting 5–6 h with NRS 9/10. The patient took 32 tablets of ibuprofen 400 mg, 12 tablets of rizatriptan 10 mg, and 10 tablets of acetaminophen 500 mg/codeine 300 mg.

In April 2012, the patient was submitted to a workup in preparation for ONS.

She was first referred for psychological assessment to evaluate the psychosocial risk factors, after which colonization by methicillin-resistant Staphylococcus aureus was excluded by nasal swabbing.

The patient was placed in the prone position. After infiltration with local anesthetic, a midline access was chosen to avoid the occipital arteries and greater occipital nerve injury (5–28 mm lateral to midline). A vertical incision was made, extending approximately 4 cm caudal from a point 1 cm below the occipital protuberance. A subcutaneous pocket was fashioned by a lateral blunt dissection, exposing the fascia for anchoring.

A curved Tuohy needle, with a plastic stylet for easy removal, was advanced laterally 6–7 cm off midline, in a subcutaneous plane under ultrasound guidance to place the lead just above the fascia to a depth of approximately 0.8–1.2 cm.

One quadripolar lead (Pisces Quad Model 3487A-45; Medtronic, Inc, Minneapolis, MN, USA) was placed on each side and then anchored to the fascia of the lateral pocket by a plastic anchor and 2-0 nonabsorbable suture. To prevent lead migration, a strain loop was positioned in the occipital pocket.

Extensions were then tunneled and externalized to be connected to an external pulse generator.

The patient reported a significant reduction in the frequency and severity of her headaches during the 2-week trial period, and proceeded to permanent implantation under local anesthesia. Two extensions were tunneled down and connected to an implantable pulse generator (IPG) (Restore Ultra Model 3; Medtronic, Inc).

At 6-month follow-up, the patient’s headache symptoms and prestimulator medications were significantly reduced.

In December 2012 the patient required an urgent visit because of worsening symptoms. In the previous month the symptoms had returned to the levels of the preimplantation period.

We tested different stimulation programs to change the polarity of contacts, rate, and pulse width, but with no therapeutic effect, so we decided to implant an additional electrode.

In January 2013 the patient underwent a new surgical procedure to activate the trigeminovascular complex, which is able to activate the parasympathetic autonomic response that involves the SPG. A left supraorbital eight-pole lead (Model 3778; Medtronic, Inc) was placed under local anesthesia and sedation.

The lead was positioned subcutaneously under ultrasound guidance, tangentially to the superciliary arch. It was tunneled under the skin above the ear to the occipital region and then to the IPG.

The occipital four-pole leads were both programmed (0−, 1−, 2−, 3+), with an amplitude of 1.6 V (right lead) and 1.4 V (left lead), pulse width of 110 ms, and frequency of 40 Hz. The eight-pole supraorbital lead was programmed (0+, 1+, 2−, 5+, 7−) with an amplitude of 1,2 V, pulse width of 90 ms, and frequency of 60 Hz.

At 6-month follow-up after supraorbital implant, the patient’s headache symptoms and the intake of medication were significantly reduced. The average pain score was NRS 2/4, with 11 crises lasting 5–6 h with NRS 6/7. She took 2 tablets of ibuprofen 400 mg and 2 tablets of rizatriptan 10 mg.

At 12-month follow-up the patient’s headache symptoms and medication regimen were stable to an extent comparable with the previous control.

The positive outcome of this case confirms the results from Slavin, Reed, Linder, and Datta, whereby the ONS combined with SONS produced a synergistic effect.

In 2009 Reed published the first report on the use of combined ONS-SONS for chronic migraine. Promising results were reported in the series of seven patients, with six describing near complete resolution of the pain and associated neurological findings.

Linder first described the use of combined ONS-SONS in a group of adolescents, and reported very good results over the long term.

According to the Melzack and Wall theory of “gate control,” the somatosensory neurostimulation of afferent fibers A and B stops the metameric nociceptive transmission. Therefore, the generally accepted clinical approach for the treatment of pain using neurostimulation is to produce a paresthesia in the same region where pain is perceived.

Despite this, previous studies have shown that ONS is able to modulate the pain even in regions metamerically discordant from C2/C3. For example, Schwedt, Magis, Dodick, Burns, Amin, and Asensio-Samper used ONS to treat frontal or supraorbital pain.

This particular analgesic effect of peripheral stimulation in the head is due to the particular and unique anatomy and physiology of the TCC, where all of the cephalic somatosensory afferents converge.

The combined neurostimulation, in particular from the occipital and trigeminal territory, synergistically activate the TCC.