Migraine-tic syndrome: Two further case reports

Abstract

Background

Migraine-tic syndrome was first reported in 2004 in a 44-year-old woman who had concomitant symptoms of both typical trigeminal neuralgia and migraine. We report here two further cases of migraine-tic syndrome and speculate on the relevance of this condition to the pathophysiology of headache.

Case reports

A 43-year-old woman presented with typical trigeminal neuralgia symptoms that preceded the onset of migraine headache; both headache types responded to treatment with sumatriptan. A 35-year-old woman presented with trigeminal neuralgia that consistently followed the onset of migraine headache. The former aspect responded to baclofen, but the migraine headache required treatment with amitriptyline.

Discussion

These two patients provide further support for the presence of an overlap syndrome of migraine-tic. We suggest that there is a common pathway for trigeminal neuralgia and migraine.

Introduction

Levin et al. (1) first described migraine-tic syndrome, a previously unreported type of headache, in 2004 in a 44-year-old woman who had concomitant symptoms of both typical trigeminal neuralgia and migraine. This case report illustrated that two otherwise distinct head pain syndromes could occur concurrently, in a similar manner to migraine and the trigeminal autonomic cephalalgias, in which cluster-tic (2–4) and paroxysmal hemicrania-tic (5,6) had already been described. We report two further cases of migraine-tic and speculate on the relevance of this disorder in the pathophysiology of headache.

Case reports

Case 1

A 43-year-old woman presented with an 11-month history of intermittent left-sided facial pain. She had been referred by her general practitioner to a local ear, nose and throat clinic, but no clinical abnormality was evident and a computed tomography scan of her sinuses was normal. A diagnosis of ‘possible trigeminal neuralgia’ or ‘unusual migraine pain’ was suggested.

The patient described stereotyped paroxysmal, side-locked, left-sided head and face pain which, until prophylactic treatment was instituted, occurred monthly, without fail, within a day or two of the onset of menstruation. In each attack, the pain would start in her left cheek and steadily worsen over 1–2 hours. At first, she experienced very sharp paroxysms of pain, lasting for a few seconds to several minutes. The paroxysms were especially likely to occur if she talked, touched her face or ate, which is typical of the trigger effects seen in patients with trigeminal neuralgia. If she needed to drink, she would have to do so through a straw using only the right side of her mouth. She found it difficult to breathe through her left nostril because the pain would immediately become more severe. However, there were no trigeminal autonomic symptoms.

Within an hour of the onset of the attack, the patient experienced a progressively worsening background left facial pain spreading from her cheek to her upper and lower jaws, the left orbital area, involving the gums and left nostril, and then into the left temporal area. The pain was constant, but pounding, and worsened with head movements and lying down.

The patient tended to sit still and upright during the attacks. There was associated nausea, photophobia and phonophobia, but not osmophobia. The symptoms were severe for three days, but then started to ease and settled completely within one week. Headache diary cards confirmed the temporal profile. Both the preceding paroxysmal pain and the following constant, throbbing pain were present in each attack. The patient had no previous history of primary headache or migraine aura, but her mother had had migraine without aura. There was a past history of mild anxiety and depression for which she was taking 25 mg of dosulepin at night.

General and neurological examinations and a magnetic resonance imaging brain scan were normal on two occasions. A magnetic resonance angiogram showed no impingement of the vessels on the cranial nerves in the posterior fossa.

The patient’s general practitioner had initially prescribed carbamazepine five months after the onset of these monthly attacks and had increased the dose gradually to 500 mg twice daily. Although this did seem to reduce the frequency of attacks to four attacks in six months, the drug made her a little drowsy. When she was subsequently admitted to her local hospital with a severe attack, gabapentin was added and quickly increased to 300 mg three times per day with a plan to withdraw carbamazepine if it was effective. It was at this stage, about 11 months after the start of the attacks, that we first saw her in our headache clinic and we have now followed her progress for 15 months.

The patient had tried various over-the-counter analgesic preparations to treat the acute attacks before seeing her general practitioner, but they had been ineffective. Her general practitioner had prescribed several other analgesic drugs without any benefit. We first prescribed 10 mg of rizatriptan (Maxalt melts) for a single attack (one dose daily for the five days of the attack) and then zolmitriptan (Zomig) nasal sprays in two consecutive attacks (nine doses in total used once or twice per day). Neither drug was helpful. However, all the acute symptoms responded rapidly and completely, albeit temporarily, to 6 mg of sumatriptan injected subcutaneously on every occasion (five attacks treated, 18 doses given). About three injections per attack minimized the symptoms before the attack finally resolved. As a result of the success of this acute treatment, the patient later decided to reduce the dose of carbamazepine to 300 mg twice a day, but continued with gabapentin, which we had increased by an extra 300 mg at night (total daily dose 1200 mg, the most she could tolerate). Overall, the patient thought that the carbamazepine had been a little more effective than gabapentin in preventing the attacks, but that both had been useful (eight attacks in 15 months on this combination of drugs).

Case 2

A 35-year-old woman began to suffer migrainous headaches in July 2013. The attacks started in association with toothache due to a dental cavity, but continued after this had been treated. She had a family history of migraine and had experienced frequent non-specific headaches when she was younger, often related to menstruation. She also had a history of endometriosis and had been found to have mutations in the genes for factor V Leiden and prothrombin after she underwent screening in view of a positive family history.

The headaches were left-sided, throbbing and associated with nausea, blurred vision and photophobia. On some occasions, but always after her typical migraine attacks had started, she developed symptoms consistent with ipsilateral trigeminal neuralgia: sharp shooting pains triggered by eating, touch and cold. Magnetic resonance brain imaging with detailed sequences through the brainstem did not reveal any abnormality of the nerve roots or vascular cross compression.

The patient had been treated for trigeminal neuralgia only in primary care. After unsuccessful trials with carbamazepine and gabapentin, the trigeminal neuralgia pain responded to baclofen at a dose of 10 mg three times per day. In the neurology clinic, the migraine headache and the fact that it preceded the trigeminal neuralgia were identified. The migraine headaches had persisted despite successful treatment of the trigeminal neuralgia. They responded to the addition of amitriptyline and an increase in the dose to 30 mg at night. The migraine headaches often responded to acute treatment with naproxen. The trigeminal neuralgia never occurred without the preceding migraine.

Discussion

The characteristics of the paroxysmal facial pains in both patients were entirely typical of trigeminal neuralgia: brief, repetitive, stereotyped and similar to an electric shock. Both patients also described typical triggering and these features, together with the location of the pain, fulfilled the ICHD 3β criteria for trigeminal neuralgia (7). However, the subsequent additional headache in patient 1 and the preceding additional headache in patient 2, with their associated features, fulfilled the ICHD 3β criteria for migraine without aura and, in the first patient (apart from the longer duration), menstrual migraine (7). The consistent concurrent expression of these two semiologies makes a diagnosis of migraine-tic inescapable.

This association prompts speculation about the mechanisms involved. Although the nature of the initial pain in patient 1 might suggest primary trigeminal nerve activation, the resolution of both the trigeminal neuralgia and the migrainous components of the attacks with subcutaneous injections of sumatriptan strongly suggests that the migraine mechanism is the prime mover. The mechanism for the failure of rizatriptan and zolmitriptan probably lies in their different routes of application – oral and nasal compared with the subcutaneous injection of sumatriptan. The perceived better response to carbamazepine compared with gabapentin in patient 1 is probably due to the more prominent role of the trigeminal neuralgia component in this patient. The consistent occurrence of typical trigeminal neuralgia exclusively within the migraine attacks in patient 2 and the continuance of the migraine attacks despite control of the trigeminal neuralgia following treatment with baclofen further support this conclusion. Furthermore, although there are reports of otherwise refractory trigeminal neuralgia responding to triptans (8), we are not aware of trigeminal neuralgia occurring only in association with menstruation, which is well documented as a migraine trigger. Gabapentin can prevent attacks of both trigeminal neuralgia (9) and migraine (10) and it reduced the frequency of migraine-tic in patient 1. It is therefore possible that these two syndromes share common pain pathways.

Trigeminal neuralgia is believed to result from vascular cross compression of the ipsilateral trigeminal nerve by an arteriosclerotic vessel and can be relieved by procedures that reverse this. However, the syndrome can also result from brainstem pathologies such as multiple sclerosis. The brainstem is known to become activated in migraine attacks, engaging the trigeminovascular system. It has been suggested that migraine could map or remap to a site of trigeminal nerve injury (11,12). Thus, in rare instances, the origin of the trigeminal nerve might become involved by the migraine mechanism, as can occur with the oculomotor nerves in ophthalmoplegic migraine (13) and the facial nerve in migrainous facial palsy and hemifacial spasm (14–16). These two patients suggest a reciprocal link, with the migraine mechanism activating the trigeminal nerve to facilitate neuralgic pain and vice versa.

The three cases of migraine-tic reported to date demonstrate that migraine can occur concomitantly with trigeminal neuralgia, as in cluster headache and paroxysmal hemicrania. Successful treatment in our patients required specific interventions for both the migraine and the trigeminal neuralgia elements. This is in keeping with experience in both cluster-tic and paroxysmal hemicrania-tic, where dual treatment is often required (17,18). It is also compatible with the view that, although clinically distinct headache and facial pain syndromes are recognized and may need individual treatment, overlap syndromes such as migraine-tic are consistent with the view that the mechanisms for headache and head pain share common pathways (19). Further support for such a common pathway is provided by a recent population-based study that found an increased incidence of trigeminal neuralgia in patients with migraine (20).

We therefore propose an ICHD 3β appendix definition of migraine-tic syndrome as one single headache episode containing two distinct headache categories fulfilling the ICHD 3β criteria for migraine and trigeminal neuralgia.

Article highlights

Migraine can occur concomitantly with trigeminal neuralgia.

Individual treatment for each headache type is often required.

This and other overlap syndromes support the view that the mechanisms for headache and head pain are likely to share common pathways.