Botulinum Toxin Type A in the treatment of sporadic hemiplegic migraine

Principle author and correspondence: Dr. Ray Goggins, Specialist Registrar in General Adult Psychiatry, Cirencester Memorial Centre, Sheep St, Cirencester, Gloucestershire, GL7 1QR
raygoggins@yahoo.co.uk
Telephone: +44 (01285) 640933 or +44 (07941) 865178
Fax: ++44 (0117) 9241750

Dr Daniel Rogers, Consultant Neuropsychiatrist, the Burden Centre, Bristol, BS16 1JB
Telephone (0117) 9186710

Recent reports suggest that Botulinum toxin type A may help patients suffering from chronic pain disorders. We report a unique case of a 47 year old woman with hemiplegic migraine responding to botulinum toxin type A injections.

There is increasing evidence that botulinum toxin type A may be a useful treatment for severe headaches and migraines. To our knowledge this is the first case report of its successful use in sporadic hemiplegic migraine.

A 44 year old woman was referred to a tertiary neuropsychiatric centre by her GP with disabling headaches. At the age of 41 she developed episodes of severe headache for the first time which gradually increased in severity. There was no family history of migraine. Episodes would start with nausea for 30 to 60 minutes followed by a pain in her forehead which could be right or left sided and occasionally bilateral and which spread gradually to the back of her head and down her neck. She also had mild photophobia, phonophobia and prostration. She often would notice "a cloudy film" over her vision but no classic migrainous visual disturbances. She described headaches as "horrendous" like "a tightening band" around her head. They occurred at any time of the day but often were present when she awakened. Simple analgesics were largely ineffective in reducing the pain.

She identified no particular precipitant. Indeed she altered her diet avoiding cheese, chocolate, alcohol etc. to no avail. There was no past psychiatric history and she had no history of head injury.

Using the International Headache Society's criteria (1) she was diagnosed with migraine with aura. She was treated with various triptans as acute treatments which were of partial benefit. Naratriptan for example, decreased the pain after 30 minutes although she continued to have pain for two days. She was also tried on various prophylactic agents at therapeutic doses for a trial period of 3 months each. These included propranolol, sodium valproate, gabapentin, pizotifen and clonidine. As each was only partial benefit they were withdrawn.

By the age of 47 these headaches were followed for the first time by pain in left jaw and shoulder and severe weakness and numbness of left upper limb for up to three days.

She was admitted to an acute medical ward. On examination her blood pressure was 110/70mmMg, pulse 75/minute. She was apyrexial. Her cardiorespiratory systems were unremarkable. Her cranial nerves were normal. There was evidence of mild hypertrophy of her left trapezius muscle. Associated with episodes of headache, she had a left hemiparesis with severe weakness, mild hypertonicity, hyperreflexia, and decreased sensation. These symptoms would resolve completely following an episode. Haematological and autoimmune screening was normal. Computerised tomography and later magnetic resonance imaging scans of her brain were normal. A lumbar puncture was unremarkable. Her electroencephalograph showed a mildly abnormal record. Intermittent sharpened theta and slow wave activities were noted on both temporal regions (right greater than left). These changes can be seen in migraine. Bilateral "H" responses (which have a statistical association with migraine) were noted.

A neurologist diagnosed sporadic hemiplegic migraine. She was treated with botulinum toxin type A, (DYSPORT) 40 units administered to her right and 40 units to her left forehead subcutaneously. After 7 days she described the result as "brilliant". Her migraines dramatically resolved as did her paresis. For six weeks following botulinum toxin type A injections, she suffered with only four headaches, all responding to paracetomol. Her headaches however then increased in frequency and she again suffered with left upper limb paresis for two days. Following three further similar injections, she had a similarly excellent recovery. For nine weeks she had approximately 2 mild headaches per week all of which responded to paracetomol. She now receives botulinum toxin type A injections every 9 to 12 weeks. She no longer takes any triptans or prophylactic agents.

To our knowledge this is the first case report of hemiplegic migraine responding to botulinum toxin type A.

Hemiplegic migraine occurs in two forms, sporadic and familial. It is characterised by the onset of unilateral sensory or motor signs during an episode of migraine. Neurological deficits vary from mild hemiparesis to full hemiplegia. The deficit may persist for some time after the headache. The hemiplegia may be part of the aura and last less than 1 hour or it may continue through the headache phase and last for days or weeks (1, 2).

Botulinum toxin type A has emerged as a promising option for patients suffering from chronic pain disorders (3). Its effect typically begins within 1 to 14 days, peaks within 2 to 6 weeks but may require re-treatment 12 to 16 weeks subsequently. It appears to be safe and easily tolerated (3). Adverse side effects are generally mild and reversible. The most common being unwanted weakness in adjacent muscles (4).

William Binder, an otolaryngologist and facial plastic surgeon, observed that botulinum toxin type A provided relief to migraine sufferers whom he was treating for cosmetic facial lines (5). Various open-label as well as two double-blind placebo controlled studies followed and reported promising results (6,7) using equivalent doses and position of injections of botulinum toxin type A as in the present report.

Interestingly, open label studies of botulinum toxin type A for the treatment of tension headache reported mixed results and three double blind placebo controlled studies reported no effect (8,9,10).

The association between botulinum toxin type A and pain relief was originally thought to relate only to its effect on muscle contraction (3). It has now been suggested that in migraine it might be acting by inhibiting the sensory trigeminal nerve endings, the release of pain-associated neurotransmitters, or the extracranial inflammatory process (3).

Clearly more research is required but we believe that botulinum toxin type A may be a promising treatment for hemiplegic migraines. Investigation of the mechanism of action may throw new light on the pathophysiology of such migraines.

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Botulinum Toxin Type A in the treatment of sporadic hemiplegic migraine: A case report.