Migrainous infarction as a complication of sporadic hemiplegic migraine in childhood

Abstract

According to the second edition of the International Classification of Headache Disorders (ICHD-II) 1 , migrainous infarction (MI) is a complication of migraine with aura. MI is diagnosed when one or more aura symptoms last longer than 60 minutes, and neuroradiological studies demonstrate ischemic stroke that potentially explains the symptoms. Stroke should not be attributed to another disorder. The ICHD-II also defines sporadic hemiplegic migraine (SHM) as a subtype of migraine with aura associated with fully reversible motor weakness in association to the typical aura symptoms (visual, sensory or dysphasic aura). Unlike familial hemiplegic migraine (FHM), familial history of migraine with motor weakness in first-or second-degree relatives is absent. MI in childhood is very rare and epidemiological studies are scarce 2 . Reports of incidence of migrainous stroke in this group range from 0.5% 2 to 1.7% 3. A fairly and diligent review of the literature found no previous reports of MI during childhood following SHM in Brazil.Herein we report a case of MI following SHM in a child. We obtained written informed consent to publish the case, as well as the results of genetic testing. The principles outlined in the declaration of Helsinki were followed. CASEA 10-year-old boy was initially referred to our service on May of 2004, with a 2-year history of attacks of bifrontal and throbbing headache, preceded by right hemi-hypoesthesia and hemiparesis that typically lasted less than 60 minutes. Headache attacks were of moderate intensity and were associated with photophobia and phonophobia. Frequency was around 3 per month and duration ranged from 10 to 72 hours.Family reported a single episode of headache in which the associated right hemiparesis lasted about three days. Although attack was longer and clearly not suggestive of a benign migraine attack, it had not been investigated.The patient had no relevant prior medical history. Relevant familial history included mother suffering from migraine with visual aura, and brother with a past episode of transient left hemiparesis (without headache) in the context of varicella infection.Two months before the first consultation, patient suffered a partial seizure, characterized by right hemi-body hypertonia, cephalic deviation and gaze shift to the right side, during a typical attack of hemiplegic migraine. Patient recovered totally and neurological exam was normal. Magnetic resonance imaging revealed a left thalamic (pulvinar) lesion, suggestive of ischemia (Fig 1). The patient was then referred to our division for investigation and treatment.A comprehensive complementary investigation with blood tests, imaging tests, genetic tests, EEG and CSF for ischemic stroke was performed and revealed no abnormalities. Treatment with flunarizine yielded adequate control of the migraine attacks, but it was suspended due to significant drowsiness and weight gain. Patient responded well to propranolol and is free of headaches since then (headache free for 2 years). Mutation screeningScreening ...

Sporadic hemiplegic migraine in children

“…SHM in children may be complicated with persistent aura without cerebral infarction, [9] and dramatic complications may lead (however, infrequently) to migrainous infarction. [10,11] The treatment of acute SHM attacks, as well as prevention, in children remains an unresolved issue due to rarity of the disease and lack of randomized controlled trials. Thus, the management is based on the management principles of migraine with aura.…”