One case of sporadic hemiplegic migraine with multiple pulmonary arteriovenous malformation

Hemiplegic migraine is a rare variation of migraine with aura (MA) which should be classified as either familial hemiplegic migraine (FHM) or sporadic hemiplegic migraine (SHM) [1]. SHM is defined as migraine attacks associated with some degree of motor weakness/hemiparesis during the aura phase and no first degree relative (parent, sibling or child) has identical attacks. FHM is the only migraine subtype for which a monogenic mode of inheritance (autosomal dominant) has been clearly established to be caused by mutations in any of the following three gene loci—CACNA1A, ATP1A2 and SCN 1A [2, 3]. Some recent studies have identified involvement of these gene loci in SHM as well [2, 3]. Moreover, SHM is more difficult to diagnose and often requires several investigations to rule out other possible diseases. Friberg et al. [4] studied regional cerebral blood flow (rCBF) in three patients in whom hemiplegic migraine was induced by focal hypoperfusion developed in the frontal lobes and spread posteriorly. Pulmonary arteriovenous malformation (PAVM) is rare pulmonary vascular anomaly. Although most patients are asymptomatic, PAVM can cause dyspnoea from right-to-left shunt (RLS) and various central nervous system complications from paradoxical emboli [5]. However, there have not been reports on SHM coexisting with RLS from PAVM presented in the case.