Identification and analysis of gene mutations in familial hemiplegic migraine (FHM) remains the focus of research of migraine genetics. Several new mutations have recently been identified in FHM1, FHM2 and FHM3 genes. Functional studies further revealed a major role for disturbed ion transport in this disorder. The findings point to an important role for cortical spreading depression in migraine pathophysiology. New genetic approaches have been tested in common migraine: novel chromosomal loci, but no gene variants have been identified.The identification of TREX1 mutations in families with retinal vasculopathy and associated diseases such as migraine may provide new insights in migraine pathophysiology.