Three different approaches to detecting the association of rare SVs with disease are shown. The association of rare structural variants with disease can be determined by comparing cases and controls with respect to the frequency of an individual mutation (single marker association, Panel a), the aggregate frequency of multiple mutations within a single gene or region (gene-wise mutational burden, Panel b), or the aggregate frequency of all rare structural variants (genome-wide mutational burden, Panel c). Panels A and B depict a typical genome browser view displaying tracks for SVs and annotated genes, c depicts an ideogram displaying the distribution of SVs genome-wide.