Comparison of CNVs >100 kb in Affected versus Unaffected Individuals at Four Selected Loci Scoring Highly for Potential Pathogenicity
Duplications, deletions, and homozygous deletions are plotted blue, red, and black, respectively, in human reference assembly coordinates (x axis in each plot). Tick marks are spaced 10 Mb apart, centromeres are indicated in gray, and hotspots are shown as two green vertical lines connected by a green diagonal. Scale in bottom right indicates 1 Mb. Rearrangement hotspots that have been associated with disease are highlighted in purple. Plotting is cropped after 30 overlapping CNVs at a given locus.
(A and B) Known disease loci.
(A) 22q11-12. Disease hotspots (left to right): VCFS, critical region; VCFS, distal region, Distal 22q11 deletion syndrome (MIM 611867). 55
(B) 15q11-q14. Disease hotspots: Prader-Willi/Angelman Syndrome BP1-BP3 (MIM 176270, 105830), and 15q13.3 (MIM 612001). 20
(C and D) Candidate disease loci.
(C) 16p11-13. An inversion-containing region found in 7/8 analyzed HapMap samples 42 has been colored orange along the x axis. Disease hotspots from left to right: 16p13 deletion syndrome distal and proximal regions, 56 16p11.2-p12.2 deletion syndrome, 15 and 16p11 region associated with autism. 19,49
(D) 15q22-25. Disease hotspots from left to right: 15q24 deletion syndrome BP0-BP1, BP1-BP2, and BP2-BP3. 17