SH3BP2 and cherubism: Previously, we have shown that heterozygous mutations in the adaptor protein SH3BP2 are responsible for cherubism.1 In the cherubism mouse model, the homozygous mutation is required to exhibit spontaneous jawbone destruction by inflammation.2 However, homozygous mice develop a phenotype that extends beyond the craniofacial skeleton, which is much more severe than human cherubism and has not been observed in human cherubism patients.