As the functional consequences of the aforementioned genetic alterations of DYNC1LI1, SRGAP3, and OR7E24 in DIPG are unknown (and the three patients with DYNC1LI1-mutant tumors had distinct mutations), these findings should be interpreted cautiously and further conclusions are limited; however, future investigation into the role that DYNC1LI1, SRGAP3, and OR7E24 aberrations may play in DIPG, in regard to potential tumorigenesis, response to irradiation, and/or prognosis, will be critical. Here, SRGAP3 is linked to diffuse intrinsic pontine glioma.