Consistent with our own findings, Kolarova et al.34 also identified patients with hypomethylation defects of MEG3 and MOV10L1. While loss of imprinting at MEG3 is a known cause of Temple syndrome, the recurrent observation of hypomethylation of MOV10L1 is significant (total n = 3 of 571 cases versus 0 of 4878 controls, p = 0.001, Fisher’s Exact test), and provides additional evidence implicating this locus with developmental defects. This evidence concerns the gene MEG3 and motor developmental delay due to 14q32.2 paternally expressed gene defect.