A recent study identified several de novo missense mutations in the chromodomain and the acetyl-CoA binding site of the histone acetyltransferase KAT5/TIP60 (henceforth referred to as KAT5) as cause for a syndromic form of orofacial clefting.13 The presence of other facial dysmorphisms points to a broader role of KAT5 in the neural crest-derived mesenchyme. Here, KAT5 is linked to orofacial cleft.