Taken alongside evidence that CCDC22(T17A), CCDC22(Y557C), and VPS35L(A830T) perturb endosomal recycling of LRP1 and LDLR and lead to hypercholesterolemia,11,65 these structural data provide a molecular explanation for the perturbed stability and assembly of the Commander complex associated with XLID and RSS. Here, CCDC22 is linked to Hypercholesterolemia.