Deletions of Bmp1/Tll1 result in short tooth roots and severe defects in dentin mineralization. Bmp1/Tll1 null mice display an unexpected delayed tooth eruption. Bmp1/Tll1 null mice show collagen accumulations but a reduction in non-collagenous proteins. This study provides novel evidence that Bmp1/Tll1 is required for maintenance of ECM homeostasis.