We reported negative staining findings and three-dimensional structure of α-dystroglycan, however, there was a structural discrepancy between electron microscopy (negative staining) findings and three-dimensional structure. The mucin-like region using negative staining was longer than three-dimensional structure. On the other hand, three-dimensional structure of α-dystroglycan, based on negative staining results, showed the mucin-like region shorter than negative staining findings. Was the mucin-like region of negative staining unfolded? Was some structure of the mucin-like region omitted? Or both? Solution of this paradox seems to require more high-resolution structure of α-dystroglycan with atomic modeling.