In the present work, we reported 2 cases of maxillary sinus cholesteatoma and related studies. The maxillary sinus cholesteatoma is primarily characterized by compression symptoms, and sometimes there are no apparent signs. Imaging examination shows expansive swelling, compression, and absorption, and its density is slightly lower compared with the brain tissue. Surgery remains the primary therapeutic option, and appropriate surgical methods and approaches can be selected according to the location, size, and scope of lesions before the operation. Because endoscopic sinus surgery has a minor injury and does not damage the face, the prelacrimal recess approach can better expose surgical field and achieve a complete radical cure.