There are very limited reports of aplasia of bilateral submandibular glands. We report the case of a 55-year-old male who presented with nontender palpable sublingual masses. On computed tomography and magnetic resonance imaging, bilateral submandibular gland aplasia and compensatory hypertrophy of bilateral parotid and sublingual glands were seen with herniation of hypertrophied sublingual glands through the bilateral mylohyoid muscles presenting as palpable sublingual region masses. Additional finding of bilateral accessory parotid glands was also noted. Prolapsed hypertrophied sublingual glands should be considered in patients presenting with bilateral sublingual masses to avoid unnecessary invasive procedures. We suggest radiological evaluation of such cases prior to any intervention.