Ankyloglossia is a congenital oral anomaly characterized by an abnormally short lingual frenulum. Ankyloglossia is less common in adults as compared to infants. Sialolithiasis usually occurs between the third and fifth decades of life. The submandibular gland is the most frequently involved salivary gland. This is the first case of ankyloglossia accompanying sialolithiasis in literature which we could access. If ankyloglossia is left untreated for a long time, this condition may cause submandibular sialolithiasis. Therefore in ankyloglossia treatment, submandibular sialolithiasis must be considered.
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