A 15-year-old girl, operated upon (surgical ankyloglossia) at birth for glossoptosis due to Pierre Robin's syndrome provoking respiratory distress, presented with lithiasis of Wharton's duct. The pathogenesis of the lithiasis is considered to be related to the wounds and scars following the sublingual surgery, findings in this case also strongly suggesting the role of mechanical and inflammatory factors in the etiology of salivary gland lithiasis in general.