| Summary: | Abstract Background The lingual frenum is a fold of mucous membrane connecting the ventral tongue to the floor of the mouth. In general, lingual frenum serves multiple roles; its main function is to support the tongue and aid in limiting its movement in different directions. Any anatomical or functional deficiency of lingual frenum may have an impact on tongue functions based on its severity. Historically, the absence of lingual frenum was linked to multiple genetic and developmental conditions such as infantile hypertrophic pyloric stenosis, non-syndromic ankyloglossia diseases, and Ehlers–Danlos syndromes and was never reported in otherwise healthy individuals. Case presentation We report the absence of lingual frenum in an otherwise healthy 21-year-old Middle Eastern woman diagnosed during a routine dental examination. Conclusion To the best of our knowledge, this is the first case to be reported in the literature with similar clinical presentation. Even without a significant impact on tongue movement or speech, it is important for health practitioners to be aware of such conditions and evaluation steps for diagnosis and management.
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