A frequent phenotype for paediatric sleep apnoea: short lingual frenulum

Abstract

A short lingual frenulum has been associated with difficulties in sucking, swallowing and speech. The oral dysfunction induced by a short lingual frenulum can lead to oral-facial dysmorphosis, which decreases the size of upper airway support. Such progressive change increases the risk of upper airway collapsibility during sleep. Clinical investigation of the oral cavity was conducted as a part of a clinical evaluation of children suspected of having sleep disordered breathing (SDB) based on complaints, symptoms and signs. Systematic polysomnographic evaluation followed the clinical examination. A retrospective analysis of 150 successively seen children suspected of having SDB was performed, in addition to a comparison of the findings between children with and without short lingual frenula. Among the children, two groups of obstructive sleep apnoea syndrome (OSAS) were found: 1) absence of adenotonsils enlargement and short frenula (n=63); and 2) normal frenula and enlarged adenotonsils (n=87). Children in the first group had significantly more abnormal oral anatomy findings, and a positive family of short frenulum and SDB was documented in at least one direct family member in 60 cases. A short lingual frenulum left untreated at birth is associated with OSAS at later age, and a systematic screening for the syndrome should be conducted when this anatomical abnormality is recognised.

FIGURE 1

Measurement of the frenulum using the commercially available Quick Tongue Tie Assessment Kit (Neo Health Services Inc., Coconut Creek, FL, USA). a) Normal frenulum [9]; b) free tongue [7]. Complete clinical protocols for lingual frenulum investigations for infants [13] and children–adolescents [25] have been published.

FIGURE 2

a–h) Examples of short frenula in children and teenagers. Consideration was given only to shortness of the frenulum and not difference in presentation of the frenulum. Histological studies have shown that different types of fibres may be present, depending on the individual. Short frenula were found in e) a 40-year-old mother and f) her 14-year-old daughter. The other subjects are aged 3–16 years. All subjects have obstructive sleep apnoea syndrome.