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Case Reports
. 2024 Feb 8;11(2):218.
doi: 10.3390/children11020218.

Ankyloglossia in Children, a Cause of Obstructive Sleep Apnoea: Case Report of Paediatric Ankyloglossia and Sleep Apnoea: DISE Resolves the Mystery

Affiliations
Case Reports

Ankyloglossia in Children, a Cause of Obstructive Sleep Apnoea: Case Report of Paediatric Ankyloglossia and Sleep Apnoea: DISE Resolves the Mystery

Johanna Ximena Valderrama-Penagos et al. Children (Basel). .

Abstract

Tongue mobility is an obstructive sleep apnoea (OSA) marker and myofunctional therapy (MFT) target. For this reason, all paediatric patients with sleep-disordered breathing should require a combined functional assessment from an ear, nose, and throat (ENT) specialist and a phonoaudiologist to confirm or rule out the presence of ankyloglossia. To our knowledge, this is the first case of a 13-year-old girl diagnosed with severe OSA and a significant decrease of 94% in her apnoea index (AI), requiring frenotomy with an immediate postoperative change in the tongue position. A drug-induced sleep endoscopy (DISE) was performed before and immediately postfrenotomy, and the anatomical changes provoked by this surgery during sleep were confirmed for the first time.

Keywords: ankyloglossia; drug-induced sleep endoscopy; frenulectomy; sleep apnoea.

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Conflict of interest statement

The authors declare no conflicts of interest.

Figures

Figure 1
Figure 1
Ankyloglossia associated with teeth malposition.
Figure 2
Figure 2
The surgical protocol used to code the DISE results according to the VOTE scale is as described in reference. a Degree of obstruction has one number for each structure: 0, Noobstruction (no vibration); 1, Partial obstruction (vibration); 2, Complete obstruction (collapse); X, Not visualized. b Oropharynx obstruction can be distinguished as related solely to thetonsils or including the lateral walls, with or without a tonsillarcomponent. c Configuration noted for structures with degree of obstructiongreater than.

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References

    1. Guilleminault C., Huseni S., Lo L. A frequent phenotype for paediatric sleep apnoea: Short lingual frenulum. ERJ Open Res. 2016;2:00043-2016. doi: 10.1183/23120541.00043-2016. - DOI - PMC - PubMed
    1. Pauws E., Peskett E., Boissin C., Hoshino A., Mengrelis K., Carta E., Abruzzo M.A., Lees M., Moore G.E., Erickson R.P., et al. X-linked CHARGE-like Abruzzo-Erickson syndrome and classic cleft palate with ankyloglossia result from TBX22 splicing mutations. Clin. Genet. 2013;83:352–358. doi: 10.1111/j.1399-0004.2012.01930.x. - DOI - PubMed
    1. Messner A.H., Walsh J., Rosenfeld R.M., Schwartz S.R., Ishman S.L., Baldassari C., Brietzke S.E., Darrow D.H., Goldstein N., Levi J., et al. Clinical Consensus Statement: Ankyloglossia in Children. Otolaryngol. Neck Surg. Off. J. Am. Acad. Otolaryngol. Neck Surg. 2020;162:597–611. doi: 10.1177/0194599820915457. - DOI - PubMed
    1. Villa M.P., Evangelisti M., Barreto M., Cecili M., Kaditis A. Short lingual frenulum as a risk factor for sleep-disordered breathing in school-age children. Sleep Med. 2020;66:119–122. doi: 10.1016/j.sleep.2019.09.019. - DOI - PubMed
    1. O’Connor-Reina C., Garcia J.M.I., Alcala L.R., Ruiz E.R., Iriarte M.T.G., Morente J.C.C., Baptista P., Plaza G. Improving adherence to myofunctional therapy in the treatment of sleep-disordered breathing. J. Clin. Med. 2021;10:5772. doi: 10.3390/jcm10245772. - DOI - PMC - PubMed

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