Relationship of ankyloglossia and obstructive sleep apnea: systematic review and meta-analysis

Purpose Recent studies have highlighted the potential role of a short lingual frenulum as a risk factor for pediatric obstructive sleep apnea syndrome. A shortened frenulum may contribute to abnormal orofacial development, leading to increased upper airway resistance and susceptibility to upper airway collapsibility during sleep. Recognizing early indicators, such as a short lingual frenulum, is crucial for prompt intervention. This systematic review aims to evaluate the association between a short lingual frenulum and the risk of obstructive sleep apnea syndrome in children. Methods This systematic review adheres to PRISMA criteria for a quantitative analysis. A comprehensive search was conducted on five databases until January 2024 to identify relevant studies. The selected articles underwent rigorous analysis, considering study design, sample characteristics, lingual frenulum characterization, sleep assessment methods, and key findings. Results A total of 239 references were initially identified. Finally, six studies were included in the qualitative synthesis, with four studies eligible for the quantitative synthesis. The Newcastle–Ottawa scale was employed to assess study quality. Meta-analysis, supported by a moderate evidence profile according to the GRADE scale, revealed statistically significant differences, with odds ratios of 3.051 (confidence interval: 1.939 to 4.801) for a short frenulum and 12.304 (confidence interval: 6.141 to 24.653) for a high-arched palate. Conclusion This systematic review and meta-analysis provide evidence supporting the association between ankyloglossia and obstructive sleep apnea in children. Nevertheless, it is crucial to consider additional factors such as tongue mobility and the presence of a high-arched palate in further evaluations. Supplementary Information The online version contains supplementary material available at 10.1007/s11325-024-03021-4.


Online Resource
Table 1.Electronic search strategy for the different databases.

Search strategy
Total (Ankyloglossia OR "lingual frenulum" OR "short lingual frenulum") AND ("obstructive sleep apnea" OR "sleep apnea" OR "sleep-disordered breathing").AUTHOR.YEAR 1a 1b 1c 2a 2b 3a 3b 3c 4a 4b 1a 1b 1c 1a 1b 1c 1d 1e 2a 2b 3a 3b 3c Is the case definition adequate?: a) Yes, with independent validation (one star); b) Yes, e.g., record linkage or based on self-report; c) No description.2) Representativeness of the cases: a) Consecutive or obviously representative series of cases (one star).b) Potential for selection biases or not stated 3) Selection of controls: a) Community controls (one star); b) Hospital controls; c) No description .4)Definition of controls: a) No history of disease (endpoint) (one star); b) No description of source.Comparability 1) Comparability of cases and controls on the basis of the design or analysis controlled for confounders: The study controls for age (one star); Study controls for other factors (list).Cohorts are not comparable on the basis of the design or analysis controlled for confounders.Exposure :1) Ascertainment of exposure: a) Secure record (e.g., surgical record) (one star); b) Structured interview where blind to case/control status (one star); c) Interview not blinded to case/control status; d) Written self-report or medical record only; e) No description; 2) Same method of ascertainment for cases and controls: Yes (one star);3) Non-response rate: a) Same rate for both groups (one star); b) Non-respondents described; c) Rate different between cases and controls with no description

Table 2 .
Quality of cohort studies assessed using the Newcastle-Ottawa Quality Assessment Form for Cohort Studies.
c) No description of the derivation of the non-exposed cohort.3) Ascertainment of exposure: a) Secure record (e.g., surgical record) (one star); b) Structured interview (one star); c) Written self-report; d) No description; e) Other.4) Demonstration that outcome of interest was not present at start of study :a) Yes ;b) No. Comparability : 1) Comparability of cohorts on the basis of the design or analysis controlled for confounders : a) The study controls for age, sex and marital status ; b) Study controls for other factors (list) ; c) Cohorts are not comparable on the basis of the design or analysis controlled for confounders .Outcome: 1) Assessment of outcome: a) Independent blind assessment; b) Record linkage; c) Self report; d) No description; e) Other.2) Was follow-up long enough for outcomes to occur: a) Yes; b) No. Indicate the median duration of follow-up and a brief rationale for the assessment above.3) Adequacy of follow-up of cohorts: a) Complete follow up-all subject accounted for; b) Subjects lost to follow up unlikely to introduce bias-number lost less than or equal to 20% or description of those lost suggested no different from those followed.(one star); c) Follow up rate less than 80% and no description of those lost; d) No statement.

Table 3 . Quality of cohort studies assessed using the Newcastle-Ottawa Quality Assessment adapted for cross sectional studies.
does not investigate potential confounders.Outcome: (Maximum 3 stars) 1) Assessment of the outcome: a) Independent blind assessment. 2 scores; b) Record linkage. 2 scores; c) Self report. 1 score; d) No description.0 score 2) Statistical test: a) The statistical test used to analyze the data is clearly described and appropriate.1 score; b) The statistical test is not appropriate, not described or incomplete.0 score