Is Ankyloglossia Correlated With Pediatric Sleep Disordered Breathing? A Systematic Review

ABSTRACT

Objectives

Sleep disordered breathing (SDB) affects 2%–11% of children, predisposing them to neurobehavioral and developmental consequences. Ankyloglossia has been proposed as a risk factor for SDB, and frenotomy as a treatment for SDB in children with ankyloglossia. With increasing ankyloglossia diagnoses, it is critical to evaluate the evidence for a linkage between SDB and ankyloglossia.

Data Sources

EMBASE, Web of Science, Medline, CINAHL, CCRCT, and SCOPUS were searched from inception to February 13, 2025. Publications assessing the relationship between ankyloglossia and SDB in non‐syndromic children ages 0 to 18 years were included. Eight studies involving 1171 patients met inclusion criteria.

Review Methods

Two reviewers independently screened abstracts and full texts for inclusion. Strength of clinical data was graded according to the Cochrane Risk of Bias Assessment and modified Newcastle–Ottawa Scale.

Results

There is mixed evidence of a relationship between ankyloglossia and pediatric SDB. The lack of standardized diagnostic criteria for ankyloglossia and the use of surveys instead of validated clinical assessment tools to assess SDB limit the generalizability of findings. There is also insufficient data to conclude that frenotomy is indicated in managing SDB in children with ankyloglossia. While two interventional studies report a positive association, their results have limited validity and generalizability.

Conclusion

There is an unclear relationship between ankyloglossia and pediatric SDB and insufficient evidence to determine if frenotomy is indicated as a treatment for SDB in children with ankyloglossia. Higher quality studies with standardized functional measures of ankyloglossia and validated assessment of SDB are needed.

Level of Evidence

N/A.

An increasing breadth of conditions and symptoms are being ascribed to ankyloglossia. As diagnosis of ankyloglossia and frequency of frenotomy exponential increase, it is important to have an understanding of the associated evidence. This systematic review summarizes the evidence for ankyloglossia's association with sleep disordered breathing in pediatric patients, and determines the link is currently uncertain.

1. Introduction

Ankyloglossia, commonly known as tongue‐tie, is a clinical diagnosis characterized by a congenitally short lingual frenulum with restriction of tongue movement [1]. Concerns have been raised regarding the potential implications of a restrictive lingual frenulum on functions such as breastfeeding, sleep, and speech amongst children, potentially leading to adverse health outcomes. Despite more recent attention to ankyloglossia, and a reported incidence rate of 2.8%–10.7% [2, 3], the relationship between the structure of the lingual frenulum and many functional limitations in children remains uncertain [4]. There is an increasing prevalence of ankyloglossia diagnosis and frenotomies performed with expanded indications [1, 5, 6, 7], as well as variability in the diagnosis of ankyloglossia with potential for overdiagnosis [4].

In particular, the association between ankyloglossia and pediatric sleep‐disordered breathing (SDB), including obstructive sleep apnea (OSA), remains unclear. The suggested mechanism for this association is that restricted tongue mobility prevents the tongue from adequately resting against and shaping the palate, potentially impairing the development of the dental arches, particularly the maxilla [8]. This abnormal maxillary growth might result in maxillary hypoplasia or a high‐arched palate, subsequently impairing nasal breathing and increasing upper airway collapsibility, resulting in disordered sleep [8]. The lack of consensus on the role of ankyloglossia in SDB in children has led to confusion and management controversies among clinicians and parents [1]. SDB affects 2% to 11% [9] of children, predisposing them to neurobehavioral consequences such as excessive daytime sleepiness, hyperactivity, anxiety, inattentiveness, and decreased school performance [10].

Frenotomy to release a short lingual frenulum has been proposed as a treatment option for SDB by some practitioners [11]. However, a surgical procedure to correct ankyloglossia is invasive, and with uncertain efficacy in pediatric SDB, this practice warrants further research. The objective of this systematic review was to better understand the relationship between ankyloglossia and SDB, with the goal of informing clinicians and facilitating definitive practice guidelines when children have concerns for both conditions.

2. Methods

This systematic review was reported in accordance with the Preferred Reporting Items for Systematic Reviews and Meta‐Analysis Protocols (PRISMA) guidelines [12]. The study protocol was registered in the PROSPERO database (ID: CRD42022341286) before initiation.

2.1. Search Strategy

Published literature assessing the relationship between ankyloglossia and SDB in non‐syndromic children ages 0 to 18 years was identified by searching the following electronic databases from inception to February 13, 2025: EMBASE, Web of Science, Medline, CINAHL, CCRCT, and SCOPUS (Full search strategy included as Supporting Information). Only literature published in English was included. Case series, retrospective cohort studies, case‐control studies, prospective cohort studies, and randomized control trials were included. Opinion papers, technical reports, editorials, literature reviews, and case reports were excluded. Inclusion and exclusion criteria are summarized in Table 1. For the primary analysis of the correlation between ankyloglossia and SDB, the following “PEO” statement was used: Population: Children under 18 years of age; Exposure: presence of ankyloglossia; Outcome: sleep‐disordered breathing. For the secondary analysis of the impact of frenotomy, we utilized the following “PICO” statement: Participants: Children under 18 years of age; Intervention: frenotomy; Control: no surgery; Outcome: sleep‐disordered breathing.

TABLE 1.

Inclusion and exclusion criteria utilized during the screening process.

Category	Inclusion criteria	Exclusion criteria
Study population	Pediatric patients (< 18 years) Diagnosis of ankyloglossia Patients are being assessed for SDB (including OSA) Patients with or without surgical treatment (i.e., frenotomy, frenulectomy, frenuloplasty), medical treatment, speech therapy and osteopathy	Syndromic patients (i.e., those with Trisomy 21, craniofacial abnormalities, Pierre Robin Sequence, etc.)
Publication languages	English	Non‐English
Study design	Case series, retrospective cohort studies, case control studies, prospective cohort studies, experimental studies, randomized control trials	Opinion papers, technical reports, editorials, literature reviews and case reports

2.2. Search Selection and Data Extraction

Screening was completed using Covidence (Covidence systematic review software, Veritas Health Innovation, Melbourne, Australia. Available at www.covidence.org). Two independent reviewers assessed abstracts for eligibility, then reviewed full texts for inclusion in the study. Reviewers were blinded to each other's decisions and met to resolve disputes through consensus. Relevant data were extracted from included studies following standardized tables developed a priori by two independent reviewers. Any unresolved disputes were brought to the senior author for resolution.

2.3. Data Analysis

The methodological quality of identified randomized clinical trials was determined using the Cochrane Risk of Bias Assessment [12]. Studies with cross‐sectional, experimental, and prospective observational designs were analyzed with the modified Newcastle–Ottawa Scale [13].

3. Results

3.1. Search Results

A total of 123 abstracts were identified, of which 22 were included for full‐text review. Eight studies, involving a total of 1171 patients, met inclusion criteria. The following study types were included: five cross‐sectional, one RCT, one prospective observational, and one experimental (Figure 1).

FIGURE 1.

Prisma flow diagram. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com]

Sample size across studies ranged from 32 to 504, with age ranging from 13 months to 17 years. Two studies examined the impact of a lingual frenotomy on sleep outcomes [14, 15], whereas the remaining studies examined the association of SDB and ankyloglossia without surgically addressing the lingual frenulum. Study characteristics are reported in Table 2.

TABLE 2.

Study characteristics.

Source, trial design, country	Financial disclosures	N	Age range (Years) [mean]	AG criteria	SDB criteria	Outcome
Brożek‐Mądry 2021, case–control, Poland	None	135	4–17 [NR]	Length of free tongue ≤ 16 mm	PSQ ≥ 8	Significant association (p = 0.0033) between free tongue length ≤ 16 mm and SDB as assessed by PSQ
Burska 2022, case–control, Poland	None	131	3–17 [NR]	Length of free tongue ≤ 16 mm	PSQ ≥ 8	Significant association (p = 0.011) between free tongue length ≤ 16 mm and SDB as assessed by PSQ
Cohen‐Levy 2020, Prospective Observational, Canada	Yes, (SickKids Foundation, Toronto, Canada)	100	2–17 [8.78]	Limited tongue elevation in relation to maximal aperture of 60% or less	HSAT; REI	No significant association (p = 0.281) between tongue mobility and REI
Villa 2019, case–control, Italy	None	504	6–14 [9.6]	Length of free tongue ≤ 16 mm	SCR ≥ 6.5	Significant association (p < 0.002) between free tongue length ≤ 16 mm and SDB as assessed by SCR
Guilleminault 2016, case–control, USA	None	150	3–12 [NR]	Length of free tongue ≤ 16 mm	PSG; AHI and SpO2 nadir	In univariate analysis, significant (p = 0.025) association between ankyloglossia and AHI/lower SpO2 nadir. In multivariate analysis controlling for age, sex, symptoms of OSA (fatigue, EDS, inattention/hyperactivity), anatomy (high palatal vault, Friedman score, Mallampati score), no significant association (p > 0.05) between ankyloglossia and AHI/lower SpO2 nadir.
Yuen 2022, case–control, Hong Kong	None	82	5–12 [8.3]	Length of free tongue ≤ 16 mm, TRMR	PSG; presence of OSA defined as ≥ 1 obstructive, mixed apneas or hypopneas per hour of sleep on PSG	Significant association (p = 0.019) between ankyloglossia as measured by TRMR and presence of OSA Significant association (p = 0.018) between free tongue length ≤ 16 mm and presence of OSA
Fioravanti 2021, RCT, Italy	Yes (Pediatric Dentistry Unit, Policlinico Umberto I)	32	4–13 [NR]	Class III–V by Kotlow; Grades 2 and 3 by Ruffoli	PSG, graded into mild, moderate and severe by unclear metrics	Significant improvement (p < 0.0001) in OSA severity for patients who were randomized to frenotomy
Baxter 2020, experimental, USA	None	37	1–12 [4.2]	Length of free tongue ≤ 16 mm	Pre‐procedural non‐standardized subjective parental reports of patient sleeping behaviors. 1 week and 1 month post‐frenectomy likert scale evaluation by parents of changes in same sleeping behaviors	Significant improvements (p < 0.005) in the behaviors “Wakes tired and not refreshed,” “Snoring,” “Gasping for air” following frenotomy

Abbreviations: AG: ankyloglossia; AHI: apnea/hypopnea index; HSAT: home sleep apnea test; OSA: obstructive sleep apnea; PSQ: Pediatric Sleep Questionnaire; REI: respiratory event index; SCR: sleep clinical record; SDB: sleep disordered breathing; SpO₂ nadir: lowest value of arterial oxyhemoglobin saturation as measured by pulse oximetry; TRMR: tongue range of motion ratio.

The population examined by each study varied substantially, with differing inclusion and exclusion criteria and differing participant demography in terms of ethnicity, mean age range, and gender distribution. Details regarding selection and population characteristics are reported in Table 3. Studies also varied in the physical and functional attributes examined, including measures such as BMI, oral cavity assessments, inattention, daytime somnolence, and quality of life. A description of structural and non‐structural factors analyzed as well as the measures of SDB are defined as Tables S1 and S2.

TABLE 3.

Clinical characteristics of included patients.

Study	Inclusion	Exclusion	% female	% white	Controlled demographic factors	Uncontrolled demographic factors	Controlled structural/clinical differences	Uncontrolled structural differences
Brożek‐Mądry 2021	Children living in rural Poland	Congenital abnormalities	51	100	Age, sex, height, weight, mean BMI, age‐stratified BMI %ile	None	Inter‐incisor distance	HFP (SG: 77.2 ± 20.0, CG: 70.2 ± 18.1; p value: 0.0306) High arched palate (SG: 24%, CG: 6%; p value 0.0033)
Burska 2022	Children living in rural Poland	Congenital abnormalities	51	100	Age, sex, height, weight, BMI (control method unspecified)	None	None	Prevalence of grade on Mallampati scale (p < 0.001) High arched palate (SG: 21.5%, CG: 6.1%; p < 0.001) Crossbite (SG: 40%, CG: 6.1%; p < 0.001)
Cohen‐Levy 2020	Symptomatic children; adenoid or tonsil surgery; chronic snoring or witnessed apneas; agreed to cranio‐facial examination. Chronic snorers or HSCS > 2.72	Syndromic or compromised medical condition	37	53	No control	No control	No control	No control
Villa 2019	Children recruited from a school in Rome who correctly filled out the SCR questionnaire	History of acute/chronic cardiorespiratory or neuromuscular disease; chronic inflammatory disease; major craniofacial abnormalities, chromosomal syndromes; epilepsy	56	Not provided	Age, sex, BMI percentile	None	Tongue strength and endurance	Malocclusion (SG: 88.1%, CG: 44.8%; p < 0.001) Nasal septum deviation (SG: 23.8%, CG: 7.5%; p < 0.001) Arched palate (SG: 95.2%, CG: 49.7%; p < 0.001) Oral breathing (SG: 61.9%, CG: 10.2%; p < 0.001) Adenotonsillar hypertrophy (SG: 17.1%, CG: 42.8%; p < 0.001)
Guillemin‐ault 2016	Non‐syndromic children referred to a clinic for “sleep disorders” diagnosed with OSA by PSG.	Syndromic, neuromuscular or major psychiatric disorders, craniofacial syndromes, major pediatric illnesses, BMI > 28.	39	Not provided	Sex	Age (SG: 9.88, CG: 8.05; p = 0.0015)	Symptoms including fatigue, EDS, inattention/hyperactivity	High arched palate (SG: 80%, CG: 9%; p = 0.0001) Average friedman score (SG: 1.8, CG: 3.2; p = 0.0001) Average mallampati score (SG: 3.4, CG: 2.9; p = 0.0001)
Yuen 2021	Non‐syndromic prepubertal children + suspected OSA after presentation to sleep disorder clinic + admitted for PSG.	Previous surgical treatment, obesity	30	0	Age, weight, height, BMI z‐score, circumference of neck, waist and hip	None	Allergic rhinitis, bruxism, turbinate size, tonsil size, inter‐incisor distance	None
Fioravanti 2021	Diagnosis of OSAS and a short lingual frenum (class III–V by Kotlow; Grades 2 and 3 by Ruffoli)	Contraindications to administration of local anesthetic, Previous frenotomy, did not consent	43	Not provided	Age, gender, nationality	None	OSA intensity (mild, moderate, severe)—grading criteria not provided	None
Baxter 2020	Children with speech, feeding, and sleep issues, + short lingual frenum (class II–V by Kotlow)	Previous frenotomy, did not consent	38	Not provided	No control	No control	No control	No control

Abbreviations: BMI: body mass index; CG: control group; EDS: excessive daytime sleepiness; HFP: head forward position; HSCS: Hierarchical Severity Clinical Scale; OSA: obstructive sleep apnea; PSG: polysomnography; SG: subject group.

3.2. Ankyloglossia Assessment

Seven studies utilized the Kotlow scale to define ankyloglossia, with six defining a short lingual frenulum as a length of free tongue ≤ 16 mm (Kotlow class ≥ I) [8, 14, 16, 17, 18, 19, 20]. Fioravanti et al. only included those with Kotlow class III–V and Ruffoli grade 2 and 3 [15]. Baxter et al. evaluated the lingual frenulum using the Kotlow rating scale, but lingual frenulum parameters were not reported [14]. Cohen‐Levy et al. defined a short lingual frenulum as limited tongue elevation in relation to “maximal aperture” of 60% or less [18]. In addition to assessing free tongue length, Yuen et al. also assessed the tongue range of motion ratio (TRMR) ratio [20]. An inventory of measures of ankyloglossia and other tongue or airway measures considered by the studies are included in Table S3.

3.3. Clinical Presentation

There was significant heterogeneity amongst the studies with respect to outcome measures, and specifically their definition of SDB. Three studies utilized polysomnography (PSG) [15, 19, 20]. Two of these studies assessed disease severity with the average apnea/hypopnea index (AHI) and SpO₂ nadir [19, 20]. Fioravanti et al. utilized the PSG to classify disease severity as mild/moderate/severe; however, they did not provide detail regarding AHI in groups or stratification criteria based on AHI [15]. Cohen‐Levy et al. assessed SDB using a home sleep apnea test (HSAT), which is a home polygraph assessment validated in adults but not children [18, 21].

Three case control studies utilized validated questionnaires or clinical assessments to identify patients with SDB and determine case assignment. Brożek‐Mądry et al. and Burska et al. employed the Pediatric Sleep Questionnaire (PSQ), a symptom questionnaire completed by parents/caregivers [16, 17, 22]. Villa et al. used the sleep clinical record (SCR), a clinical assessment that includes both a symptom survey and physical examination component [8, 23]. Baxter et al. utilized a non‐validated survey based on caregiver reports to identify sleep‐related issues during participant recruitment [14]. To measure post‐procedure outcomes, caregivers were posed the same questions with Likert scale responses regarding change from baseline.

3.4. Treatment

Both experimental studies reviewed herein performed frenotomy using lasers with post‐operative myofunctional exercise. Baxter et al. utilize a CO₂ laser while Fioravanti et al. utilize a diode laser at 90 nm, 7.5 W, 25 Hz [14, 15].

3.5. Study Findings and Outcomes

Three observational studies reported a significant association between a short lingual frenulum and SDB [8, 16, 17]. Yuen et al. did not report a significant association between short lingual frenulum and SDB but did note a significant negative association between “tongue mobility” and SDB [20]. Guilleminault et al. reported a significant association between ankyloglossia and SDB with univariate analysis, though this association became insignificant with multivariate analysis [19]. Cohen‐Levy et al.'s observational study also did not report a significant association [18].

Both the experimental studies reported improvement in SDB measures following laser frenotomy [14, 15]. Fioravanti et al. reported significant improvement in PSG‐derived clinical grade of SDB (mild/moderate/severe) in the experimental group while Baxter et al. found significant improvement in parental reports of “snoring”, “gasping for air” and “waking tired and not refreshed” following the procedure [14, 15]. Findings and outcomes for individual studies are summarized in Table 2.

Amongst the three observational studies with PSG‐derived outcomes, one found a significant association between ankyloglossia and SDB. All three observational studies utilizing questionnaires to assess SDB found significant relationships between ankyloglossia and SDB.

3.6. Quality Assessment

A risk of bias assessment [24] demonstrated that Fioravanti et al.'s randomized control study was of poor quality (Figure 2). The study reported a double‐blinded design with computer‐generated randomization and inclusion of all participant outcomes that limited selection and attrition bias. While participants and health care workers were blinded to assignment, given the invasive nature of the intervention, steps such as including a sham procedure would be required to maintain blinding. Given this aspect of blinding is not addressed, insufficient blinding likely led to performance and detection bias. Without the initial study protocol including planned analyses, attrition bias could not be assessed.

FIGURE 2.

Results of Cochrane collaboration's tool for assessing risk of bias, used for the sole included RCT, Fioravanti et al. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com]

The Newcastle‐Ottawa quality assessment of the remaining studies demonstrated three had fair quality and four had poor quality (Table 4). Brożek‐Mądry et al., Burska et al., and Villa et al. lacked response rates, ascertainment of exposure, and case definition. These studies recruited with surveys leading to poor response rate. They also utilized free length of tongue to determine exposure, a metric that is only moderately correlated with restricted tongue movement [23]. Additionally, assessment of SDB status was performed with symptom questionnaires or clinical assessments with varying degrees of validity compared with the gold‐standard PSG.

TABLE 4.

Modified Newcastle‐Ottawa quality assessment of non‐RCT studies. Each category is rated out of either one or two, with “0” indicating a score of 0, “★” indicating a score of 1, and “★★” indicating a score of 2. The maximum score for each category is indicated below the category name.

Study	Selection				Comparability	Outcomes
	Representatives of sample (Max ★)	Sample size justified (Max ★)	Response rate (Max ★)	Ascertainment of exposure (Max ★★)	Confounding controlled (Max ★★)	Outcome assessment (Max ★★)	Statistical test (Max ★)
Brożek‐Mądry 2021	★	★	0	★	★★	★	★
Burska 2022	★	★	0	★	★	★	★
Cohen‐Levy 2020	0	★	0	★★	N/A a	★★	★
Villa 2019	★	★	★	★	★★	★	★
Guilleminault 2016	0	★	★	★★	0	★★	★
Yuen 2021	0	0	★	★★	★★	★★	★
Baxter 2020	N/A b	N/A b	0	★	N/A a	0	★

^a Confounding could not be controlled in prospective observational and experimental studies given the lack of a control group.

^b Experimental studies are designed to draw from a symptomatic population and, by the nature of the invasive nature of the intervention, cannot have as large sample sizes as case–control studies; thus, these aspects cannot be assessed.

Villa et al. and Guilleminault et al. utilized well‐validated or gold‐standard methods for evaluating ankyloglossia and SDB and hence were of fair quality. While the HSAT test by Cohen‐Levy et al. is not validated for use in evaluating SDB in children, the study was considered fair quality given the test's validity in adults. However, these studies lack representativeness as they do not draw from the general pediatric population. Patients were instead recruited from those presenting to sleep clinics or those who had already undergone structural surgery, which limits the generalization of these studies' findings.

The experimental design of Baxter et al. limits evaluation by the Newcastle‐Ottawa quality assessment tool. Nonetheless, the study is of poor quality due to its unclear exposure and biased outcome definition. The free tongue length parameter used to diagnose ankyloglossia is not specified, and the results of the unvalidated symptom survey used as an outcome are unlikely to correlate to the incidence of true clinical SDB.

4. Discussion

There have been conflicting reports regarding the association between ankyloglossia and SDB, thereby creating confusion on best management practices when a child with a diagnosis of ankyloglossia presents with SDB or a child with SDB is suspected to have concomitant ankyloglossia [1, 4]. The papers included in this systematic review report a positive association between ankyloglossia and SDB; however, it is not possible to conclude an association is present given the lack of high‐quality RCT trials as well as poor design of existing studies. The studies evaluated have high heterogeneity in patient population (Table 3), and vary in the reporting of key outcomes, especially definitions of ankyloglossia and SDB (Table 2), limiting the comparison and interpretation of data between studies.

While there is increasing consensus that pathological ankyloglossia is defined by functional limitation in tongue movement [1], there is limited agreement on a standardized assessment tool. Functional assessments such as the Tongue‐tie and Breastfed Babies (TABBY) or Hazelbaker assessment tool have been shown to be reliable and clinically valid measures of tongue mobility in young infants, though they have more limited generalizability outside of the first 6 months of life [25, 26]. Functional tests of tongue mobility in older children include the maximal interincisal mouth opening (MIO), mouth opening with tongue tip to maxillary incisive papillae at roof of mouth (MOTTIP), tongue range of motion deficit (equal to MIO—MOTTIP), and tongue range of motion (equal to MOTTIP/MIO) [27]. These functional assessments are arduous to perform, leading many clinicians to assess anatomical measurements between landmarks as a proxy for tongue mobility [28, 29]. In particular, the Kotlow scale assesses the length of the free tongue while the Ruffoli scale assesses the length of the frenulum [28, 29]. Yoon et al. showed the Kotlow scale demonstrates only modest correlation with functional assessments of tongue mobility and that the Ruffoli scale is dependent on age and height [27]. As a result, these anatomic assessments are not ideal measures of ankyloglossia as compared to direct measures of tongue mobility. In summary, all current measures of ankyloglossia have limitations, and “gold standard” metrics do not presently exist.

The majority of studies examined in this review utilize the Kotlow scale to diagnose ankyloglossia, with only two—Cohen‐Levy et al. and Yuen et al. utilizing direct assessments of tongue mobility (tongue elevation and TRMR respectively). The variability in definitions makes it challenging to compare results and associations with SDB between studies. In addition, the reliance on the Kotlow scale as an imperfect proxy measure of tongue mobility and hence ankyloglossia weakens the internal validity of these studies. Future studies should consider utilizing a measure of ankyloglossia that directly measures tongue movement, and especially a functional metric of tongue mobility to increase finding validity and generalizability. In addition, the presence of ankyloglossia was presented as a binary variable in all studies when the severity of ankyloglossia can vary dramatically—future studies can consider exploring the association between a continuous measure of ankyloglossia severity and SDB to better define a link between the two, if one exists.

PSG is the gold standard for OSA assessment; however, it remains a costly and resource‐intensive study. Perhaps as a result, only three of the eight studies examined were able to definitively confirm a diagnosis of OSA with PSG. Another study, Cohen‐Levy et al., used validated ambulatory polysomnography with reported efficacy similar to PSG [21]. Three of the remaining studies used a symptom questionnaire (PSQ) or clinical assessment (SCR) as a proxy measure of SDB. While both assessments fare well in validation against the PSG (PSQ Sensitivity: 0.85, Specificity: 0.87; SCR Sensitivity: 0.96, Specificity 0.67) (Table S2), the use of non‐clinical proxy measures limits these studies' validity. The use of different outcome metrics makes comparison between studies difficult. Even among the studies with PSG‐derived outcomes, there was significant variance in the specific outcome metric being evaluated (AHI, REI, ODI, SpO₂ nadir). For instance, the RCT Fioravanti et al. reported outcomes from the PSG as mild/moderate/severe without clarifying the stratification method, preventing clear comparison with other studies that reported specific PSG metrics.

Frenotomy is an option for the treatment of ankyloglossia, if the condition is present and the symptoms of concern can be attributed to a restrictive lingual frenulum [30]. Due to limited literature available in the pediatric population, only two studies examining the effect of frenotomy on SDB were included in our review. Data were insufficient to conclude that laser frenotomy is indicated in managing SDB in children with ankyloglossia despite a positive association reported in both studies. Although Fioravanti et al. performed a randomized, double‐blinded, controlled clinical study and utilized the PSG for diagnosis of SDB, the methodology was poor, blinding was not possible despite being described, and objective outcome measures and diagnostic measures were not available. Baxter et al. unclearly defined “symptomatic tongue restriction,” did not randomize participants and used non‐standardized parental reports for SDB symptoms. Patients who underwent frenotomy in both studies were followed up for a maximum of 1 month. Fioravanti et al. did not describe complications, and Baxter et al. reported few instances of parents who reported worsening of sleep symptoms.

Beyond ankyloglossia, many of the studies explored the relationship between other structural and non‐structural factors and SDB. In particular, the three studies that analyzed arched palate and adenotonsillar hypertrophy, known structural factors associated with OSA [31], found their presence significantly associated with SDB (Table 3). Despite the number of structural factors examined, no study examined the association between ankyloglossia and known structural variants associated with SDB. An example is the variation in palatal dimensions, which present potential confounding anatomical variables when assessing the impact of the lingual frenulum [4]. Future studies should consider controlling for anatomical factors in addition to tongue mobility in order to evaluate for a specific causative link between ankyloglossia and SDB.

The comprehensive search strategy employed in this study involving multiple databases, a wide range of search terms, and the inclusion of both observational and interventional studies allowed for a broad examination of available evidence. However, this approach also introduced significant heterogeneity in study design and outcomes, which made direct comparison challenging and meta‐analysis impossible. Additionally, the wide age range of pediatric patients under consideration (13 months‐18 years) and wide range of ages included in each study (Table 2) limit comparison, as the impact of ankyloglossia on SDB may vary significantly across developmental stages from infancy to adolescence. Future studies should consider including subgroup analysis by age cohort to allow for better characterization of the impact of ankyloglossia on SDB in each age group. The exclusion of non‐English language studies may have resulted in relevant research from non‐English speaking countries being overlooked. Despite these limitations, this review provides a complete synthesis of the current state of knowledge regarding the association between ankyloglossia and SDB in children, identifying gaps in the existing literature and highlighting high‐value areas for future investigation. Our systematic review builds upon prior scoping reviews, such as that by Codray et al., by providing a detailed methodological critique of the existing evidence linking ankyloglossia to pediatric SDB by specifically evaluating study quality, diagnostic standardization gaps, and frenotomy's potential therapeutic role. In doing so, we have been able to provide specific evidence‐based recommendations for future high‐quality trials.

5. Conclusion

There is insufficient quality evidence to determine if ankyloglossia is correlated with SDB and further whether frenotomy is indicated for treatment of SDB in children with ankyloglossia. Existing studies are notable for limited quality and small sample sizes. Synthesis of findings between studies is challenging given the lack of a standardized definition of ankyloglossia and the use of non‐validated assessments of SDB. Additional studies utilizing functional and validated assessments of ankyloglossia and PSG, as well as randomized clinical studies for frenotomy, will be needed to clarify the clinical importance of ankyloglossia in pediatric SDB as well as the role of frenotomy in the treatment of the condition.

Conflicts of Interest

The authors declare no conflicts of interest.

Supporting information

Table S1: Inventory of anatomical measures used across all studies.

Table S2: Inventory of SDB measures used across all studies.

Table S3: Structural and non‐structural ankyloglossia and SDB factors analyzed by study.

Data S2: Search Strategy.

Venugopal N., Neposlan J., Bysice A., et al., “Is Ankyloglossia Correlated With Pediatric Sleep Disordered Breathing? A Systematic Review,” The Laryngoscope 136, no. 3 (2026): 1088–1098, 10.1002/lary.70134.