Clinical Consequences of Ankyloglossia from Childhood to Adulthood: Support for and Development of a Three-Dimensional Animated Video

Abstract

Ankyloglossia causes impairment of normal tongue motility and disrupts the average balance of the muscle forces that form the orofacial complex. Inadequate swallowing from birth can cause long-term anatomical and functional consequences in adult life. Using the video presented herein, we describe the current knowledge about the long-term implications of ankyloglossia. After a literature review of the Medline, Google Scholar, and Embase databases on the relations between ankyloglossia and sleep-disordered breathing, we designed and created a three-dimensional (3D) video using Adobe After Effects based on the anatomical and functional changes produced by repeated deglutition, with and without ankyloglossia, from childhood to adulthood. The animated video (Blender 3D, Amsterdam, The Netherlands, 2024) presented herein was based on the most recent literature review of dentition, breathing, posture, and abnormal swallowing, emphasizing the importance of the potential consequences of sleep-disordered breathing. The resulting animated 3D video includes dynamic sequences of a growing child, demonstrating the anatomy and physiology of deglutition with and without ankyloglossia, and its potential consequences for the surrounding structures during growth due to untreated ankyloglossia. This visual instructional video regarding the impacts of ankyloglossia on deglutition/swallowing may help motivate early childhood diagnosis and treatment of ankyloglossia. This instrument addresses the main myofunctional aspects of normal deglutition based on the importance of free tongue motion and can be used by students or professionals training in myofunctional disorders.

1. Introduction

The lingual frenulum is a fibrotic fold of connective tissue that joins the floor of the mouth between the sublingual caruncles and may occasionally insert into the genioglossus muscle in the anterior portion of the tongue, typically between the apex and the middle third. A pathological frenulum can be attached as far anteriorly as the lower alveolar ridge, potentially restricting tongue mobility and leading to functional limitations. These anatomical variations may contribute to altered oral function and its potential consequences [1] (Figure 1). European otolaryngology specialists play a secondary role in both the diagnosis and treatment of ankyloglossia, unlike in other countries [2]. Therefore, it has been suggested that all health professionals be aware of, and work collaboratively to meet, ankylofrenula management practice guidelines [3].

In neonates, ankyloglossia symptoms can present as difficulty and pain during breastfeeding and can lead to early breastfeeding cessation [4]. Prevalence varies among studies, from 2.8 to 12.11%, among whom 62% are males [2,5]. One in four children with ankyloglossia have a family history of a short lingual frenulum [2,5].

Understanding the influences of restriction on tongue movement is based on the concept of the balance of forces in the orofacial musculature, in which the tongue is ultimately responsible for the development of the stomatognathic system [6]. Under normal conditions, there is a balance between the masseter and buccinator muscles, which exert a lateral force. The orbicularis oris muscle exerts a posterior force on the dental arch, which makes it easier for the lips to seal the oral cavity, and the mentalis muscle exerts a passive force. Most importantly, the tongue acts by resting on the incisive papilla as a catapult, always in a posterior direction, to facilitate the propulsion and oral transit of the food bolus during swallowing [6]. These patterns of oral deglutition are reproduced approximately 500–700 times per day, although some of the literature suggests this occurs up to 2000 times, and the pressure on the orofacial complex by the tongue during this process ranges from 750 g to 3 kg [7]. When this balance of forces is broken by the presence of an abnormal lingual frenulum, orofacial myofunctional disorders develop, in which the frenulum plays a determining role [8]. The teeth are continuously subjected to a pressure of 25 KM/cm² by the tongue and orofacial muscles [7,8].

The pediatric consequences of ankyloglossia (

Table 1. Consequences of lingual frenulum and assessment tools.

Clinical Consequences	Reference	Type	Follow-Up Time	Number of Patients	Assessment Tools
Breastfeeding and swallowing	Saccomanno et al. [9]	Pilot study	3 weeks	6 children	Hazelbaker Assessment Tool for Lingual Frenulum Function (HATLFF)
	Pompeia et al. [11]	Systematic review	N/A	N/A	Marchesan Lingual Frenulum Protocol
	Yoon et al. [12]	Prospective cohort case series	3 months	1052	Kotlow Classification Tongue Range of Motion Ratio (TRMR) *
Craniofacial growth	Pompeia et al. [11]	Systematic review	N/A	N/A	Marchesan Lingual Frenulum Protocol
	Yoon et al. [14]	Prospective cohort case series	Occasional	302	TRMR
	Srinivasan et al. [13]	Prospective cohort case series and control	Occasional	57 vs. 60	Nonspecific standardized tool
Dental occlusion	Bhattad et al. [10]	Prospective case series	1 year	3	Kotlow Classification
	Yoon et al. [12]	Prospective case series	3 months	1052	Kotlow Classification TRMR
	Vaz et al. [15]	Prospective case series	Occasional	700	Nonspecific standardized tool
Articulation	Bhattad et al. [10]	Prospective case series	1 year	3	Kotlow Classification
	Suzart et al. [16]	Case series and control	Occasional	52	Marchesan Lingual Frenulum Protocol
Mouth breathing	Bhattad et al. [10]	Prospective case series	1 year	3	Kotlow Classification
	Pompeia et al. [11]	Systematic review	N/A	N/A	Marchesan Lingual Frenulum Protocol
	Yoon et al. [12]	Prospective case series	3 months	1052	TRMR
Social relationships	Bhattad et al. [10]	Case series	1 year	3	Kotlow Classification
Sleep apnea	Guilleminault et al. [17]	Retrospective case series	Occasional	150	Nonspecific standardized tool
	Huang et al. [18]	Retrospective case series	Occasional	27	Nonspecific standardized tool
Head posture	Brożek-Mądry et al. [19]	Case series and control	Occasional	128	Nonspecific standardized tool

* TRMR: tongue range of motion ratio evaluates function without ankyloglossia. N/A not available

) include multiple difficulties [9] that can affect breastfeeding and deglutition [10,11,12], craniofacial growth [11,13,14], dental occlusion [10,14,15], articulation and phonological disorders [11,16], social relationships [10], respiratory disorders [10,11,12], and disorders related to obstructive sleep apnea (OSA) [17,18] and head posture [19]. OSA is a particular concern given that its evaluation is not included in most official anatomical examination guides by the international sleep societies [20]. Although there is consensus on the definition of ankyloglossia, the many potential factors involved in its development are unclear [21].

Sleep Apnea

Sleep surgeons have specific tools, such as drug-induced endoscopy, to evaluate patients with OSA. Using this technique, they have observed different anatomical patterns related to the collapsibility of the pharynx, oropharynx, tongue, and hypopharynx in sedated patients with OSA [22]. Our hypothesis was that disorders of the orofacial musculature complex propagate through linkages between the buccinator and superior pharyngeal constrictor through the hamulus [23] attachment in a progressive sequence.

Others [24] have used videos for educational and health promotion strategies to train students and health professionals, toward improving patient health. The Spanish Otolaryngology Society sponsored a three-dimensional [3D] video to emphasize the importance of early ankyloglossia diagnosis by otolaryngologists. Prior investigations by our group demonstrated a lack of interest in ankyloglossia among sleep specialists [20] and the importance of its diagnosis and treatment in certain patients with OSA [25,26,27].

The objective herein was to review the current knowledge about long-term implications of ankyloglossia and to present an animated 3D video showing dynamic sequences of a growing child with ankyloglossia.

2. Materials and Methods

To support the evidence-based development roadmap of the video, we performed a narrative literature review based on material obtained from the PubMed, Cochrane, Scopus, WEB of Science, Google Scholar, ProQuest, Medline, Google Scholar, and Embase databases, and selected scientific works with information on the upper airway anatomy and pathophysiology of children and adults, with and without ankyloglossia. The literature inclusion was limited to studies of child participants who had clinical and objective diagnosis of ankyloglossia, and who were followed using a standardized, reproducible assessment of its long-term consequences into adulthood; treatment consequences must also have been evaluated by objective criteria.

The exclusion criteria were as follows: syndromic patients with craniofacial malformations; severe obesity; and neurological comorbidities with swallowing or speech impairment.

Keywords used for the literature search were as follows: ankyloglossia, dentition, breathing, posture, craniofacial growth, sleep apnea, childhood, adulthood, and anatomy. The literature review was conducted independently by two otolaryngologists, one dentist, and one speech therapist, all with clinical experience in ankyloglossia and sleep-disordered breathing. Each reviewer independently screened titles, abstracts, and full-text articles against the inclusion and exclusion criteria. Disagreements were resolved by consensus. The final selection of studies was discussed within a multidisciplinary panel that included two imaging engineers to ensure clinical relevance and applicability for the development of the 3D animation.

Herein, we describe the narrative outline (Blender 3D, Netherlands, 2024) based on a review of the most recent literature about dentition, breathing, posture, and abnormal swallowing, which emphasizes the importance of potential consequences of sleep-disordered breathing (SDB).

3. Results

After identifying 108 records, 24 duplicates were removed before screening. A total of 84 records remained, but only 35 were screened after excluding those not relevant based on the title and abstract. Of these, three reports could not be retrieved. The remaining 24 full-text articles were assessed for eligibility, and 13 were excluded for not fulfilling the inclusion and exclusion criteria. Finally, 11 studies were included in this narrative review (Figure 2). The creation of the 3D visualization was guided by a narrative outline, which was developed through the review of these 11 sources (Table 1), which helped shape the representation of ankyloglossia into two main parts. The first part focused on describing the anatomy of a normal neonatal face, skull, mouth, throat, and esophagus, and its development to adulthood. The second part covered the physiological processes involved in pathological consequences of development with ankyloglossia.

After finishing the narrative outline based on these academic references, a designer used the outline to create 3D visuals for the anatomy video, and the mechanisms of growth and changing stomatognathic functions. This process involved collaboration between the healthcare professionals and digital designers, allowing them to discuss the narrative outline, review image details, and ensure the visuals accurately reflected neonatal and adult anatomy.

No articles included in this review confirmed the hypothesis about the development of disorders in the orofacial musculature complex that are sequentially propagated through linkages between the buccinator and superior pharyngeal constrictor through the hamulus attachment. All identified articles described an isolated vision of ankyloglossia consequences, depending on the investigators’ specializations [6,7,8,9,10]. For example, there was no functional evaluation of ankyloglossia in a paper written by otolaryngologists [21] and no otolaryngologist evaluations in papers written by dentists [10,12]. Long-term, ankyloglossia has implications for the development of the stomatognathic system, which should be treated by multidisciplinary teams. To our knowledge, to date, there are no articles in which a consensus was developed between different specialists treating ankyloglossia.

4. Discussion

Ankyloglossia is increasingly recognized not only as a structural anomaly, but as a functional impairment with potential long-term orofacial development, airway patience, and quality of life consequences. This condition impacts multiple physiological domains, from infancy through adulthood, reinforcing the need for a multidisciplinary diagnostic and therapeutic approach.

The tongue plays a critical role in oral posture, swallowing, and maxillofacial growth [17]. When its mobility is restricted, as in ankyloglossia, it can alter the balance of orofacial muscles and contribute to craniofacial developmental changes. Studies such as those by Yoon et al. [12,14] and Srinivasan et al. [13] demonstrate correlations between ankyloglossia and maxillary hypoplasia, narrow palates, and altered dental occlusion. These findings suggest that functional limitation due to a short lingual frenulum may impair proper skeletal growth, particularly during critical developmental periods (Figure 3).

In addition to structural impacts, ankyloglossia is associated with functional disturbances such as atypical swallowing and mouth breathing. These behaviors not only affect feeding and speech, but they also influence airway dynamics. Guilleminault et al. [17] and Huang et al. [18] found a strong relation between short lingual frenula and pediatric OSA. Moreover, a large pediatric study by Villa et al. supports the link between short lingual frenula and SDB in school-age children [28]. This reinforces the idea that oral dysfunction may extend beyond feeding and articulation problems to more serious respiratory issues, particularly in the presence of other anatomical risk factors.

Breastfeeding difficulties are often among the earliest signs of ankyloglossia [29]. Saccomanno et al. [9] and Pompeia et al. [11] found that frenotomy can improve latch and milk transfer, reduce maternal nipple pain, and potentially support orofacial muscle development in infants. These early functional improvements may prevent downstream structural consequences and underline the importance of timely diagnosis and treatment.

Speech production is also affected by tongue mobility, although causality must be considered carefully. While studies like those by Suzart et al. [16] link ankyloglossia to articulation disorders, it is essential to differentiate between speech delays due to developmental versus mechanical causes. Restricted lingual range of motion may influence articulation of specific phonemes, but speech pathologists must evaluate this within the broader context of each child’s development.

The integration of findings across these domains—breastfeeding, craniofacial development, airway patency, and speech—supports the value of a collaborative approach in both research and clinical care. In line with this, pediatric dental associations, such as the American Association of Pediatric Dentistry (AAPD), have issued clinical policies for frenulum management, underscoring the importance of early intervention [30]. Similarly, the Australian Dental Association has provided policy statements highlighting the impact of ankyloglossia and the role of dental professionals in its management [31]. However, as our review revealed, the literature remains fragmented, with most studies reflecting the narrow perspective of a single specialty.

A cephalometric study of children with oral breathing observed alterations in head posture [32]. Studies have also assessed head posture in children with different forms of malocclusion, facial types, and oral malformations [19,33,34] (Figure 4). These abnormalities can also affect respiratory tract anatomy, which can contribute to OSA. Knowledge about these conditions may be useful for the objective diagnosis of atypical swallowing and ankyloglossia, and for its treatment. Rehabilitation of this dysfunction should not be limited to orofacial structures but should also include cervical structures (Figure 4).

Based on our review, the evidence suggests that ankyloglossia may represent a significant health concern that is currently approached inconsistently, potentially allowing a functional issue in childhood to evolve into a more serious condition such as OSA. These findings highlight the lack of a multidisciplinary approach to this issue, which has contributed to its growing prevalence, and is further compounded by a lack of focus on preventive measures for OSA [20,28,35].

Otolaryngologists are uniquely positioned to address this issue, due to their comprehensive understanding of the anatomy and function of head and neck structures. Our discipline has pioneered demonstrating that, in certain cases, frenuloplasty can remodel structures such as the base of the tongue and epiglottis, which are involved in OSA, as evidenced by drug-induced sleep endoscopy findings [26,27]. Unfortunately, ankyloglossia is not currently recognized as a cause of OSA in children or adults [20,21]. Thus, our group has initiated the first randomized controlled trial to demonstrate that treating ankyloglossia in patients with specific anatomical conditions can improve or potentially cure OSA [36].

Our video was produced based on our cumulative clinical experience, including attending to many patients with SDB and ankyloglossia, despite the limited awareness in some specialties about the relevance of this condition. The project herein aimed to address this gap through the creation of an educational 3D video grounded in anatomical, physiological, and clinical evidence. By visualizing the consequences of ankyloglossia across the lifespan, we hope to foster interdisciplinary dialogue and improve awareness among health professionals. The animation is not a substitute for evidence-based guidelines but was developed to serve as a pedagogical tool to highlight the potential downstream effects of untreated ankyloglossia.

This project was not without limitations. These include the narrative review methodology, which revealed the lack of any prior comprehensive visual studies, and limited evidence quality. The overall low quality of evidence in the included papers necessitates a cautious interpretation of the results. This is largely due to the absence of a multidisciplinary perspective, small sample sizes, a lack of randomized clinical trials, and limited follow-up periods in the studies reviewed.

5. Conclusions

This paper provides an overview of the clinical consequences of ankyloglossia across childhood and adulthood and describes a visual animation of ankyloglossia from infancy to adulthood. Such videos may promote awareness of this condition and promote further multidisciplinary investigation into its clinical implications.