J Korean Acad Pediatr Dent > Volume 52(4); 2025 > Article
Lee: Tongue-Tie in Children: Clinical Controversies and a Comprehensive Review

Abstract

This review examines the ongoing controversy surrounding the treatment of tongue-tie, particularly the lack of evidence supporting the long-term efficacy of diagnosis and surgical intervention (tongue-tie release). It identifies the need for further research to support evidence-based practice and proposes a multidisciplinary, function-centered treatment approach. Furthermore, it explores various theories and causes of tongue movement restriction in children, including neurological, muscular, structural, acquired, and functional factors that must be differentiated from tongue-tie.

Introduction

1. Overview and Historical Context of Ankyloglossia

Ankyloglossia is a congenital condition characterized by a restrictive lingual frenulum that limits the normal range of tongue movement[1]. This anatomical restriction can significantly impair essential oral functions such as swallowing and speaking[2]. The medical concept of ankyloglossia has been recognized since the 1960s. As initially described by Wallace, it was defined as a condition in which the tongue’s frenulum linguae is too short, preventing the tip of the tongue from extending beyond the lower incisors[3].
Despite long-standing debate, the clinical management of ankyloglossia remains highly controversial[1]. There is still no consensus on its precise definition, classification system, diagnostic criteria, or optimal treatment strategy[1].
A notable trend observed over the past two decades is the dramatic increase in infant frenulum diagnoses and subsequent treatments in countries such as the United States, Canada, and Australia, despite declining birth rates[4,5]. This surge is partly attributable to the diverse and inconsistent definitions of the frenulum used by various healthcare providers and researchers[1,6]. The lack of a universal and precise definition of tongue-tie leads to inconsistent diagnostic practices[1]. This inconsistency contributes to inflated reported prevalence rates and the perceived rise in patient numbers. Consequently, surgical interventions have increased, some of which may not be medically justified[1,6,7].
Establishing a universally accepted, function-based definition of tongue-tie is essential. Without such a definition, efforts to standardize diagnosis, accurately assess prevalence, conduct meaningful research, and develop evidence-based guidelines will be significantly hindered. This could result in the overmedicalization of a condition that often resolves naturally or can be managed with non-surgical treatment[1,6].

2. Prevalence and Causes in the Pediatric Population

The reported prevalence of lingual frenulum adhesions varies widely, ranging from 0.1% to 10.7%. This broad range primarily results from the lack of a universally accepted definition and the differing diagnostic criteria used in various studies[1,8].
Interestingly, mild cases of lingual frenulum adhesions often resolve naturally over time. This natural progression helps explain why studies focusing solely on newborns (1.72 - 10.7%) report significantly higher rates than studies examining older children, adolescents, and adults (0.1 - 2.08%)[8,9]. A consistent finding across multiple studies is that ankyloglossia is more common in males, with estimates suggesting it occurs approximately three times more frequently in boys than in girls[10,11].
This condition occurs when the frenulum, located beneath the tongue, fails to fully separate from the tongue before birth. This developmental abnormality may be influenced by genetic factors and often exhibits a familial pattern. As a congenital anomaly, a shortened lingual frenulum may also be associated with other syndromes[12].

Definition and Diagnosis of Tongue-tie

1. Anterior vs. Posterior Tongue-tie

The greatest challenge in managing tongue-tie is the lack of universally agreed definitions, classification systems, and standardized diagnostic criteria, which leads to significant variation in treatment approaches[1]. To address this issue, a panel of otolaryngologists published a clinical consensus statement in 2020, defining tongue-tie as “a condition in which restricted tongue mobility is caused by a limited lingual frenulum[1].” The American Academy of Pediatrics (AAP) also recommends that healthcare providers focus on this general definition, emphasizing limited tongue mobility[6].
Ankyloglossia can be classified into two main types.
• Anterior ankyloglossia: This is the classic and more easily identifiable form, characterized by the frenulum attaching to or very near the tip of the tongue, significantly restricting tongue movement[1].
• Posterior ankyloglossia: This term remains highly controversial within the medical community. It generally refers to a frenulum attachment located further back on the ventral surface of the tongue or to submucosal adhesions that are believed to restrict tongue mobility. However, many experts consider what is termed a “posterior frenulum” to be a normal frenulum attachment. Ankyloglossia is an ill-defined term lacking expert consensus and should not be used as a justification for performing surgical procedures on infants[6].
• Recent anatomical studies describe the frenulum not as a simple fibrous band but as a complex fascial fold containing motor and sensory nerve branches. This advanced understanding suggests that there may be no anatomical basis for posterior tongue-tie and that frenotomy, even if seemingly minor, could potentially worsen the condition due to possible nerve involvement[1].

2. Clinical Evaluation and Diagnostic Tools

Diagnosis of tongue-tie should primarily focus on demonstrating restricted tongue mobility, which is typically identified when the tongue cannot extend beyond the vermilion border or the lower gum or incisors. A characteristic indented or heart-shaped appearance is often observed when the tongue tip is protruded[1].
A comprehensive medical history and physical examination are essential for an accurate diagnosis. This should include a detailed breastfeeding history, feeding methods, and an assessment of actual milk production[8]. Some healthcare professionals may have limited knowledge of breastfeeding techniques. This underscores the importance of early consultation with a lactation consultant[8]. A collaborative, multidisciplinary approach involving lactation consultants, pediatricians, family physicians, nurses, surgeons, and dentists is crucial for accurate diagnosis and effective management[6,13].
Several assessment tools are used to evaluate tongue-tie; however, their widespread acceptance and validation vary (Table 1)[2,14,15].
There is a gap between the clinical imperative to diagnose tongue-tie based on demonstrable functional impairment and the reality that many existing classification and diagnostic tools, as well as much of the literature, still rely heavily on anatomical appearance[2,14,16]. This can lead to diagnosis and intervention based on perceived anatomical “adhesion” despite the absence of clear functional limitations, resulting in overtreatment[16]. The controversy surrounding posterior tongue-tie exemplifies this issue. The lack of functional criteria fuels debate and may lead to unnecessary procedures[1,2]. This highlights a fundamental flaw in the current diagnostic paradigm, which is an urgent problem. Function-centered diagnostic criteria and assessment tools must be developed and rigorously validated[14,16]. Such changes are essential to ensure that treatment targets only children experiencing actual functional limitations, thereby reducing unnecessary interventions, associated costs, and the risk of potential complications[2,16,17].

3. Differential Diagnosis of Infant Feeding Disorders

When a newborn experiences difficulties with breastfeeding, it is essential to systematically rule out other potential pathologies before assuming the issue is due to tongue-tie[18]. Many symptoms attributed to tongue-tie overlap significantly with those caused by other common breastfeeding problems[8,18].
Key pathological features to consider include the following:
• Craniofacial pathologies, such as a receding jaw (underbite) and cleft palate
• Nasal obstruction: Conditions such as stenosis of the nasal aperture and nasal atresia
• Airway obstruction, such as bilateral vocal cord paralysis or laryngeal flaccidity
• Laryngopharyngeal reflux: a common condition that can mimic or worsen feeding difficulties
• Neurological or cardiovascular disorders that may impair an infant’s feeding ability
It is crucial to gather comprehensive information on various feeding factors. For example, improper feeding posture is often the primary cause of breastfeeding difficulties, rather than issues with the lingual frenulum itself[8,18].

Clinical Symptoms and Their Effects

1. Primary Impact: Breastfeeding Challenges from the Perspectives of Infants and Mothers

Frenulum adhesion is most commonly associated with breastfeeding and primarily interferes with the process. For infants, breastfeeding complications include difficulty latching, inability to maintain the latch, fussiness during feeding, low milk extraction efficiency, and, consequently, poor weight gain or growth failure[13,18,19]. Infants with tongue-tie struggle to extend their tongue beyond the lower gum line for effective latching and often compensate by pressing the nipple with their jaw[18,20].
Mothers frequently report severe pain during breastfeeding, insufficient milk let-down, or milk remaining in the breast because the baby cannot suck effectively. Persistent nipple pain affects up to 80% of these women, and approximately 25% consider switching to bottle feeding due to extreme pain or difficulty, even after six weeks postpartum[20,21].
Tongue-tie release can offer short-term benefits, such as reducing nipple pain for the mother. Improvements in breastfeeding efficiency may also be observed. However, there is currently no definitive evidence that it extends the overall duration of breastfeeding. Methodological limitations in existing studies, including small sample sizes and lack of blinding, make it difficult to draw definitive conclusions about long-term breastfeeding success. For example, the Cochrane Collaboration’s 2017 review concluded that frenotomy reduced maternal nipple pain in the short term but did not consistently improve infant breastfeeding outcomes[13,18].

2. After Breastfeeding

Symptoms linked to the lingual frenulum are part of a connected physiological system, not isolated local problems. Tongue movement limitations may lead to compensatory changes and secondary effects in respiratory, digestive, musculoskeletal, language, and emotional development. Research suggests the oral cavity’s function is deeply integrated with systemic health[22,23]. Therefore, a holistic, multidisciplinary approach for diagnosis and management considering overall quality of life is essential. Early, effective intervention might help prevent additional complications, but further study is needed to confirm this[21,22].

1) Language and Articulation Development

Tongue-tie has been identified as a potential cause of language disorders, particularly those affecting articulation. However, a direct relationship between tongue-tie and language disorders remains highly controversial[23,24]. Some experts argue that tonguetie rarely causes severe language disorders, and it is commonly believed that speech onset is generally not delayed[23,24]. Nevertheless, when tongue movement is significantly restricted, children may develop maladaptive compensatory movements during speech, which can result in articulation disorders[23,24].

2) Oral Hygiene and Dental Health Outcomes

Tongue-tie may be associated with worsened oral hygiene and various dental problems, including malocclusion, gum recession, increased cavities, and the formation of gaps between the lower front teeth[22,23]. This is partly because the tongue’s ability to naturally remove food debris from the teeth and mouth is impaired. Additionally, this condition may affect craniofacial development, potentially leading to maxillary hypoplasia, palatal elongation, and broader craniofacial deformities. The hypothesis linking ankyloglossia to mandibular prognathism and maxillary hypoplasia suggests that the tongue’s low position causes abnormal pressure on the dental arches[25]. However, direct evidence supporting the influence of tongue-tie on malocclusion remains limited at present[22,23].

3) Sleep-Disordered Breathing and Obstructive Sleep Apnea (OSAS)

Ankyloglossia is associated with OSAS and other sleep-disordered breathing symptoms in children. Alterations in craniofacial growth potentially caused by ankyloglossia can reduce the size of the upper airway, increasing the risk of sleep-related breathing disorders[26,27]. Furthermore, if the tongue cannot properly contact the palate, it may cause airway obstruction and reduce sleep quality. One randomized controlled trial suggested that frenectomy may help alleviate the severity of apnea. Frenectomy has been reported as an effective intervention to improve sleep in children with restricted tongue movement[26,27].

4) Gastrointestinal Symptoms: Aerophagia and Reflux

A literature review identified reflux, vomiting, and regurgitation as common gastrointestinal (GI) symptoms observed in infants with tongue-tie. Aerophagia, characterized by increased air swallowing, is considered a key mechanism linking tongue-tie to these GI symptoms[26,28]. Frenulum ankylosis is believed to cause excessive air swallowing, leading to reflux, pain, and abdominal distension due to difficulties in latching during feeding and impaired tongue movement control. A significant finding is that improved tongue mobility following frenulum release surgery results in a noticeable improvement in gastroesophageal reflux symptoms. Multiple studies have demonstrated that infants who underwent surgical resection via frenectomy experienced a statistically significant reduction in reflux scores[26,28,29].

5) Psychosocial and Quality of Life Impact

Tongue-tie affects more than just physical feeding or speech. It can significantly reduce a child’s overall quality of life. While mild cases sometimes resolve naturally, persistent symptoms, including challenges with more complex eating and speaking tasks, can appear as children grow older[21,26]. Difficulties include chewing, choking, stuffing food, and social issues like teasing or trouble with licking foods, playing instruments, and kissing. Long-term impacts may include social withdrawal, emotional distress, and reduced self-esteem, with these effects sometimes only becoming clear later in childhood[21,26].

Treatment Approaches

1. Non-Surgical Interventions: Feeding Support and Speech Therapy

Consulting a lactation consultant is strongly recommended before considering surgical intervention, especially when breastfeeding difficulties are present. It is often observed that the primary cause of breastfeeding issues is an improper feeding position rather than the frenulum itself[8,30]. Because this is a complex issue, a thorough breastfeeding assessment by a comprehensive, multidisciplinary team is necessary. Ideally, this team should include breastfeeding specialists, lactation consultants, and pediatricians to provide a holistic evaluation and management plan[30].
For children experiencing language difficulties, consultation with a speech-language pathologist is essential. This is especially important for issues related to bottle feeding or articulation exercises. The speech therapist may also refer the patient to a multidisciplinary team if the problem appears more complex. Some studies suggest that language problems associated with ankyloglossia can be effectively addressed and resolved through professional speech therapy alone, without the need for frenotomy[23,24,31].

2. Surgical Intervention: Frenectomy (Conventional Technique vs. Laser Technique)

Frenectomy is the most common surgical procedure used to correct tongue-tie. This procedure can be performed using traditional methods, typically involving scissors to release the lingual frenulum. Alternatively, laser frenectomy is also an option and is often described as safe, simple, and comfortable. Although studies comparing various frenotomy techniques exist, current research does not indicate that any one method is definitively superior to the others[32].
Surgical intervention should be considered a reasonable option for symptomatic tongue-tie, but only after thoroughly evaluating and, if possible, addressing other potential causes of breastfeeding difficulties. It is important that the frenotomy be performed by a trained specialist with extensive experience in the medical care of newborns and infants. Furthermore, the process should include a comprehensive discussion of all possible alternatives, the potential risks and benefits of the proposed procedure, and various pain management options[32].

3. Research on the Effects of Frenectomy

1) Impact on Breastfeeding Outcomes

Research indicates that frenotomy may help reduce nipple pain in mothers. Some studies suggest that breastfeeding outcomes may significantly improve for both mother and infant compared to receiving only intensive support from a lactation consultant. However, despite these short-term improvements, there is insufficient strong evidence to support the claim that frenotomy leads to positive long-term outcomes beyond immediate relief during breastfeeding. Furthermore, there is no sufficient evidence that frenotomy helps prolong breastfeeding duration. The lack of definitive long-term data, combined with methodological shortcomings in many studies such as small sample sizes and the absence of double-blinding, limits the generalizability and reliability of these conclusions. A 2017 Cochrane Review noted short-term pain reduction but found no consistent positive effect on infant breastfeeding[18,33,34].

2) Effects on Language, Dental, Sleep, and Gastrointestinal Issues

Some studies suggest that frenectomy may be an effective intervention for improving language in children with restricted tongue movement. However, the overall evidence supporting a direct causal relationship between tongue-tie and speech disorders, and consequently the efficacy of frenectomy for speech improvement, remains inconclusive or insufficient. Additionally, evidence directly linking tongue-tie to malocclusion and demonstrating the long-term effects of frenectomy on dental outcomes is currently limited[23,24].
In some cases, frenectomy has been shown to effectively alleviate the severity of apnea. It has also been reported to improve sleep in children with restricted tongue movement[23,26].
One area of recent research focus is the impact of tongue-tie release on gastrointestinal symptoms. This procedure has been associated with significant improvements in gastrointestinal issues in infants, particularly reflux symptoms. Several studies have demonstrated a statistically significant reduction in reflux scores following tongue-tie release[28,33].

4. Risks and Complications of Frenectomy

Frenectomy is generally considered a safe procedure. Minor bleeding is the most common complication. However, more serious complications have been reported in several studies[32,35].
• Delayed Differential Diagnosis: Frenectomy may postpone the identification of more serious underlying causes of malnutrition, such as congenital heart disease or metabolic disorders[35].
• Bleeding: Cases of severe bleeding and hemorrhagic shock have been reported.
• Infectious diseases: Submandibular abscess, Ludwig’s angina, and others.
• Respiratory disorders: Apnea and acute life-threatening events.
• Refusal to eat: Potential for prolonged refusal of oral intake after the procedure.
• Discomfort: Potential refusal to breastfeed due to pain[8].

5. Importance of a Multidisciplinary Approach to Treatment

The challenges associated with effective breastfeeding and the broad, multifaceted impact of tongue-tie highlight the inherent complexity of this condition. A multidisciplinary approach is essential for achieving successful outcomes. This collaborative model ideally includes lactation consultants, pediatricians, family physicians, nurses, surgeons, lactation therapists, and dentists working together to provide comprehensive care and support shared decision-making with families[30].

Current Research and Controversies

1. Increase in Publications vs. Robust Evidence

Although research and publications on tongue-tie and frenotomy have risen globally, robust scientific evidence has not kept pace with clinical activity. Most studies lack quality design, including randomized controlled trials and reliable outcome measures, leading to weak evidence-based guidelines. Methodological flaws like small sample sizes and inconsistent blinding are common, and objective data on outcomes such as breastfeeding duration and infant growth are scarce. Clinical practice and treatment rates are growing faster than scientific validation, raising concerns about overtreatment and resource misuse, with potential risks to patients if long-term efficacy remains uncertain[34,36].

2. Ongoing Controversies

Key controversies in the study and practice of adhesions revolve around several core areas:
• Definition and Diagnostic Criteria: The absence of standardized definitions and universally accepted diagnostic criteria continues to hinder consistent diagnosis and research[36].
• Posterior Tongue Adhesions: The existence and clinical significance of posterior tongue adhesions remain highly contentious. Many experts consider them normal anatomical variations that do not require intervention[1,37].
• Optimal Timing and Intervention Methods: There is no clear consensus on the ideal age for surgery, nor is there agreement on whether conventional frenectomy or laser surgery yields superior outcomes[32].
• Treatment vs. Spontaneous Resolution: The degree to which observed improvements can be attributed to frenectomy, as opposed to natural resolution in mild cases or the advantages of non-surgical feeding support, remains unclear[13,18,36].
• Long-Term Outcomes: The long-term efficacy of frenectomy beyond the immediate relief of breastfeeding discomfort remains insufficiently explored[34,36].

Definition of Tongue Movement Restriction and the Importance of Differential Diagnosis

Tongue movement restriction refers to an impaired ability to perform essential oral functions due to limitations in the tongue’s normal range of motion, strength, or coordination[6,15]. Symptoms such as feeding difficulties, speech disorders, and swallowing disorders are common in various pediatric conditions beyond ankyloglossia, making a thorough differential diagnosis critically important[6]. Considering alternative etiologies is essential to avoid unnecessary or ineffective procedures and ensure the underlying problem is properly addressed[6,38].

1. Neurological Causes of Restricted Tongue Movement

Neurological disorders can significantly impair tongue movement by affecting the nerves that control the tongue muscles, the brain regions responsible for planning and executing speech and swallowing, or the overall motor control system[39,40]. Restrictions in tongue movement often serve as key indicators of various neurological disorders that impact not only oral function but also systemic health, including respiratory function and overall developmental processes[41].

1) Hypoglossal Nerve Palsy (HNP)

Hypoglossal Nerve Palsy (HNP) involves damage to the twelfth cranial nerve, which primarily controls the movement of all intrinsic tongue muscles and most extrinsic tongue muscles. Damage to this nerve can result in tongue weakness, atrophy, and deviation[42]. In children, tumors are the most common cause of HNP, followed by trauma, vascular anomalies, and autoimmune diseases. Rarely, infections such as influenza B virus, enterovirus, adenovirus, Epstein-Barr virus, or Streptococcus species may be involved. Although uncommon, HNP can occur in isolation without other neurological symptoms[42,43].
Symptoms of HNP include unilateral tongue atrophy and muscle contraction, asymmetry in tongue protrusion (typically toward the affected side), dysarthria (difficulty speaking), and dysphagia (difficulty swallowing). In cases of bilateral paralysis, saliva may accumulate within the oral cavity[43]. Diagnosis typically involves a clinical examination assessing tongue movement, atrophy, and muscle contraction, supplemented by a brain MRI/MRA to rule out structural abnormalities, and occasionally a lumbar puncture. Electromyography can be particularly useful in distinguishing between upper motor neuron and lower motor neuron lesions. Treatment primarily focuses on addressing the underlying cause, such as tumor management or infection treatment[42,43].

2) Oral Motor Disorders Related to Cerebral Palsy (CP)

Cerebral Palsy (CP) is a group of disorders caused by abnormal brain development or injury, affecting movement, balance, and posture. In children with CP, this condition can cause dyskinesia (involuntary muscle movements) or hypotonia (reduced muscle tone) in oral muscles, including the tongue, disrupting coordinated muscle function. These neurological impairments often lead to a range of functional problems. For example, oral motor dysfunction can directly contribute to breathing difficulties, obstructive sleep apnea, and recurrent respiratory infections[41].
Restricted tongue movement in neurological disorders can lead to feeding and swallowing difficulties, increasing the risk of aspiration and respiratory problems. In children with cerebral palsy, common symptoms include abnormal tongue and lip movement, hypersensitivity, refusal of food, and difficulty with different food textures, resulting in long feeding times. Diagnosis is based on observation of feeding patterns and oral motor function, with early signs such as poor sucking at birth. Management is multidisciplinary, focusing on strengthening oral muscles, improving chewing, and coordinating sucking, swallowing, and breathing[41,44].

3) Childhood Apraxia of Speech (CAS) and Dysarthria

Childhood Apraxia of Speech (CAS) and dysarthria are distinct speech disorders that affect oral motor control. In CAS, the brain has difficulty planning the precise movements required for speech, resulting in incoordination of the lips, jaw, and tongue. In contrast, dysarthria is characterized by impaired articulation caused by weak or uncoordinated speech muscles[40].
Similar symptoms in neurological conditions such as CP, stroke, and CAS pose significant diagnostic challenges and require expert evaluation beyond surface-level observation[39].
Accurate diagnosis involves detailed history-taking, comprehensive neurological examination, and instrumental assessments to distinguish between causes. CAS is marked by limited and unclear speech, sound errors, and articulation difficulties, while dysphonia involves voice changes and slow or weak speech due to muscle weakness. Diagnosis is made by speech-language pathologists, and treatment focuses on intensive speech therapy and oral motor exercises[39,40].

4) Effects of Stroke and Traumatic Brain Injury (TBI)

Stroke and Traumatic Brain Injury (TBI) can damage brain regions that control numerous muscles involved in swallowing and speaking, severely limiting tongue movement. This damage often results in dysphagia (difficulty swallowing) and dysarthria (speech impairment). Swallowing difficulties are common after a stroke, affecting the preparation of food in the mouth, the propulsion of food into the pharynx, and overall swallowing efficiency. Tongue weakness, a direct consequence of nerve damage, can cause problems with controlling the food bolus, premature food leakage, or impaired swallowing timing, potentially leading to safety concerns such as food penetration and aspiration. Speech disorders, particularly dysarthria, are also frequently observed[41,44].
Diagnosing acquired neurological disorders requires detailed clinical evaluation, thorough feeding history, and instrumental tests. Video Fluoroscopic Swallowing Study (VFSS) (or Modified Barium Swallow Study (MBSS)) uses X-rays to assess swallowing in real-time and identify structural or physiological issues, mucus problems, or aspiration. FEES offers a non-radiation way to directly observe swallowing via nasal endoscopy. Treatment may include speech therapy for tongue strength, dietary changes, and eating habit adjustments[41].

2. Muscular and Genetic Disorders Affecting Tongue Mobility

Restricted tongue movement may result from muscle weakness (hypotonia), stiffness (myotonia), or abnormal size (macroglossia), not solely from physical causes like lingual ties[45].

1) Congenital Myopathies (CM) and Myotonia Congenita

Congenital myopathies (CM) are rare muscle disorders present at birth that often result in weak or small muscles, sometimes caused by CLCN1 gene mutations leading to myotonia. The main symptoms include a “floppy baby” appearance, delayed motor milestones, and difficulties with breathing and feeding, particularly with sucking, chewing, and extended feeding times. Myotonia causes muscle stiffness, which typically improves with movement, known as the “warm-up effect”[45].
Diagnosis is based on clinical assessment, measurement of creatine kinase levels, and genetic testing and may include imaging studies or muscle biopsy for confirmation. Treatment commonly consists of physical therapy, sensory stimulation, and interventions aimed at managing feeding and respiratory challenges.

2) Other Genetic Syndromes Associated with Oral Motor Symptoms

Several genetic syndromes can affect muscle tone, craniofacial development, or overall neurological function, presenting with oral motor symptoms such as restricted tongue movement. Early intervention is crucial. However, the long-term prognosis varies widely. The immediate impact on feeding is critical for growth and often results in issues such as failure to thrive[46,47].
• Down Syndrome: Infants with Down syndrome often exhibit hypotonia. This can cause the tongue to position too far back in the mouth, leading to glossoptosis (tongue-falling-back syndrome), which obstructs the airway and impairs breathing, eating, and swallowing[ 48].
• Beckwith-Wiedemann Syndrome (BWS): Macroglossia is a common feature of BWS, affecting approximately 90% of children with this condition. The enlarged tongue can interfere with proper tooth alignment, eating, and speech development[49].
• Ehlers-Danlos Syndrome: This syndrome is characterized by connective tissue disorders and may present with an absent lower labial frenulum and lingual frenulum. While it does not directly cause movement restrictions, it reflects a broader connective tissue abnormality that can indirectly affect oral structure and function[50].
• Pierre Robin Sequence: This congenital condition involves an abnormally small mandible, causing the infant’s tongue to be positioned too far back. This anatomical configuration directly affects breathing, eating, and swallowing[51].
Diagnosis is based on clinical presentation, genetic testing for specific syndromes, and imaging studies to identify craniofacial anomalies. Treatment is typically multidisciplinary, involving speech therapy, nutritional support (including tube feeding in severe cases), and sometimes surgical interventions such as tongue reduction for macroglossia or jaw growth management for Pierre Robin sequence.

3. Structural and Anatomical Abnormalities (Excluding Those Related to the Frenulum)

This category includes primary structural abnormalities such as a small jaw, tongue hypertrophy, or skeletal growth anomalies, which may lead to a range of secondary functional problems affecting breathing, feeding, speech, dental health, and even psychosocial development. Addressing the underlying structural issue is often essential to resolving these functional limitations, rather than treating the symptoms.

1) Glossoptosis

Glossoptosis refers to the tongue being positioned too far back in the mouth toward the throat or being improperly positioned. It is commonly associated with conditions such as Pierre Robin syndrome, Down syndrome, and cerebral palsy.
Key symptoms of glossoptosis include breathing difficulties caused by airway obstruction, snoring, and obstructive sleep apnea. Additionally, it can significantly impair eating and swallowing, potentially leading to failure to thrive in infants, and may also affect speech and language development. Early diagnosis and intervention are vital[51].

2) Macroglossia

Macroglossia is characterized by an abnormally enlarged tongue relative to the size of the oral cavity, which directly interferes with normal oral function. Although it is most commonly associated with genetic syndromes such as Beckwith-Wiedemann syndrome and Down syndrome, macroglossia can also result from cancer or severe infections.
Diagnosis is primarily based on clinical observation and identification of the underlying cause. If symptoms are mild, immediate surgical intervention may not be necessary. However, in more severe cases, surgical reduction of the tongue size may be performed. Comprehensive treatment involves managing the underlying syndrome[46]

3) Oral Tumors and Cysts

Tumors or lesions in the jawbone or soft tissues of the mouth and face can physically obstruct tongue movement, displace surrounding structures, or cause pain, potentially limiting oral function. These may include various types of benign tumors such as ameloblastoma, central giant cell granuloma, odontogenic myxoma, odontoma, fibroma, and warts. Cysts, including odontocysts, odontokeratocysts, and mucous cysts, may also develop. While most of these tumors are benign, some may exhibit aggressive behavior. Some tumors are asymptomatic and may be discovered incidentally during routine dental X-rays. Diagnosis typically involves a clinical examination, imaging studies such as X-rays, and a biopsy for definitive identification. Treatment often requires surgical removal[52].

4) Craniofacial Anomalies (e.g., Cleft Palate, Retrognathia)

Structural congenital defects affecting the head and face can significantly alter the anatomy of the oral cavity, limiting tongue movement or the ability to form an adequate seal during eating or speaking. Examples include cleft palate and retrognathia.
Diagnosis is typically made at birth or during early infancy through a physical examination. Management involves a multidisciplinary team approach, including surgeons, speech therapists, and other specialists, with surgical correction often serving as a key component of treatment[53].

4. Acquired and Iatrogenic Causes

Tongue movement restrictions can result not only from congenital or developmental issues but also from external factors, including iatrogenic causes or injuries. Medical interventions can unintentionally lead to tongue movement limitations, emphasizing the importance for healthcare professionals to be aware of potential iatrogenic complications.

1) Postoperative Complications (e.g., Head and Neck Surgery, Intubation)

Surgical procedures in the head and neck region, particularly those involving the oral cavity, pharynx, or adjacent structures, carry the risk of inadvertently damaging nerves or muscles, such as the hypoglossal nerve, or causing scar tissue formation that restricts tongue movement. Additionally, prolonged or forceful intubation can lead to hypoglossal nerve paralysis due to mechanical compression or nerve-stretching injuries.
Symptoms include weakness, numbness, tongue deviation, dysarthria, and dysphagia. These complications may be temporary, with nerve damage typically resolving within three months. However, recovery can be slow, as observed in axonal neuropathy. Diagnosis is based on a history of recent surgery or endotracheal intubation, a thorough clinical examination, and occasionally electromyography. Treatment varies depending on the severity and specific type of nerve damage, ranging from careful observation to speech therapy or other supportive measures[54].

2) Radiation Therapy

Radiation therapy to the head and neck can cause stiffness in the oral muscles and mucosa , leading to stiffness as well as reduced tongue mobility. Additionally, radiation can damage the salivary glands, resulting in xerostomia which further impairs oral function and causes discomfort.
Symptoms include stiffness in the mouth and jaw, altered taste perception, dry mouth, pain, and difficulty swallowing. These complications may arise acutely during treatment or persist as chronic issues[55].
Diagnosis is based on the patient’s history of radiation therapy, a clinical examination of oral structures, and an assessment of swallowing function. Management focuses on pain control, meticulous oral hygiene, addressing symptoms of xerostomia, and swallowing therapy[55].

3) Traumatic Injury

Direct trauma to the tongue, jaw, or surrounding structures can restrict tongue movement due to muscle damage, nerve injury (e.g., to the hypoglossal nerve), swelling, or scarring. Symptoms typically include pain, swelling, visible injury, difficulty moving the tongue, dysarthria, and dysphagia. Diagnosis involves a clinical examination, imaging studies, and a neurological evaluation. Treatment varies depending on the nature and severity of the injury, ranging from wound care and surgical intervention to subsequent rehabilitation, such as speech therapy[56,57].

Functional and Developmental Considerations

In some cases, restricted tongue movement does not result from obvious anatomical defects or specific neurological damage but rather from developmental delays, learned behaviors, or general oral motor dysfunction. It is important to emphasize that not all cases of restricted tongue movement are solely due to anatomical or neurological causes. Long-term habits such as thumb sucking or pacifier use, as well as the feeding environment, can contribute to or worsen oral motor dysfunction. This suggests that interventions including behavioral modification and environmental adjustments are necessary, requiring an understanding that goes beyond purely medical causes to encompass learned patterns and habits[58,59]

1. General Oral Motor Dysfunction

General oral motor dysfunction refers to difficulties in the movement and coordination of muscles within the mouth, face, and throat, including the lips, tongue, jaw, and palate. This condition can result from reduced muscle tone, oral sensory dysfunction (either hyposensitivity or hypersensitivity), or movement imbalances. The causes of such dysfunction are often unclear, arising from the complex interaction of multiple factors. When the cause is uncertain, a multidisciplinary team approach is essential to identify the intricate interplay of sensory, motor, developmental, and behavioral factors[41,44,60].
Causes may be related to neurological disorders such as prematurity, underdeveloped oral muscles, cerebral palsy, or other neuromuscular conditions. Developmental delays and low muscle tone are also contributing factors. Diagnosis involves an oral sensory-motor assessment conducted by a speech-language pathologist. This assessment includes detailed observation of facial structure, muscle strength, range of motion, sensory responses, and feeding behaviors. Specialized tools, such as the Pediatric Eating Assessment Tool (PediEAT) or the Schedule for Oral Motor Assessment (SOMA), may be utilized. Treatment focuses on oral motor therapy and speech therapy to improve oral muscle strength, coordination, and control. Therapeutic activities may include tongue exercises, bubble blowing, drinking through a straw, and oral sensory stimulation[61,62].

2. Tongue Thrusting

Tongue thrusting refers to the action of pushing the tongue forward against or between the teeth during swallowing or speaking. It is associated with normal swallowing patterns in infancy and usually resolves as the child grows[63].
However, persistent tongue thrusting can result from prolonged thumb or finger sucking, extended pacifier use, enlarged tonsils or adenoids, or upper airway obstruction caused by allergies. Reduced strength or tone in the oral muscles, genetic factors, and the persistence of the infant’s swallowing reflex can also contribute[58]. Tongue thrusting may cause or exacerbate an open bite. Articulation disorders affecting sounds such as /s/, /z/, /t/, /d/, /n/, and /l/ are also common. Diagnosis is typically made through clinical observation by a speech-language pathologist, often in collaboration with an orthodontist. A physician’s airway examination is also important to rule out airway obstruction as a contributing factor[63].
Treatment includes orofacial myofunctional therapy and speech therapy, aiming to establish a normal oral resting posture in which the tip of the tongue rests on the alveolar ridge and to ensure proper tongue movement during swallowing. Exercises include lip closure, tongue elevation, and training in specific tongue positions[63].

3. Developmental Delays Affecting Oral-Motor Skills

Children with overall developmental delays may also show delays in acquiring the fine motor control and coordination skills required for complex oral movements. This indicates broader neurological immaturity rather than a specific oral pathology[64].
Symptoms resemble those observed in general oral motor dysfunction, including difficulties during feeding developmental stages (e.g., transitioning to solid foods), delayed language development, and challenges with oral hygiene. Diagnosis requires a comprehensive developmental assessment, incorporating a detailed evaluation of oral motor skills. Early intervention through occupational and speech therapy is essential, emphasizing the strengthening and coordination of oral muscles via play-based activities and targeted exercises[64,65].

Comprehensive Diagnostic Approach

Accurate diagnosis of tongue movement restriction without ankyloglossia requires a thorough, often multidisciplinary evaluation that combines clinical observation with instrument-based assessment. The diagnostic process has significantly evolved from subjective visual assessment to reliance on objective, functional evaluation. Individualized treatment strategies can be developed by addressing the root biomechanical and physiological causes rather than only superficial symptoms[15,66].

1. Clinical Evaluation and History Taking

A comprehensive history, including prenatal and birth details, family history of similar conditions, growth patterns, comorbidities, and allergies, is essential. The physical examination should encompass an oral assessment to evaluate oral reflexes and a perioral examination to assess symmetry, sensation, strength, tone, range, speed of movement, and coordination of the lips, jaw, tongue, palate, pharynx, and larynx. Observing infant-caregiver interactions, conducting developmental motor assessments, and evaluating respiratory status, alertness, and self-regulatory behaviors are also critical. A detailed feeding history and observation of feeding patterns, including latching, sucking, chewing, swallowing, meal duration, and signs of distress such as coughing, choking, gagging, hoarseness, or food refusal provide valuable insights into functional limitations[67].
When differentiating from lingual frenulum issues, clinicians must specifically determine whether a restrictive lingual frenulum is the primary limiting factor. If typical symptoms of lingual frenulum syndrome, such as a dimpled or heart-shaped tongue tip or inability to extend the tongue beyond the lower incisors, are not present, other causes should be considered for accurate diagnosis[16].

2. Instrumental Evaluation

Instrumental evaluation is essential for objectively capturing and visualizing the biomechanical processes involved in swallowing. It is also crucial for assessing structures that are not easily visible during a clinical examination.
• Video Fluoroscopic Swallowing Study (VFSS)/Modified Barium Swallow Study (MBSS): This X-ray examination visualizes swallowing movements in real time using various food consistencies and evaluates oral, pharyngeal, and esophageal function. It is highly effective for identifying anatomical or physiological abnormalities, esophageal bolus flow issues, and the presence of aspiration. However, due to radiation exposure and the examination environment, it can be challenging for uncooperative children[68,69].
• Fiberoptic Endoscopic Evaluation of Swallowing (FEES): This procedure involves inserting a flexible endoscope through the nose to directly visualize swallowing function. It is radiation-free and portable. FEES assesses structural integrity, the presence of secretions, and the patient’s response to food and liquids[70].
• Ultrasound Examination: This non-invasive technique allows dynamic observation of the oral phase of swallowing by monitoring the movements of the tongue and hyoid bone[71].
• High-resolution manometry: This technique measures pressure within the pharynx and esophagus, providing objective data on muscle contractions during swallowing[72].

3. Specialized Oral Motor Assessment Tools

Several specialized tools are available to objectively assess oral motor function.
• Schedule for Oral Motor Assessment (SOMA): This observation-based assessment is designed for infants and toddlers aged 0 to 2 years. It is a reliable method for evaluating oral motor function in children with swallowing difficulties and is especially useful for those who may be uncooperative during a Video Fluoroscopic Swallowing Study (VFSS). SOMA evaluates a child’s ability to manipulate various food textures over approximately 20 minutes[62].
• Dysphagia Disorders Survey (DDS): An observation-based assessment tool designed for children with developmental disabilities aged 2 to 21 years. It is recognized for its high reliability and validity in identifying and characterizing swallowing and feeding disorders[73].
• Pediatric Eating Assessment Tool (PediEAT): A parent-reported instrument designed to measure feeding problem symptoms in children aged 6 months to 7 years. Age-specific screening tools are provided to facilitate initial evaluation[61].
• Oral Motor Measurement Scale (OMMS): Developed by the Kennedy Krieger Institute, the OMMS quantifies oral motor and swallowing functions during inpatient rehabilitation for children aged 2 years through adolescence following acquired or traumatic brain injury. It assesses solid and liquid food intake, chewing patterns, tongue lateralization, secretion management, and functional eating speed[67].
• Tongue-o-meter: A biofeedback device that objectively measures tongue strength and endurance. It is useful both for the initial assessment to determine whether tongue weakness is causing dysphagia and for guiding and monitoring progress in tongue-strengthening exercises[74,75].

Management and Therapeutic Interventions

Treatment for tongue movement restrictions in the absence of a lingual frenulum is highly individualized, depending on the underlying cause, severity of symptoms, and the child’s age and developmental stage. A multidisciplinary team approach involving specialists such as speech-language pathologists, neurologists, geneticists, otolaryngologists, dentists, occupational therapists, and nutritionists is often essential[76,77].

1. Speech-Language Pathology and Oral-Motor Therapy

Speech-language pathologists play a central role in assessing and treating oral motor dysfunction, including restrictions in tongue movement. Treatment focuses on improving the strength, coordination, and control of the muscles involved in eating, drinking, and speaking[44,63].
• Oral Motor Exercises: Targeted exercises designed to strengthen muscle strength and coordination, such as pressing the tongue against a spoon or tongue depressor and curling the tongue toward the palate. Playbased therapy for younger children often incorporates activities like blowing bubbles, drinking through a straw, whistling, and pursing the lips[23,44,63].
• Sensory Stimulation Techniques: Explore a variety of food textures and oral sensations to enhance oral sensory awareness and tolerance, particularly in children with hypersensitivity or hyposensitivity[78].
• Swallowing Strategies: Collaborate with children to develop more effective chewing and swallowing techniques and specific maneuvers to enhance the safety and efficiency of swallowing[44].
• Dietary Modifications: Adjust the viscosity, texture, temperature, or portion size of foods and liquids to ensure safe swallowing and adequate nutritional intake[44].
• Adaptive Devices: Utilizing specialized equipment and tools to control bolus size or liquid flow rate, thereby enhancing independence and improving swallowing safety[44].
• Oral-Facial Muscle Function Therapy: This therapy is especially beneficial for conditions like tongue thrusting. It focuses on establishing a normal oral resting position, with the tongue tip placed against the alveolar ridge, and promoting proper tongue movement during swallowing. The therapy typically includes targeted tongue positioning exercises and activities designed to enhance lip mobility[23,63].

2. Treatment and Surgical Management by Condition

Surgical intervention is performed based on the underlying cause of restricted tongue movement.
• Neurological Conditions: For conditions such as hypoglossal nerve palsy, treatment primarily focuses on addressing the underlying cause, such as managing tumors or treating infections. In cases of stroke or brain injury, rehabilitation is essential and is often combined with speech therapy[76].
• Muscular and Genetic Disorders: The focus is often on supportive care for feeding and breathing difficulties, including physical therapy and sensory stimulation programs for congenital myopathies. For syndromes such as macroglossia, surgical reduction of tongue size may be considered if symptoms are severe and cause significant functional impairment. Severe feeding disorders may require nutritional support, including tube feeding[46].
• Structural and Anatomical Abnormalities: For mandibular fractures, posture modifications (e.g., prone sleeping position) or respiratory support (e.g., breathing tubes) may be provided until jaw growth stabilizes. Oral tumors and cysts are often surgically removed if they interfere with function or cause discomfort. Craniofacial abnormalities, such as cleft palate, typically require surgical correction by a multidisciplinary team[79].
• Acquired and Iatrogenic Causes: Following postoperative complications or traumatic injury, careful observation until nerve recovery occurs may be necessary, followed by rehabilitation therapy. Management of the side effects of radiation therapy focuses on symptom control (pain, xerostomia) and swallowing therapy[76].

Conclusion

Tongue movement restriction in children is often attributed to the lingual frenulum, but can arise from various causes that require careful differential diagnosis[6,15]. The increasing prevalence of tongue-tie diagnoses, coupled with a lack of clear evidence linking it causally to reported symptoms, underscores the need for clinicians to be highly skilled in identifying and distinguishing other causes of tongue restriction[6,76]. The overlapping symptoms observed in conditions such as feeding disorders, language impairments, and swallowing difficulties further complicate diagnosis[6,44]. Therefore, a comprehensive approach is essential, extending beyond superficial observation to include a detailed medical history, thorough physical examination, and objective instrumental assessments[15].
Tongue movement restriction does not merely indicate morphological constraints[76]. It can also result from intrinsic muscle weakness, rigidity, unbalanced size, or even learned behaviors and environmental factors[44]. Furthermore, oral motor dysfunction rarely presents as an isolated symptom, which often reflects broader systemic issues, particularly those related to respiratory function and overall development[76]. The fact that iatrogenic tongue restriction can be induced highlights the dynamic nature of oral motor function and underscores the need for ongoing vigilance in patient care[15,76].

NOTES

Conflicts of Interest

The author has no potential conflicts of interest to disclose.

CRediT authorship contribution statement

Hyeon-Heon Lee: Conceptualization, Methodology, Investigation, Writing - original draft, Writing - review and editing.

Table 1.
Comparative overview of assessment tools
Tool (Abbreviation) Assessment Items Score Strengths Weaknesses
Assessment Tool for Lingual Frenulum Function (ATLFF) 5 appearance + 7 function items (e.g., tongue lift, extension, spread, peristalsis, and frenulum elasticity) 0 - 14 each for appearance/function (Total up to 28; <11 or 12 function = significant tie) Comprehensive; research-based; both structure and function; used for infants and children Complex to apply; requires examiner training; subjective in some scoring
Bristol Tongue Assessment Tool (BTAT) 4 items: tongue tip appearance, frenulum attachment to gum, lift, and protrusion (each 0 - 2 points) 0 - 8 Quick, simple, and objective; minimal training needed; suitable for screening Does not assess feeding function; limited to physical observation
TABBY (Tongue-tie And Breastfed Babies assessment) Visual version of BTAT-the same 4 items represented with pictures 0 - 8 Very easy training; helpful for visual learners; available in many languages Same as BTAT-no direct feeding assessment; pictorial judgments can be subjective
Lingual Frenulum Protocol for Infants (LFPI) Anatomy, non-nutritive and nutritive sucking, tongue movements, functional signs Multiple domains scored; e.g., anatomy 0 - 2, function 0 - 6 points (different by domain) Extensive; covers anatomy, function, and feeding; high clinical value Lengthy; requires training; not yet standardized globally
Neonatal Tongue Screening Test (NTST) Appearance (e.g., tongue shape, frenulum), limited sucking/function assessment 0 - 4 or 0 - 5 Very fast; efficient for mass screening; minimal training Not validated in all regions; lower sensitivity/ specificity in some studies
Siriraj Tongue-Tie Score (STT) Breastfeeding, tongue shape, lift, extension, and frenulum; both appearance- and feeding-focused Each item scored 0 - 2; cumulative scoring Emphasizes breastfeeding (practical in lactation clinics); simple scale Limited literature outside Thailand; not universally validated

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Hyeon-Heon Lee
https://orcid.org/0009-0004-1449-7553

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