Identification and Management of Ankyloglossia and Its Effect on Breastfeeding in Infants: Clinical Report.

Ankyloglossia refers to a congenitally tight lingual frenulum that limits the motion of the tongue. Whether the release of a tight lingual frenulum in neonates improves breastfeeding is not clear. Because many of the symptoms of ankyloglossia overlap those of other breastfeeding difficulties, a team partnership is necessary.

Ankyloglossia in Australia: Experiences and perspectives of parents and caregivers.

OBJECTIVES: To investigate the perspectives and experiences of parents of infants with ankyloglossia in Australia. METHOD: Two hundred and sixty-seven parents across Australia responded to an online survey including their experiences of having a child with ankyloglossia. Descriptive statistics were used to analyse quantitative data. RESULTS: Participants were from all Australian states and territories, with the majority residing in New South Wales and Victoria. Ankyloglossia diagnoses typically occurred within the first two weeks of life, driven mainly by difficulties with feeding. Lactation consultants played a prominent role in diagnosis, particularly in rural areas. Surgical interventions were common, with frenotomy using scissors or scalpel being the primary treatment. Aftercare recommendations, including stretching the frenotomy wound, were prevalent. Most parents received education about ankyloglossia, primarily through verbal and written information. Parents generally reported high satisfaction with the effectiveness of treatments, favouring surgical interventions. However, satisfaction with health professionals' support did not consistently correlate with the likelihood of consenting to the treatment again, and those who conducted their own research on ankyloglossia expressed lower satisfaction with health professionals' support. CONCLUSIONS: The experiences of Australian parents in their infant's diagnosis, management and education of ankyloglossia varied greatly. Clinical guidelines for all relevant health professionals are needed to ensure standardised diagnosis and management processes. In future, this will help guide evidence-based diagnosis and intervention for infants with ankyloglossia.

Maxillomandibular Fusion Along with Fusion of Dorsal Surface of the Tongue to the Hard Palate-A Case Report.

RATIONALE: This case report presents a rare combination of congenital anomalies in an otherwise healthy male infant born at 36 weeks. The infant was diagnosed with congenital maxillomandibular synechia, ectrodactyly, and ankyloglossia superior syndrome (ASS). PATIENT CONCERNS: Inability to open the mouth completely, feeding challenges, and a cleft palate. The infant was stabilized through successful positive pressure ventilation via a face mask at birth and enteral feeding was initiated via a feeding gastrostomy. EXAMINATION: Diagnostic tests revealed a midline palatal cleft, hypoplastic jaws, persistent metopic suture, and a bony fusion at the midline. TREATMENT: Sectioning of the bony spur along the midline and achieving a mouth opening of 2 cm post-manipulation. The patient is under follow-up, with future treatment plans including cleft palate correction at 12 months and potential frontomandibular and lower jaw advancement depending on growth trajectories. TAKEAWAY LESSONS: This case underscores the complexity of managing multiple congenital anomalies and the need for individualized treatment plans.

Posterior tongue tie: that is a thing?

PURPOSE OF REVIEW: The purpose of this review is to examine the current research of the posterior tongue tie and how it relates to breast feeding, solid feeding, and speech. RECENT FINDINGS: Recent findings show that the posterior tongue tie may play a role in effective breast feeding. SUMMARY: Ankyloglossia is the term used for the restriction of the movement of the tongue that impairs certain functions such as breastfeeding or bottle feeding, feeding with solids, and speech. Cadaver studies have shown that there can be a restriction of the tongue and oral tissues in some people relative to others. In some breast-feeding studies, releasing the posterior tie has been shown to improve certain aspects of tongue movement. There is little evidence for or against posterior tongue ties contributing to other problems such as speech and solid feeding. This article goes into depth about the current studies on posterior ankyloglossia.

Influence of Neonatal Ankyloglossia on exclusive breastfeeding in the six first months of life: a cohort study. / Influência da Anquiloglossia neonatal na amamentação exclusiva nos seis primeiros meses de vida: estudo de coorte.

PURPOSE: To analyze the influence of ankyloglossia on the prevalence and duration of exclusive breastfeeding of full-term infants up to the sixth month of life. METHODS: Prospective cohort study, carried out with 225 mother-infant dyads who were followed up in the first six months of life in a center specialized in breastfeeding in a tertiary hospital. Full-term infants with asymptomatic ankyloglossia (no need for surgery) were compared with infants without change at monthly follow-up. Ankyloglossia was diagnosed using the Bristol Tongue Assessment Tool, with a positive diagnosis being considered for those with a score less than or equal to 5 considering functional and anatomical aspects. Statistical analyzes were performed using descriptive statistics, logistic regression (weaning determinants), relative risk, and survival curves (to analyze breastfeeding duration between groups with and without ankyloglossia). RESULTS: Ankyloglossia was associated with weaning (considered even partial) before the sixth month of life. After adjusted analysis, a higher risk of weaning was detected in infants with this alteration, with a risk present from the second month of life. In the survival analysis, the duration of breastfeeding in infants with ankyloglossia was shorter when compared to children without alterations. CONCLUSION: Compared to infants with normal lingual frenulum, babies with ankyloglossia had shorter exclusive breastfeeding time, but well above the average observed in the general population. The risk of weaning for this group was also higher.

OBJETIVO: Analisar a influência da anquiloglossia na prevalência e no tempo de exclusividade do aleitamento materno de lactentes a termo até o sexto mês de vida. MÉTODOS: Estudo de coorte prospectivo, realizado com 225 díades mãe-bebê que foram acompanhadas nos seis primeiros meses de vida em centro especializado em amamentação em um hospital terciário. Lactentes a termo com anquiloglossia do tipo assintomática (sem necessidade de cirurgia) foram comparados com lactentes sem alteração em um acompanhamento mensal. O diagnóstico de anquiloglossia foi realizado através do Bristol Tongue Assessment Tool, sendo considerados com diagnóstico positivo aqueles com escore menores ou iguais a 5 considerando os aspectos funcionais e anatômicos. As análises estatísticas foram realizadas através de estatística descritiva, regressão logística (determinantes do desmame), risco relativo e curvas de sobrevivência (para analisar o tempo de aleitamento entre os grupos com e sem anquiloglossia). RESULTADOS: A anquiloglossia esteve associada com o desmame (considerado ainda que parcial) antes do sexto mês de vida. Após análise ajustada, foi detectado maior risco de desmame nos lactentes com a alteração presente, com risco presente a partir do segundo mês de vida. Na análise de sobrevida, o tempo de aleitamento nos lactentes com anquiloglossia foi menor quando comparadas às crianças sem alteração. CONCLUSÃO: Em comparação com lactentes com freio lingual normal, os bebês com anquiloglossia apresentaram tempo menor de aleitamento exclusivo, porém bem acima da média observada na população geral. O risco de desmame para este grupo também foi maior.

The influence of tight tongue-ties and tongue-tie clips on speech development.

Ankyloglossia or tongue-tie is a condition where the anatomical variation of the sublingual frenulum can limit normal tongue function. In Denmark, as in other countries, an increase in the number of children treated for ankyloglossia has been described over the past years. Whether or not ankyloglossia and its release affect the speech has also been increasingly discussed on Danish television and social media. In this review, the possible connection between ankyloglossia, its surgical treatment, and speech development in children is discussed.

Comparison of ChatGPT knowledge against 2020 consensus statement on ankyloglossia in children.

OBJECTIVE: This paper evaluates ChatGPT's accuracy and consistency in providing information on ankyloglossia, a congenital oral condition. Assessing alignment with expert consensus, the study explores potential implications for patients relying on AI for medical information. METHODS: Statements from the 2020 clinical consensus statement on ankyloglossia were presented to ChatGPT, and its responses were scored using a 9-point Likert scale. The study analyzed the mean and standard deviation of ChatGPT scores for each statement. Statistical analysis was conducted using Excel. RESULTS: Among the 63 statements assessed, 67 % of ChatGPT responses closely aligned with expert consensus mean scores. However, 17 % (11/63) were statements in which the ChatGPT mean response was different from the CCS mean by 2.0 or greater, raising concerns about ChatGPT's potential influence in disseminating uncertain or debated medical information. Variations in mean scores highlighted discrepancies, with some statements showing significant deviations from expert opinions. CONCLUSION: While ChatGPT mirrored medical viewpoints on ankyloglossia, alignment with non-consensus statements raises caution in relying on it for medical advice. Future research should refine AI models, address inaccuracies, and explore diverse user queries for safe integration into medical decision-making. Despite potential benefits, ongoing examination of ChatGPT's power and limitations is crucial, considering its impact on health equity and information access.

Alternative Therapies for Ankyloglossia-Associated Breastfeeding Challenges: A Systematic Review.

Background: Ankyloglossia (AG) diagnoses are increasingly common, and management is not standardized. Nonsurgical alternative therapies are frequently recommended in conjunction with or instead of frenotomy, with uncertain evidence. Objective: To evaluate the efficacy of nonsurgical alternative therapies (chiropractic care, myofunctional therapy, and osteopathy) in improving breastfeeding for infants diagnosed with AG. Methods: PubMed, Embase, CINAHL, Scopus, Web of Science, Clinicaltrials.gov, and Google Scholar were searched (September-October 2023). Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines were followed. A librarian-designed search included the terms "Ankyloglossia," "Non-surgical," "myofunctional therapy," "chiropractic," "osteopathy," and related therapies, with no date restrictions. English language studies of infants <24 months with AG and alternative therapy were included. Risk-of-bias evaluation used Newcastle-Ottawa Scale (NOS). Results: Of 1,304 identified articles, four studies (2016-2022) met inclusion criteria (two cross-sectional, one case report, and one case series). All studies reported frenotomy in combination with alternative therapy yielded favorable outcomes for maternal pain, weight gain, feeding duration, and maintenance of latch. The risk of bias was moderate for two studies, low for the case series, and not calculated for the case report, which has an inherent high risk of bias. All studies lacked control or comparator groups preventing definitive conclusions about the role of alternative therapies in AG. Conclusion: Although some studies suggest the potential benefits of combining alternative therapies with surgery for AG-related breastfeeding issues, the lack of control groups renders the evidence inconclusive. Nonsurgical approaches alone currently lack sufficient evidence. As these alternative therapies gain popularity, rigorous research is crucial to determine their cost-effectiveness and role in managing AG.

Oral Structural Dysphagia in Children.

Oral causes of dysphagia in infancy may involve the lips, the tongue, or the palate. Whereas ankyloglossia is commonly diagnosed in infants with dysphagia, assessment of the need for surgical intervention may be less straightforward. Tongue size (macroglossia) may be associated with dysphagia as it may cause limitation of movement of the food or milk bolus by the lips or cheeks. Congenital conditions such as cleft lip and palate, micrognathia, or craniofacial microsomia may also be associated with dysphagia. Diagnosis and treatment of these conditions can be improved with the engagement of lactation and feeding experts as well as multidisciplinary craniofacial teams.

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

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

Speech Outcomes of Frenectomy for Tongue-Tie Release: A Systematic Review and Meta-Analysis.

OBJECTIVE: Tongue-tie, which is also known as ankyloglossia, is a common condition where the lingual frenulum is unusually tight or short. While most literature investigates the impact of tongue-tie on breastfeeding, recent articles have examined its role in speech production in children. However, these have not previously been reviewed systematically. This study aims to determine the impact of tongue-tie on speech outcomes and assess whether frenectomy can improve speech function. METHODS: In this systematic review, we conducted a comprehensive search of PubMed/MEDLINE, Cochrane Library, Embase, and speechBITE to analyze primary studies investigating the impact of frenectomy for tongue-tie on speech outcomes. We extracted data regarding patient age, male to female ratio, procedure type, follow-up time, and speech outcomes and ran statistical analyses to determine if frenectomy for tongue-tie leads to improvement in speech issues in pediatric patients. Speech outcomes extracted were subjectively measured based on the interpretation of a speech and language pathologist or parent. RESULTS: Our analysis included 10 studies with an average patient age of 4.10 years, and average cohort size of 22.17 patients. Overall, frenectomy for tongue-tie was associated with an improvement in speech articulation (0.78; 95% CI: 0.64-0.87; P < .01). Increasing patient age was found to be negatively correlated with post-frenectomy speech outcomes (P = .01). However, this relationship disappeared in the adjusted model. CONCLUSION: Overall, we conclude that frenectomy is a suitable treatment to correct speech issues in select patients with tongue-tie if caught early in childhood. Despite the limited investigations around speech outcomes post-frenectomy, these results are informative to providers treating tongue-tie.

Is Public Interest Associated with Real-World Management of Ankyloglossia?

OBJECTIVES: Assess the relationship between public interest in ankyloglossia as determined by internet search volume and real-world medical claims data. STUDY DESIGN: Retrospective Cohort Study. SETTING: This retrospective cohort study was conducted using claims data from the Merative™ Marketscan® Research Databases. The internet search data was collected from Google Trends. METHODS: Annual Google Trends data were compiled using search terms associated with "ankyloglossia" and "frenotomy" for the years 2011 to 2021. We obtained incidence of ankyloglossia diagnoses and frenotomy procedures in children under 12 months from Marketscan relative to all infants enrolled. We compared associations between search and incidence data among US states and over time. RESULTS: Google search correlated with ankyloglossia incidence (r = 0.4104, P = .0031) and with frenotomy incidence (r = 0.4062, P = .0034) per state. Ankyloglossia diagnoses increased with Google search index (coefficient = 0.336, 95% confidence interval [CI] 0.284, 0.388) and year (coefficient = 0.028, 95% CI 0.025, 0.031). Similarly, frenotomy procedures increased with Google search index (coefficient = 0.371, 95% CI 0.313, 0.429) and year (coefficient = 0.027, 95% CI 0.024, 0.030). CONCLUSIONS: Associations between online ankyloglossia search trends and both diagnosis and treatment rates, persist across US regions and timeframes. Internet search trends are pivotal in shaping pediatric health care decisions, driving clinical consensus, and disseminating evidence-based information.

Ankyloglossia and Breastfeeding Duration: A Multicenter Birth Cohort Study.

Background and Objective: The prevalence of ankyloglossia and its impact on breastfeeding practices may be overestimated, leading to surgical overtreatment in newborns. The study was conducted to estimate the prevalence of ankyloglossia in the first year of life and investigate the association with exclusive and total breastfeeding duration in different regions of Brazil. Materials and Methods: This multicenter prospective cohort study involved the recruitment of mother-infant pairs soon after childbirth in public hospitals in three state capitals in Brazil. Interviews were held with the mothers after birth, at 6 and 12 months to collect sociodemographic variables and data on exclusive and total breastfeeding duration. At 12 months of age, the children were submitted to a dental examination for classification of the lingual frenulum using the Bristol Tongue Assessment Tool. Data analysis involved Poisson regression with robust variance, with the calculation of unadjusted and adjusted relative risk (RR). Results: The final sample was composed of 293 children. The prevalence of defined and suspected ankyloglossia was 1% and 4.8%, respectively, totaling 5.8% (confidence interval [95% CI]: 3.1-8.5). No significant difference was found in the prevalence of exclusive and total breastfeeding at 1, 4, and 6 months between children with defined/suspected ankyloglossia and those without ankyloglossia. The multivariable analysis showed that the probability of the child achieving 6 months of breastfeeding did not differ between groups (RR = 0.98; 95% CI: 0.79-1.23; p = 0.907). Conclusion: The prevalence of defined ankyloglossia was very low and defined/suspected ankyloglossia was not associated with exclusive or total breastfeeding duration. Clinical Trial Registration: Registered with clinicaltrials.gov (n° NCT03841123).

The buccal frenum: Trends in diagnosis and indications for treatment of buccal-ties among 466 healthcare professionals.

BACKGROUND: The buccal frenum is connective tissue that adheres the mucosa of the cheek to the alveolar process. When restricted, this condition is commonly known as a buccal- or cheek-tie. Restrictive buccal frena are often treated during tongue- and lip-tie procedures, yet widely accepted classification, diagnostic and treatment guidelines are lacking. OBJECTIVE: Provide a scoping review on the evaluation and management of buccal-ties, including diagnosis, classification, symptoms and treatment, by surveying healthcare providers with experience evaluating and managing oral restrictions. METHODS: Literature review and IRB-approved survey to assess practice patterns among healthcare providers identified from online directories of tongue-tie release providers and associated allied health professionals. RESULTS: A multidisciplinary group of 466 providers responded. About 87% indicated that they assess buccal restrictions. Evaluation methods included finger sweep (89.1%), visual inspection (76.4%), tissue blanching (66.5%) and functional assessment (53.4%). Around 94% of providers reported that objective and subjective findings are both needed for diagnosis and that an estimated 5%-10% of infants may be affected. About 70% of providers release buccal-ties (if needed) simultaneously with tongue-ties, and 76.8% recommend post-operative stretches as necessary for optimal healing. Respondents indicated a need for further research, evidence-based assessments, a classification system and treatment protocols. CONCLUSION: Evaluating a buccal frenum to diagnose a symptomatic buccal-tie relies upon visual inspection, palpation and assessment of oral function. Survey data and clinical experience are summarized to review classification systems, diagnostic/evaluation criteria and treatment recommendations as a foundational cornerstone for future works to build upon.

Ankyloglossia and malocclusion: A systematic review and meta-analysis.

BACKGROUND: Ankyloglossia is a congenital alteration that affects the tongue's mobility, influencing craniofacial development; however, its association with malocclusion is still unclear. This systematic review and meta-analysis investigated the association between ankyloglossia and malocclusion in patients. TYPES OF STUDIES REVIEWED: The authors conducted a systematic review and meta-analysis of observational studies that investigated the association between malocclusion and the presence of ankyloglossia in any age group, used any assessment tool to diagnose ankyloglossia, and considered all types of malocclusion. The authors conducted searches in 8 electronic databases through July 1, 2022. They used Joanna Briggs Institute appraisal tools to assess the methodological quality and the Grading of Recommendations, Assessment, Development and Evaluations approach to assess the certainty of the evidence. RESULTS: Eleven studies (5 cross-sectional, 6 case-control) included 2,904 patients and over 13 occlusal alterations. The certainty of the evidence ranged from low through very low. Patients with Angle Class III malocclusion had similar or more inserted lingual frenula than patients with Class I malocclusion (standard mean difference, 0.37; 95% CI, -0.50 to 1.25) and Class II malocclusion (standard mean difference, 0.55; 95% CI, -0.52 to 1.63). Patients with Class III malocclusion had clinically significant increased mouth opening reduction compared with patients with Class I malocclusion (mean difference, 6.67; 95% CI, 4.01 to 9.33) and Class II malocclusion (mean difference, 5.04; 95% CI, 2.35 to 7.72) patients. PRACTICAL IMPLICATIONS: There is uncertain evidence that ankyloglossia may be associated with the development of occlusal alterations. Ankyloglossia did not influence the Angle classification. Clinicians should closely follow children with ankyloglossia to evaluate whether this condition interferes with the occlusion. Future research should prospectively follow up on the long-term association between ankyloglossia and malocclusion. The protocol was registered a priori in the PROSPERO database (CRD42021248034). No amendments were deemed necessary after the registration of the protocol. Some subgroup analyses planned a priori were not possible, such as the severity of ankyloglossia, sex, and age, due to the lack of studies reporting data for these subgroups.

Quantitative impact of frenotomy on breastfeeding: a systematic review and meta-analysis.

BACKGROUND: Symptoms related to infant ankyloglossia/tongue-tie may deter mothers from breastfeeding, yet frenotomy is controversial. METHODS: Databases included PubMed, Embase, CINAHL, PsycINFO, Web of Science, and Google Scholar from 1961-2023. Controlled trials and cohort studies with validated measures of surgical efficacy for breastfeeding outcomes were eligible. Meta-analyses synthesized data with inverse-variance weighting to determine standardized mean differences (SMD) between pre-/postoperative scores. RESULTS: Twenty-one of 1568 screened studies were included. Breastfeeding self-efficacy improved significantly post-frenotomy: medium effect after 5-10 days (SMD 0.60 [95% CI: 0.48, 0.71; P < 0.001]), large effect after 1 month (SMD 0.91 [CI: 0.79, 1.04; P < 0.001]). Nipple pain decreased significantly post-frenotomy: large effect after 5-15 days (SMD -1.10 [CI: -1.49, -0.70; P < 0.001]) and 1 month (SMD -1.23 [CI: -1.79, -0.67; P = 0.002]). Frenotomy had a medium effect on infant gastroesophageal reflux severity at 1-week follow-up (SMD -0.63 [CI: -0.95, -0.31; P = 0.008]), with continued improvement at 1 month (SMD -0.41 [CI: -0.78, -0.05; P = 0.04]). From LATCH scores, breastfeeding quality improved after 5-7 days by a large SMD of 1.28 (CI: 0.56, 2.00; P = 0.01). CONCLUSIONS: Providers should offer frenotomy to improve outcomes in dyads with ankyloglossia-associated breastfeeding difficulties. PROTOCOL REGISTRATION: PROSPERO identifier CRD42022303838 . IMPACT: This systematic review and meta-analysis showed that breastfeeding self-efficacy, maternal pain, infant latch, and infant gastroesophageal reflux significantly improve after frenotomy in mother-infant dyads with breastfeeding difficulties and ankyloglossia. Providers should offer frenotomy to improve breastfeeding outcomes in symptomatic mother-infant dyads who face challenges associated with ankyloglossia.

Relationship between Ankyloglossia and Breastfeeding: A Bibliometric Review

ABSTRACT Objective: To assess global trends in the publication of studies investigating the association between ankyloglossia and breastfeeding. Material and Methods: An electronic search was performed in the Scopus database without restrictions. Observational studies and clinical trials were included. Bibliometric indices such as publication year, authors, co-authors, journals, field of knowledge, countries, and the most cited keywords were analyzed using the VOSviewer program. Results: The search retrieved 350 studies, and 68 were selected. The first article was published in 2000 in the United States. The United States presented the highest number of publications (n=21), followed by Brazil (n=9) and the United Kingdom (n=9). An increase in publications on this theme was observed in 2013; 2021 was the year with the highest number of publications (n=14). The most common word was "frenulum". The authors with the highest number of publications were Botze and Dollbert from Israel (n=3), Ghaheri, and Mace from the United States (n=3). Among the journals, "Breastfeeding Medicine" presented the highest number of publications (n=7), followed by the "International Journal of Pediatric Otorhinolaryngology" (n=6), "CODAS" (n=5), "Journal of Human Lactation" (n=4) and "Pediatrics" (n=3); the latter published the top-cited studies, with 412 citations. Conclusion: There has been an increase in recent articles evaluating the correlation between ankyloglossia and breastfeeding, indicating the growing interest of researchers in this field.