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).

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.

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.

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.

The development of a specialist tongue tie assessment clinic for neonates.

OBJECTIVES: Ankyloglossia or tongue tie is a condition where the lingual frenulum restricts tongue movement, negatively impacting breastfeeding. An increasing number of infants diagnosed with ankyloglossia may lead to unnecessary procedures. To limit the overtreatment of this problem, we established a specialist clinic to provide the best available evidence-based care to the mother-infant dyad. METHODS: We discuss the development of a specialist tongue tie assessment clinic in our unit. RESULTS: From January to October 2023, there were 162 visits to the clinic by 157 patients. During this time, there were 96 frenotomies performed. CONCLUSIONS: The mother-infant dyad is a vulnerable patient group. Establishing a specialist tongue tie assessment clinic with otolaryngology and lactation expertise provides the best available evidence-based care.

Nota técnica reforça procedimentos para identificação da anquiloglossia em recém-nascidos

O Ministério da Saúde, por meio da Secretaria de Atenção Primária (SAPS), acaba de publicar nota técnica para orientar os profissionais e serviços de saúde sobre a identificação precoce da anquiloglossia em recém-nascidos e, assim, estabelecer o fluxo do atendimento dessa população na rede de atenção à saúde. A condição congênita, conhecida popularmente como “língua presa”, pode causar problemas no desenvolvimento oral da criança, além de dificultar a amamentação.

Manejo odontológico del paciente con frenillo lingual corto: a propósito de un caso / Dental management of the patient with short lingual frenulum: a case report

Introducción: El frenillo lingual corto puede provocar dificultades en la lactancia durante los primeros meses de vida en el neonato. A partir del desarrollo del lenguaje puede ser la causa de una fonética inadecuada. El tratamiento quirúrgico del frenillo debe ir acompañado por un adecuado tratamiento multidisciplinar para favorecer la corrección de las dificultades en la pronunciación. El objetivo del presente artículo es presentar un caso clínico representativo del diagnóstico, tratamiento quirúrgico y rehabilitación miofuncional del frenillo lingual corto, así como analizar la evidencia científica disponible. Caso clínico: Se presenta un paciente varón de 6 años diagnosticado de anquiloglosia severa que acude por dificultad en la pronunciación del fonema “RR”. Se realiza el tratamiento quirúrgico del frenillo mediante electrobisturí y su seguimiento por un logopeda. Conclusiones: El diagnóstico de la patología y la planificación quirúrgica y miofuncional deben tener en cuenta la clasificación del frenillo, la edad del paciente y la anatomía de la región. El tratamiento de la patología asociada al frenillo lingual corto requiere de un equipo multidisciplinar para evitar la recidiva. (AU)

Introduction: The short lingual frenulum can cause breastfeeding difficulties during the first months of life in the neonate. From language development they can be the cause of inadequate phonetics. Surgical treatment of the frenulum must be accompanied by adequate multidisciplinary treatment to improve the correction of pronunciation difficulties. The objective of this article is to present a representative clinical case of the diagnosis, surgical treatment and myofunctional rehabilitation of the short lingual frenulum, as well as to update the available scientific evidence. Case Report: A 6-year-old male patient is presented, diagnosed with severe ankyloglossia who came due to difficulty in pronouncing the phoneme “RR”. Surgical treatment of the frenulum was performed using electrocautery and its follow-up by a speech therapist. Conclusions: The diagnosis of the pathology and the surgical and myofunctional planning must take into account the classification of the frenulum, the age of the patient and the anatomy of the region. The treatment of the pathology associated with short lingual frenulum requires a multidisciplinary team to avoid recurrence. (AU)

Manejo odontológico del paciente con frenillo lingual corto: a propósito de un caso / Dental management of the patient with short lingual frenulum: a case report

Introducción: El frenillo lingual corto puede provocar dificultades en la lactancia durante los primeros meses de vida en el neonato. A partir del desarrollo del lenguaje puede ser la causa de una fonética inadecuada. El tratamiento quirúrgico del frenillo debe ir acompañado por un adecuado tratamiento multidisciplinar para favorecer la corrección de las dificultades en la pronunciación. El objetivo del presente artículo es presentar un caso clínico representativo del diagnóstico, tratamiento quirúrgico y rehabilitación miofuncional del frenillo lingual corto, así como analizar la evidencia científica disponible. Caso clínico: Se presenta un paciente varón de 6 años diagnosticado de anquiloglosia severa que acude por dificultad en la pronunciación del fonema “RR”. Se realiza el tratamiento quirúrgico del frenillo mediante electrobisturí y su seguimiento por un logopeda. Conclusiones: El diagnóstico de la patología y la planificación quirúrgica y miofuncional deben tener en cuenta la clasificación del frenillo, la edad del paciente y la anatomía de la región. El tratamiento de la patología asociada al frenillo lingual corto requiere de un equipo multidisciplinar para evitar la recidiva. (AU)

Introduction: The short lingual frenulum can cause breastfeeding difficulties during the first months of life in the neonate. From language development they can be the cause of inadequate phonetics. Surgical treatment of the frenulum must be accompanied by adequate multidisciplinary treatment to improve the correction of pronunciation difficulties. The objective of this article is to present a representative clinical case of the diagnosis, surgical treatment and myofunctional rehabilitation of the short lingual frenulum, as well as to update the available scientific evidence. Case Report: A 6-year-old male patient is presented, diagnosed with severe ankyloglossia who came due to difficulty in pronouncing the phoneme “RR”. Surgical treatment of the frenulum was performed using electrocautery and its follow-up by a speech therapist. Conclusions: The diagnosis of the pathology and the surgical and myofunctional planning must take into account the classification of the frenulum, the age of the patient and the anatomy of the region. The treatment of the pathology associated with short lingual frenulum requires a multidisciplinary team to avoid recurrence. (AU)

Maternal folic acid supplementation and the risk of ankyloglossia (tongue-tie) in infants; a systematic review.

BACKGROUND: Maternal folic acid supplementation is protective against the development of neural tube defects (NTDs) in babies. However, recent public-facing communications have raised concerns about a causal relationship between folic acid supplementation, particularly after the first trimester, and ankyloglossia (tongue-tie) in infants. Non-evidence-based communications are potentially harmful because they could adversely affect adherence to folic acid supplementation, increasing NTD occurrence. This study aimed to review evidence on the relationships between maternal folic acid supplementation during preconception and/or pregnancy and the risk of ankyloglossia in infants. METHODS: We searched the databases MEDLINE, EMBASE, Cochrane CENTRAL, and Scopus. We searched for observational, and interventional studies, and systematic reviews investigating the effect of maternal folic acid supplementation during preconception or pregnancy on the occurrence of ankyloglossia in offspring. The search was registered on PROSPERO on 01/12/2022, ID: CRD42022375862. RESULTS: The database searches yielded 93 articles. After removing duplicates and screening titles and abstracts, 26 remained. One article was judged relevant for inclusion in analyses; a case-control study that directly mentions the relationship between folic acid supplementation and ankyloglossia. This study reported that regular intake of folic acid supplements was higher in women with infants with ankyloglossia. However, this study has limitations regarding design, selection bias, and confounding, calling the findings into question. CONCLUSIONS: Insufficient evidence exists for a relationship between folic acid supplementation and ankyloglossia. Currently, the benefits of folic acid supplementation far outweigh the risks. This must be clearly communicated to patients by their clinicians during preconception and antenatal care.

Frenotomy with breastfeeding support versus breastfeeding support alone for infants with tongue-tie and breastfeeding difficulties: the FROSTTIE RCT.

Background: Tongue-tie can be diagnosed in 3-11% of babies, with some studies reporting almost universal breastfeeding difficulties, and others reporting very few feeding difficulties that relate to the tongue-tie itself, instead noting that incorrect positioning and attachment are the primary reasons behind the observed breastfeeding difficulties and not the tongue-tie itself. The only existing trials of frenotomy are small and underpowered and/or include only very short-term or subjective outcomes. Objective: To investigate whether frenotomy is clinically and cost-effective to promote continuation of breastfeeding at 3 months in infants with breastfeeding difficulties diagnosed with tongue-tie. Design: A multicentre, unblinded, randomised, parallel group controlled trial. Setting: Twelve infant feeding services in the UK. Participants: Infants aged up to 10 weeks referred to an infant feeding service (by a parent, midwife or other breastfeeding support service) with breastfeeding difficulties and judged to have tongue-tie. Interventions: Infants were randomly allocated to frenotomy with standard breastfeeding support or standard breastfeeding support without frenotomy. Main outcome measures: Primary outcome was any breastmilk feeding at 3 months according to maternal self-report. Secondary outcomes included mother's pain, exclusive breastmilk feeding, exclusive direct breastfeeding, frenotomy, adverse events, maternal anxiety and depression, maternal and infant NHS health-care resource use, cost-effectiveness, and any breastmilk feeding at 6 months of age. Results: Between March 2019 and November 2020, 169 infants were randomised, 80 to the frenotomy with breastfeeding support arm and 89 to the breastfeeding support arm from a planned sample size of 870 infants. The trial was stopped in the context of the COVID-19 pandemic due to withdrawal of breastfeeding support services, slow recruitment and crossover between arms. In the frenotomy with breastfeeding support arm 74/80 infants (93%) received their allocated intervention, compared to 23/89 (26%) in the breastfeeding support arm. Primary outcome data were available for 163/169 infants (96%). There was no evidence of a difference between the arms in the rate of breastmilk feeding at 3 months, which was high in both groups (67/76, 88% vs. 75/87, 86%; adjusted risk ratio 1.02, 95% confidence interval 0.90 to 1.16). Adverse events were reported for three infants after surgery [bleeding (n = 1), salivary duct damage (n = 1), accidental cut to the tongue and salivary duct damage (n = 1)]. Cost-effectiveness could not be determined with the information available. Limitations: The statistical power of the analysis was extremely limited due to not achieving the target sample size and the high proportion of infants in the breastfeeding support arm who underwent frenotomy. Conclusions: This trial does not provide sufficient information to assess whether frenotomy in addition to breastfeeding support improves breastfeeding rates in infants diagnosed with tongue-tie. Future work: There is a clear lack of equipoise in the UK concerning the use of frenotomy, however, the effectiveness and cost-effectiveness of the procedure still need to be established. Other study designs will need to be considered to address this objective. Trial registration: This trial is registered as ISRCTN 10268851. Funding: This project was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment Programme (project number 16/143/01) and will be published in full in Health Technology Assessment; Vol. 27, No. 11. See the NIHR Journals Library website for further project information. The funder had no role in study design or data collection, analysis and interpretation. The views expressed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health and Social Care.

Many mothers and babies experience difficulties in establishing breastfeeding. In some babies it is thought that their difficulties may be linked to a condition called tongue-tie, in which a piece of skin tightly joins the middle part of the underside of the tongue to the base of the baby's mouth. This can be treated by an operation to divide the tight part/skin in the middle of the underneath of the tongue. We planned to carry out a trial of 870 babies to find out whether an operation together with breastfeeding support helps more mothers and babies with tongue-tie to continue breastfeeding until the baby is 3 months old compared to breastfeeding support on its own and whether the costs were different between the two groups of mothers and babies. We were only able to recruit 169 babies as the trial was stopped because of slow recruitment, changes to services in the COVID-19 pandemic and a high proportion of the babies in the breastfeeding support group going on to have an operation. There were no differences in the rate of breastfeeding at 3 months between the babies in the group who had an operation straightaway and those in the group that had breastfeeding support alone, or had an operation later. More than four in every five babies in both groups were still breastmilk feeding at 3 months. Three babies who had an operation, around 1 in 50 babies, had a complication of the operation (bleeding, scarring or a cut to the tube that makes saliva). Because of the small size of the study, we cannot say whether an operation to divide a tongue-tie along with breastfeeding support helps babies with tongue-tie and breastfeeding difficulties or has different costs. We will need to try different types of research to answer the question.

Orofacial-Myofunctional therapy after lingual frenectomy in patient with tongue-tie: a systemic postural approach with mezieres method and postural bench.

AIM: In this study we present the new postoperative oro-myofunctional protocol following frenectomy by atmospheric plasma associated with a systemic postural approach, which determines functional recovery through body symmetry restoration based on the concepts of the French "Mezieres Method" and postural bench. METHODS: A total of 130 patients (76 female, 54 male) affected by ankyloglossia of class III/IV, according to Kotlow's Tongue Tie Classification have been treated with atmospheric plasma followed by oro-myofunctional therapy associated with a systemic postural approach. The overall change, improved/worsened speech, feeding, or sleep has been evaluated through the 10 points Parents Speech Satisfaction (PSS) Score after one week and two months. CONCLUSION: Based on the findings of the current investigation, myofunctional therapy in tongue-tie results in a consistent significant functional ameliorating of feeding capability, speech and sleeping of subject treated as reported with traditional oro-myofunctional therapy.

Ankyloglossia: Typical Characteristics, Effects on Function, and Clinical Implications.

The purpose of this article is to (1) define the diagnostic characteristics of ankyloglossia, (2) identify potential problems associated with ankyloglossia, and (3) discuss treatment options, when treatment is appropriate. This article is based on a review of the literature, including recent systematic reviews, and the author's experience as a cleft and orofacial specialist. Ankyloglossia is a common congenital condition characterized by an anterior attachment of the lingual frenulum on the tongue. This causes difficulty elevating and/or protruding the tongue tip. As such, ankyloglossia has been thought to affect neonatal feeding, speech, and other functions. Although systematic reviews have concluded that most infants with ankyloglossia can be fed normally, a small percentage of affected infants will show improved efficiency of feeding post-frenotomy. They also concluded that frenotomy may relieve nipple pain in the breastfeeding mothers of affected infants. Regarding speech, the systematic reviews concluded that there is no evidence that ankyloglossia causes speech disorders. This may be because simple compensations will result in normal acoustics of the sounds. Therefore, frenotomy should be recommended sparingly for newborn infants, and it should rarely, if ever, be recommended for speech disorders.

Ranula Formation After Lingual Frenectomy in a Newborn.

Ankyloglossia is a developmental anomaly characterized by a shortened lingual frenulum, which can range from cases with no clinical relevance to severe cases, where frenectomy may be indicated. In newborns, this indication has increased exponentially. The purpose of this report is to discuss obstruction of the sublingual caruncle, with a recurrent ranula, as a post-surgical complication of laser lingual frenectomy performed in a newborn. The treatment provided is also discussed (marsupialization and removal of the affected sublingual glands). Lingual frenectomy should be performed with great care, especially in newborns, because the sublingual caruncles can be affected and complications can occur.