Familial Presentation of Ankyloglossia and Localized Periodontitis.

An association between ankyloglossia and periodontitis has not previously been reported. This case series describes three sisters who each had ankyloglossia and a molar/incisor pattern of localized periodontitis. The concurrent presentation of both conditions within the family suggests that further investigation of genetic factors that might concurrently affect the pathogenesis of both disorders may be warranted.

Geometric model to predict improvement after lingual frenulectomy for ankyloglossia.

OBJECTIVES: Frenulectomy for ankyloglossia is an intervention that often improves breastfeeding quality for both the mother and infant. Current classification systems assess and identify patients with ankyloglossia, but they do not predict the degree of improvement after lingual frenulectomy. We propose an idealized geometric model to quantify the potential effect of frenulectomy for ankyloglossia. METHODS: Our geometric model depicts the intact lingual frenulum as a triangular pyramid of mucosa on the floor of mouth. After incising one edge of the pyramid, as is performed during a frenulectomy, the structure unfolds to a two-dimensional diamond whose dimensions can be calculated. Utilizing this calculation, we can predict percent improvement in tongue extension after frenulectomy based off the original dimensions of the pyramid. RESULTS: Our multivariable equation that allows for the calculation of the percent increase in tongue extension is based on the frenulum thickness, frenulum length, tongue length, and insertion point of the frenulum on the tongue. The initial height of the frenulum and the proximity of the frenulum insertion to the tip of the tongue had the largest impact on tongue extension, whereas frenulum width had the smallest impact. CONCLUSION: Lingual frenulectomy has subjectively been reported to improve lingual tongue movement. Our mathematical model identifies multiple anatomic variables that lead to an increase in tongue extension after frenulectomy. Our model is the first step in supporting this subjective improvement with quantifiable measurements, and can allow for future validation studies.

Characteristics and considerations for children with ankyloglossia undergoing frenulectomy for dysphagia and aspiration.

OBJECTIVE: The purpose of this study is to analyze the basic demographics of patients who underwent frenulectomy at our institution as well as additional considerations regarding age, location of procedure, and possible effects on aspiration. METHODS: A retrospective chart review was performed based on CPT codes for frenulectomy and basic demographic data was collected. Other information such as presenting symptoms, type of ankyloglossia, location of the procedure, and modified barium swallow study (MBSS) information were also obtained. RESULTS: A total of 226 (66.4% male) patients underwent frenulectomy in the study time frame. Younger patients underwent frenulectomy for feeding symptoms (average age 6.5 months) and older children typically presented with speech related symptoms (average age 3.8 years). Of patients who had MBSS before and after the procedure, 5/11 (43%) had improvement of their aspiration after frenulectomy. CONCLUSIONS: Symptomatic ankyloglossia is more common in boys. Two age groups typically present for frenulectomy, infants for feeding difficulties and toddlers/preschoolers for speech related difficulties. Children with aspiration may benefit from frenulectomy, though aspiration is unlikely to resolve if other comorbidities are present. Proper evaluation and documentation of anatomy and functional tongue movement is important for future studies and decision-making regarding frenulectomy.

Anomalies of the oral cavity in newborns.

Examination of the oral cavity should be an essential part of the newborn assessment. Early detection of congenital disorders is essential to begin appropriate medical or surgical therapy and to prevent complications that could profoundly affect a child's life. The present review aims to describe the main anomalies of the oral cavity in infants and provide images in order to help the physician in current clinical practice.

The development and evaluation of a picture tongue assessment tool for tongue-tie in breastfed babies (TABBY).

Background: The presence of a tongue-tie (ankyloglossia) in an infant may lead to breastfeeding difficulties, but debate continues about which babies should be treated with frenotomy. The Bristol Tongue Assessment Tool (BTAT), a clear and simple evaluation of the severity of tongue-tie, is being used worldwide and translated into different languages. We aimed to produce a simple picture version of the BTAT to aid and enhance consistent assessment of infants with tongue-tie. Methods: The Tongue-tie and Breastfed Babies (TABBY) assessment tool was developed from the BTAT by a graphic designer, with iterative discussion with four practicing NHS midwives. The TABBY tool consists of 12 images demonstrating appearance of the infant tongue, its attachment to the gum and the limits of tongue mobility. The TABBY tool is scored from 0 to a maximum of 8.Two initial audits of the TABBY were undertaken at a large maternity unit in a secondary care NHS Trust, in Bristol UK from 2017 to 2019. TABBY was evaluated by five midwives on 262 babies with tongue-ties and experiencing breastfeeding difficulties who were referred for assessment to a tongue-tie assessment clinic using both BTAT and TABBY. Each pair of scores was recorded by one midwife at a time. A further training audit with 37 babies involved different assessors using BTAT and TABBY on each baby. Results: All midwives found the TABBY easy to use, and both audits showed 97.7% agreement between the scores. We suggest that a score of 8 indicates normal tongue function; 6 or 7 is considered as borderline and 5 or below suggests an impairment of tongue function. Selection of infants for frenotomy required an additional breastfeeding assessment, but all infants with a score of 4 or less in the audits had a frenotomy, following parental consent. Conclusions: The TABBY Assessment Tool is a simple addition to the assessment of tongue-tie in infants and can provide an objective score of tongue-tie severity. Together with a structured breastfeeding assessment it can inform selection of infants for frenotomy. It can be used by clinical staff following a short training and will facilitate translation into other languages.

Defining the anatomy of the neonatal lingual frenulum.

The lingual frenulum is recognized as having the potential to limit tongue mobility, which may lead to difficulties with breastfeeding in some infants. There is extensive variation between individuals in the appearance of the lingual frenulum but an ambiguous relationship between frenulum appearance and functional limitation. An increasing number of infants are being diagnosed with ankyloglossia, with growing uncertainty regarding what can be considered "normal" lingual frenulum anatomy. In this study, microdissection of four fresh tissue premature infant cadavers shows that the lingual frenulum is a dynamic, layered structure formed by oral mucosa and the underlying floor of mouth fascia, which is mobilized into a midline fold with tongue elevation and/or retraction. Genioglossus is suspended from the floor of mouth fascia, and in some individuals can be drawn up into the fold of the frenulum. Branches of the lingual nerve are located superficially on the ventral surface of the tongue, immediately beneath the fascia, making them vulnerable to injury during frenotomy procedures. This research challenges the longstanding belief that the lingual frenulum is a midline structure formed by a submucosal "band" or "string" and confirms that the neonatal lingual frenulum structure replicates that recently described in the adult. This article provides an anatomical construct for understanding and describing variability in lingual frenulum morphology and lays the foundation for future research to assess the impact of specific anatomic variants of lingual frenulum morphology on tongue mobility. Clin. Anat. 32:824-835, 2019. © 2019 The Authors. Clinical Anatomy published by Wiley Periodicals, Inc. on behalf of American Association of Clinical Anatomists.

Acute presentation of an intraoral dermoid cyst causing airway compromise in a young child.

A 4-month-old boy presented with a cystic swelling at the floor of the mouth causing acute airway compromise. The only previous history of note, was a tongue tie release at 3 days old. CT scan suggested a dermoid cyst with extensive floor of mouth abscess. He had an excision of the cyst and drainage of the superimposed abscess and made a good recovery. The histology report revealed a dermoid cyst which is a rare diagnosis in a child, particularly within the oral cavity. Early treatment is required to remove these lesions especially when they cause airway compromise or swallowing difficulties. This is the first case to our knowledge which suggests tongue tie release procedures causes a predisposition to the development of dermoid cysts in the oral cavity.

What is a tongue tie? Defining the anatomy of the in-situ lingual frenulum.

Surgical release of the lingual frenulum (frenotomy) has become an increasingly common procedure, performed from birth through to adulthood. Surprisingly, detailed anatomy of the in-situ lingual frenulum has never been described, and no anatomical basis has been proposed for the individual variability in frenulum morphology. The lingual frenulum is frequently referred to as a "cord" or "submucosal band" of connective tissue, yet there is no evidence to support this anatomical construct. This paper aims to describe the anatomy of the in-situ lingual frenulum and its relationship to floor of mouth structures. Fresh tissue microdissection of the lingual frenulum and floor of mouth was performed on nine adult cadavers with photo-documentation and description of findings. The lingual frenulum is a dynamic structure, formed by a midline fold in a layer of fascia that inserts around the inner arc of the mandible, forming a diaphragm-like structure across the floor of mouth. This fascia is located immediately beneath the oral mucosa, fusing centrally with the connective tissue on the tongue's ventral surface. The sublingual glands and submandibular ducts are enveloped by the fascial layer and anterior genioglossus fibers are suspended beneath it. Lingual nerve branches are located superficially on the ventral surface of the tongue, immediately deep to the fascia. The lingual frenulum is not a discrete midline structure. It is formed by dynamic elevation of a midline fold in the floor of mouth fascia. With this study, the clinical concept of ankyloglossia and its surgical management warrant revision. Clin. Anat. 32:749-761, 2019. © 2019 The Authors. Clinical Anatomy published by Wiley Periodicals, Inc. on behalf of American Association of Clinical Anatomists.

Is the Neonatal Tongue Screening Test a valid and reliable tool for detecting ankyloglossia in newborns?

BACKGROUND: Although there is a lack of strong evidence for the association between ankyloglossia in newborns and impaired breastfeeding, screening for ankyloglossia using the Neonatal Tongue Screening Test (NTST) is mandated by law in Brazilian maternities. AIM: To assess the reliability and validity of the NTST. DESIGN: cohort study; baseline sample comprised 268 mother-newborn dyads. At follow-up, 169 mothers were contacted by telephone. Interviews with the mothers for data collection were performed up to 48 h and at 1-3 months after childbirth. Trained and calibrated personnel performed the oral examinations of the newborns. Thirty newborns were examined for inter-reproducibility assessment. RESULTS: Of the 268 newborns included, 212 had a lingual frenulum that could be visually inspected and their NTST scores ranged from zero to nine (mean = 2.0, ±2.0). Interexaminer reproducibility was acceptable (Intraclass correlation coefficient = 0.77). Internal consistency of the NTST was poor (Cronbach's alpha = 0.28). Construct validity was investigated through the association between NTST scores and difficulties in breastfeeding at baseline and follow-up, and infants' weight gain at follow-up (mean age 32 ± 6.7 days). No statistically significant associations were found. CONCLUSION: NTST is neither reliable nor valid for detecting ankyloglossia that may interfere with breastfeeding in newborns.

Familial autosomal dominant severe ankyloglossia with tooth abnormalities.

Ankyloglossia is a congenital oral anomaly characterized by the presence of a hypertrophic and short lingual frenulum. Mutations in the gene encoding the transcription factor TBX22 have been involved in isolated ankyloglossia and X-linked cleft palate. The knockout of Lgr5 in mice results in ankyloglossia. Here, we report a five-generation family including patients with severe ankyloglossia and missing lower central incisors. Two members of this family also exhibited congenital anorectal malformations. In this report, male-to-male transmission was in favor of an autosomal dominant inheritance, which allowed us to exclude the X-linked TBX22 gene. Linkage analysis using short tandem repeat markers located in the vicinity of LGR5 excluded this gene as a potential candidate. These results indicate genetic heterogeneity for ankyloglossia. Further investigations with additional families are required in order to identify novel candidate genes.

A influência da anquiloglossia no crescimento e desenvolvimento do sistema estomatognático / Ankyloglossia and its influence on growth and development of the stomatognathic system

RESUMO Objetivo: Avaliar criticamente os artigos existentes na literatura brasileira e estrangeira acerca da influência do frênulo lingual encurtado sobre o crescimento e desenvolvimento do sistema estomatognático, bem como sobre a conquista do equilíbrio forma-função. Fontes de dados: A busca eletrônica nas bases MEDLINE/PubMed, Google Scholar, LILACS, SciELO e ScienceDirect com os descritores "freio lingual" e "desenvolvimento" e seus equivalentes em inglês resultou inicialmente em 51 resultados de trabalhos publicados entre janeiro de 1997 e a presente data. Após aplicados os critérios de exclusão, 14 artigos de estudos clínicos foram selecionados para a leitura completa. Síntese dos dados: A revisão integrativa da literatura ajudou a confirmar a proposição de que algumas maloclusões estão intimamente relacionadas à anquiloglossia. Ainda que a quantidade de estudos clínicos publicados até a atualidade seja pequena, há um consenso entre os autores acerca do efeito negativo de desequilíbrios funcionais sobre o crescimento e o desenvolvimento corretos do sistema estomatognático, tanto que a metade dos estudos pesquisados relata que intervenções cirúrgicas para a liberação do frênulo lingual são seguras e eficazes no que diz respeito à melhora na amamentação. Ainda, dos 14 estudos incluídos nesta revisão, 4 reportam influências negativas da anquiloglossia sobre a musculatura orofacial. Conclusões: Há um consenso entre os autores acerca dos efeitos negativos das alterações anatômico-funcionais do frênulo lingual sobre o crescimento e o desenvolvimento craniofacial, ainda que a opinião sobre a intervenção cirúrgica precoce não seja unânime.

ABSTRACT Objective: To critically examine the existing Brazilian and International scientific literature regarding the influence of short lingual frenulum over growth and development of the stomatognathic system, as well as how it impacts the achievement of the shape-function balance. Data sources: An electronic literature search was conducted in databases, including MEDLINE/PubMed, Google Scholar, LILACS, SciELO, and ScienceDirect, using the key words "lingual frenum" and "development", as well as their equivalents in Brazilian Portuguese. The literature search yielded 51 papers published between January 1997 and the present date; 14 articles of clinical trials were selected for meeting the inclusion criteria and were read in full. Data synthesis: The integrated literature review supported the proposition that some malocclusions are closely related to the presence of ankyloglossia and, although very few clinical trials on this topic have been published so far, there is a consensus among authors concerning the negative effects of functional imbalances over the stomatognathic system's proper growth and development. Half of the studies found state that surgical interventions for releasing the lingual frenum are both safe and effective, concerning improvement in breastfeeding scores. Moreover, 4 out of the 14 studies included in this integrated review, report a negative influence of ankyloglossia over the orofacial muscular system. Conclusions: There is a consensus among the authors concerning the negative effects of lingual frenulum's anatomic and functional alterations over craniofacial growth and development. The opinion about the early surgical intervention, however, is not unanimous.