Labial ankyloglossia: A rare case report.

Tongue-tie or ankyloglossia is the congenital short thick lingual frenulum resulting in reduced mobility of the tongue. It limits the possibilities of the extension such as the protrusion and elevation of the tip of the tongue due to either the short of frenulum or genioglossus muscle or both. It can be observed at different ages with specific indications for treatment for each group and cause problems in the feeding, dental hygiene, speech, appearance, and self-esteem of affected patients. Although various degrees of the tongue-tie are mostly observed from the tip of tongue to the base of tongue, sometimes it may present unusually. The present article reports an unusual unique pattern of ankyloglossia where lingual frenum was exceptionally extended and merged with mandibular labium frenum. Reported case is one of the rarest types of ankyloglossia and emphasizes on its clinical implications, need for modifying diagnostic criteria, surgical management, and postoperative exercises.

Introduction

Ankyloglossia (partial) is defined as a limitation of the possibilities of the protrusion and elevation of the tip of the tongue due to either the shortness of the frenulum or the genioglossus muscles or both.[1] Ankyloglossia is a congenital origin with genetic etiology or environmental factors with a prevalence of about 5% and male:female ratio 2:1 was reported in early age.[23] Ankyloglossia can be observed at different ages with specific indications for treatment for each group. It has also been associated with problems in breastfeeding among neonates, dental caries, malocclusion, gingival recession, and restricted alveolar bone growth in growing children.

Mostly tongue-tie presents as partial ankyloglossia from tongue tip to the base of tongue with variable degrees classified by Kotlow’s[4] and classification of ankyloglossia based on the degree of limitation of lingual mobility due to the hypertrophic lingual frenulum.[5]

Case Report

An 8-year-old boy reported to the Department of Pedodontics and Preventive dentistry, Faculty of Dental Sciences, KGMC, Lucknow, with the chief complaint of difficulty in brushing the lower anterior teeth both labially and lingually, rotated in lower anteriors, and produced problems in fluent speech. Intraoral examination revealed the presence of tongue-tie. The lingual frenum was thick and fibrotic inserted close to the tip of the tongue. Anteriorly, it was continuous with the mandibular labial frenum along the floor of the mouth and between the mandibular central incisors [Figure 1]. The child was able to protrude his tongue just beyond the mandibular anterior teeth. The tongue showed clefting on the protrusion and typical heart shape on elevating it toward the palate. On pulling the lower lip outwardly, the tongue was inadvertently forced out due to pull exerted by the labial frenum. Similarly, on retruding the tongue into the floor of the mouth, the lower lip was strained inward. On forcibly separating the lip and the tongue away from each other, blanching was observed over the frenum. There was spacing between the central incisors and a distolingually rotated lower right central incisor. The patient suffered from malnutrition, poor oral hygiene, and low confidence levels.

Figure 1

Frenum attachment at the tip of the tongue and insertion into labial lip protracted tongue on outward lip movement

Frenectomy was done under local anesthesia. Using two hemostats, the lingual frenum was clamped and then clipped with a scalpel. The incision was deep enough to sever and remove the intrinsic muscle extension which was enmeshed into the frenum [Figure 2]. The labial frenectomy was done in continuation with lingual frenectomy [Figure 2]. Immediately, after surgery, tongue protrusion of 3 cm was seen. The site was closed with interrupted sutures using 3-0 black silk. The patient was prescribed analgesics, antibiotics, and mouthwash, instructed for regular tongue exercises for 3–5 min, once or twice daily, for 3–4 weeks, and maintaining proper oral hygiene. The healing was smooth, and at a 2-week follow-up, the patient showed improvement in tongue mobility. After 3 years follow-up, the patient showed normal speech and tongue movement [Figure 3].

Figure 2

Frenectomy of lingual frenum and labial frenum with removal of muscle extention

Figure 3

Follow-up of 3 years

Discussion

In the oral cavity, frenula are cords of tissue beneath the tongue and in the labial and buccal vestibules, which during the intrauterine development, apparently guide the growth of various structures of the oral apparatus. After birth, they are largely redundant although they seem to help in the positioning of primary teeth. Ankyloglossia may be the result of failure in cellular degeneration leading to a much longer anchor between the floor of the mouth and tongue.[6]

Ankyloglossia, though an innocuous condition, it does mar the normal function of the tongue. Although there is ambiguity regarding the definition or grading system to describe tongue-tie, Kotlow[4] has suggested a protocol for the diagnosis of the condition which is the most commonly acceptable classification.

In the present case, frenum attachment was observed at the tip of the tongue continuous with the mandibular labial frenum along the floor of the mouth and between the mandibular central incisors, attached to the lower lip. It is the rarest type of ankyloglossia yet to be documented. Thus, the Kotlow's classification must be modified to include it as class 5 type of ankyloglossia. The movement of tongue was carried out by movement of the floor of mouth. Speech problems as the tip of the tongue cannot rise high enough to articulate sounds such as “t, d, z, s, th, n, and l” or arch off the floor of the mouth while pronouncing “r.” Thus, the patient was not able to speech fluently and correctly. This promotes an infantile swallow and prevents its conversion into an adult-like swallow which can result in an open-bite deformity or mandibular prognathism due to the tongue contacts the anterior portion of the mandible with exaggerated anterior thrusts.[7]

Most children with ankyloglossia are asymptomatic,[3] even in severe form of ankyloglossia.

Severe ankyloglossia in early age may result in inadequate milk intake, prolonged feeding times, maternal nipple pain or bleeding, and rarely, failure to thrive.[8] Personal and social activities as licking ice cream, cleaning food off the teeth with the tongue, playing wind instruments, and kissing are impaired.[4]

Thus, depending on the extent of tongue movement and Hazelbaker scoring system to assess the severity of ankyloglossia,[9] the following treatments are planned. Class 1 and Class 2 cases are self-correcting; thus wait and watch along with speech therapy is indicated. Class 3 and Class 4 surgical interventions are desirable.

Delays in language acquisition after frenectomy have been noticed in some young patients, possibly due to habitual lack of practicing a more mature and structured language and an inherent tendency to converse using short phrases and telegraphic speech. The longer these maladaptations have lasted, more the patient will cling to them, and much motivation is required to boost progress toward developing proper oro-motor skills. The patient had developed a good and fluent speech as well as correct wording after speech therapy in 3 years follow-up period.

Conclusion

Optimal management of tongue-tie including well-timed and proper surgical intervention followed by proper tongue exercises and speech therapy as and when indicated holds the potential to deliver pleasing results in complete ankyloglossia.

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Conflicts of interest

There are no conflicts of interest.