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Ankyloglossia: does it matter?
Pediatr Clin N Am 50 (2003) 381 – 397
Ankyloglossia: does it matter? M. Lauren Lalakea, MDa,b, Anna H. Messner, MDa,c,* a
Division of Surgery/Otolaryngology/Head & Neck Surgery, Stanford University, Stanford, CA 94305, USA b Division of Otolaryngology/Head & Neck Surgery, Santa Clara Valley Medical Center, 751 S. Bascom Avenue, San Jose, CA 95128, USA c Lucile Salter Packard Children’s Hospital, Stanford Pediatric Otolaryngology, 725 Welch Road, Palo Alto, CA 94304-5654, USA
An author nearly 40 years ago aptly commented that ‘‘much entertaining nonsense has been written about tongue-tie’’ [1]. Tongue-tie (more formally known as ankyloglossia) is a congenital anomaly characterized by an abnormally short lingual frenulum, which may restrict mobility of the tongue tip. The clinical significance of this anomaly and the best method of management have been the subject of debate for some time [2]. Much of the controversy about management of ankyloglossia probably is related to the paucity of relevant scientific data demonstrating efficacy of intervention. To quote a prominent pediatrician (PD), ‘‘much of the information needed for making rational treatment decisions in cases of tongue-tie is lacking’’ [3]. In a recent edition of a popular pediatric textbook, only one sentence is devoted to the discussion of ankyloglossia [4]. The purpose of this article is to summarize fact, fiction, and areas of controversy about ankyloglossia, to create a greater awareness and understanding of this condition.
Historical context Historical references to tongue-tie may be found beginning in biblical times: ‘‘. . .and the string of his tongue was loosened and he spoke plain,’’ (Mark 7:35). In the eighteenth century, several references, as cited by Catlin and De Haan [5] and Marmet et al [6], recommended clipping the frenulum in tongue-tied infants to facilitate breast-feeding. Horton et al [7] reported that it was the habit of midwives in this period to use their fingernails to divide the lingual frenulum of all infants. Since that time, support for tongue-tie as a cause of feeding or speech problems * Corresponding author. Division of Surgery/Otolaryngology/Head & Neck Surgery, Stanford University, Stanford, CA 94305, USA. E-mail address: [email protected] (A.H. Messner). 0031-3955/03/$ – see front matter D 2003, Elsevier Inc. All rights reserved. doi:10.1016/S0031-3955(03)00029-4
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has waxed and waned. By the early part of the twentieth century, strenuous opposition was raised to the practice of frenotomy [6,7]. Moreover, by the 1940s, some authors were quite vehement in their opinion that ankyloglossia should never be considered causally related to speech difficulties of any type [8]. Current opinion ranges from the belief that tongue-tie only rarely interferes with feeding or speech, and generally requires no treatment [3,4]; to enthusiastic support for frenotomy in the lactation literature [9 –11]. Illustrating this diversity of opinion, a recent article reported the results of a survey of hundreds of US and Canadian PDs, otolaryngologists (OTOs), lactation consultants (LCs), and speech pathologists (SPs), and concluded that there was little consensus among and within these groups with regard to the significance of ankyloglossia or its management [2].
Incidence and natural history Ankyloglossia is uncommon, but not rare. Incidence figures reported in the literature vary widely, ranging from 0.02% to 4.8% [5,12 – 15]. Tongue-tie occurs more commonly in males—with a male-to-female ratio on the order of 3 to 1, and shows no racial predilection [13,14]. The variation in reported incidence may be attributed, in part, to the lack of a uniform definition and objective grading system for tongue-tie. In addition, some of the variation may reflect age-related differences in the presence of this anomaly. Contemporary studies conducted in well-baby nurseries have yielded incidence figures for neonates in the range of 1.7% to 4.8% [12 – 15]. Tongue-tie is thought to be less common in adults, although there are no exact figures in this regard [3,5]. Ankyloglossia occurs most frequently as an isolated anatomic variation. Increased prevalence has been noted in infants with a history of maternal cocaine abuse (incidence = 10.4%, odds ratio = 3.5) [16]. Ankyloglossia also may occur with increased frequency in association with various congenital syndromes, including Opitz syndrome, orodigitofacial syndrome, and in association with X-linked cleft palate [17 – 19]. There is little definitive information on the natural history of untreated ankyloglossia. Certain authors [1,7] have postulated (but not substantiated) that the short frenulum can elongate spontaneously due to progressive stretching and thinning of the frenulum with age and use. This explanation might account for the general perception that this anomaly is more frequently seen in young children, as compared with adults. There is, however, no prospective longitudinal data on the fate of the congenitally short lingual frenulum. This lack of scientific data complicates clinical decision making with regard to the need for treatment of ankyloglossia, if any, and its timing.
Potential manifestations Opinions range widely regarding the clinical significance of ankyloglossia. Some authors feel that ankyloglossia is only rarely symptomatic [3,4], whereas
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others believe it may lead to a host of problems, including infant feeding difficulties, speech disorders, and various mechanical and social issues related to the inability of the tongue to protrude sufficiently [6,7,20 – 22]. The ankyloglossia survey study mentioned above [2] found that as a group, PDs were less likely than their OTO, SP, and LC colleagues to believe that this anomaly was associated with symptoms of any kind. Ninety percent of PDs surveyed indicated that ankyloglossia is never or rarely a cause of feeding dysfunction, whereas 69% of LCs stated that ankyloglossia was frequently or always associated with feeding difficulty. Seventy-seven percent of PDs noted that ankyloglossia is never or rarely a cause of speech dysfunction, whereas only 40% of OTOs believed similarly. Lastly, 67% of OTOs as compared with 21% of PDs believed that tongue-tie is at least sometimes associated with a variety of social and mechanical issues. Although some of the above differences may reflect referral biases, it is clear that beliefs about the significance of ankyloglossia are quite divergent. Information that is relevant to the potential clinical manifestations of tongue-tie is outlined below. Effect on feeding With the increasing popularity of breast-feeding in the past 2 decades, there has been a resurgence of interest in ankyloglossia as a cause of breast-feeding problems [6,9]. Although much of the current literature relating to this topic is in the form of case reports and small uncontrolled case series [6,11,23,24], many LCs and some physicians believe that ankyloglossia can make breast-feeding difficult for at least some infants with this condition [2,6,25]. Multiparous mothers who have breast-fed their unaffected infant(s) successfully in the past note an obvious difference when nursing a subsequent newborn with tongue-tie. Moreover, mothers of affected infants frequently report a marked improvement in breast-feeding after tongue-tie release. Problems reported previously include sore nipples, poor latching and sucking mechanics, poor infant weight gain, and early weaning. A recently published prospective study of infants with ankyloglossia has shed some light on this controversy [14]. Mothers of 36 infants with ankyloglossia noted at birth and mothers of a matched control group of unaffected newborns were followed for up to 6 months to determine the incidence of breast-feeding difficulties. There was no significant difference in the percentage of mothers with affected infants as compared with mothers in the control group who were able to successfully breast-feed for at least 2 months (83% and 92%, respectively). Breastfeeding problems, defined as nipple pain lasting longer than 6 weeks or infant difficulty latching onto the breast, occurred significantly more frequently in the ankyloglossia group, however (25%, versus 3% for controls). This study concluded that ankyloglossia may adversely affect breast-feeding in selected infants. There is general agreement that infants with ankyloglossia do not have trouble with bottle-feeding, nor with handling solid foods once they are introduced. Bottle-feeding, however, should not be proposed as a solution for ankyloglossiarelated problems in a mother who otherwise desires to breast-feed. The role of frenotomy in this situation is discussed later in the article.
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Effect on speech The effect of ankyloglossia on speech has not been defined clearly. Although speech problems historically have been viewed as the hallmark manifestation of symptomatic tongue-tie, particularly among the lay public, others in the literature have vociferously denied that any such relationship exists [8]. Current opinion among speech therapists is evenly divided; 49% of those responding to a survey on this topic believed that ankyloglossia was never or rarely associated with speech problems, and an equal number believed that it was sometimes or frequently associated with speech problems [2]. Anecdotal evidence indicates that some children with ankyloglossia develop normal speech, and compensate for limited tongue-tip mobility without surgical repair or need for speech therapy [1,7,21]. In at least some individuals, however, tongue-tie contributes to articulation errors or difficulty with the rate and range of articulation, thereby decreasing speech intelligibility [1,7,20,21,26]. Speech sounds that may be affected by impaired tongue-tip mobility include lingual sounds and sibilants such as T, D, Z, S, TH, N, and L [1,5,27]. The compensatory techniques used by children with ankyloglossia typically include restricted mouth opening while speaking, and alternate tongue placement for sounds requiring tongue-tip elevation. An excellent study by Fletcher and Meldrum [20] provided strong evidence regarding the relationship of tongue mobility to speech articulation. In this study, normal children, 11 to 12 years of age, underwent careful intraoral measurements of the relative lengths of the tongue and frenulum, to determine the ratio of ‘‘free’’ tongue to total tongue length. Participants were separated into ‘‘limited lingual freedom’’ and ‘‘greater lingual freedom’’ groups on this basis. These investigators found a highly significant increase in the number of articulation errors in the limited lingual freedom group as compared with the greater lingual freedom group, and concluded that these findings might be particularly pertinent to the entity of ankyloglossia. When present, the severity of the articulation problems in affected individuals may vary; problems may be so pronounced as to be evident at the single word level, or be mild enough so as to be noticeable only in connected speech. In our experience, up to one half of young children with ankyloglossia referred for otolaryngology evaluation will have articulation difficulties that may be detected in the context of a formal speech pathology consultation [26]. In addition, among adults with ankyloglossia, even those with grossly normal speech still may complain of speech problems, believing that their speech is more effortful than that of others. It is important to keep in mind that ankyloglossia is not a cause of speech delay. Children with ankyloglossia are expected to acquire speech and language at a normal rate, although some may experience articulation difficulties for certain speech sounds, as indicated above. Occasionally, parents and others who care for a child with speech delay may erroneously ascribe the delay to tonguetie, and demand surgical intervention in the hope that normal speech and
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language will result. In such a patient, potential causes of speech delay should be sought carefully, and the patient should be directed for other evaluations as appropriate (eg, audiologic, speech/language, or neurodevelopmental assessments). Surgical repair may be considered at a later time, once the child has been evaluated fully, and other issues have been addressed. Mechanical and social effects The potential mechanical and social consequences of ankyloglossia have received little attention in the medical literature. Many clinicians are unaware that tongue-tie may have consequences beyond those of speech and feeding difficulties [2]. Mechanical problems related to tongue-tie may include difficulty with intraoral toilet (licking the lips and sweeping the teeth free of food debris), local discomfort or cuts beneath the tongue, dental issues such as a diastasis between the lower central incisors due to pressure from a tight frenulum, and difficulty wearing dentures later in life due to poor fit. Other issues that may be associated with limited lingual range of motion may include decreased facility in playing a wind instrument, difficulty licking an ice cream cone, and difficulty with ‘‘french kissing’’ [1,21,28,29]. In addition, as might be expected, these types of symptoms may be accompanied by a sense of social embarrassment, due to teasing and ridicule from peers [30,31]. In our experience, approximately 50% of older children and adults with persistent ankyloglossia report one or several of the above complaints when their presence is sought in the course of obtaining a complete problem-directed history. These mechanical limitations and social issues may occur even in the absence of other ankyloglossia-related complaints (ie, without a history of feeding or speech problems). It is important to note that mechanical and social concerns may not manifest until later in childhood. Younger children indeed may have symptoms, but may not recognize or be able to report them. In addition, complaints related to activities such as playing a wind instrument and kissing, and issues relating to teasing and ridicule from peers, may not become apparent or relevant until later childhood and beyond. Finally, owing to a sense of embarrassment regarding their condition, patients may refrain from expressing or volunteering their concerns unless questioned directly.
Diagnosis Although the diagnosis of ankyloglossia may be suggested by history, it is confirmed by characteristic physical examination findings. The frenulum may be thick and fibrous or thin and membranous, and is abnormally short, inserting at or near the tongue tip (Figs. 1, 2). The tongue may have a notched or heart shape on protrusion, due to tethering by the frenulum (Fig. 3). Protrusion is limited, and in some cases, may fail to extend past the lower lip. The tongue may appear to roll or curl with attempted protrusion as the midportion of the tongue moves forward,
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Fig. 1. One-day-old male with mild ankyloglossia and difficulty latching onto the breast.
while the tip itself is drawn inferiorly by the frenulum with little forward extension. Affected individuals also characteristically will have difficulty lifting or elevating the tip of the tongue toward the upper dentition and upper lip. Patients may be completely unable to lift the tongue, or in those cases when they are able to elevate the tip to some degree, there is often dimpling noted behind the tongue tip due to tethering. Side-to-side motion of the tongue may be impaired as well. A space or diastasis between the lower teeth is occasionally present, due to the repeated thrusting action of the frenulum through the teeth with attempted tongue protrusion (Fig. 4).
Fig. 2. Preschooler with moderate ankyloglossia and articulation problems.
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Fig. 3. Heart-shaped tongue on protrusion.
A variety of schemas have been proposed to verify the presence of ankyloglossia and grade its severity. Hazelbaker [32], an LC, devised an ‘‘Assessment Tool for Lingual Frenulum Function’’ to be used with neonates. This tool evaluates seven tongue movements, including lateralization, lift, extension, spread of anterior tongue, cupping, peristalsis, and snap-back. Each movement is graded on a 0 to 2 scale; if an infant scores poorly, frenotomy is recommended.
Fig. 4. Four-year-old male with diastasis between his lower central incisors due to ankyloglossia.
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Kotlow [28] used a measurement termed ‘‘free’’ tongue (distance in mm from tongue tip to the insertion of the frenulum) to divide tongue-tie into four classes: mild, moderate, severe, and complete. Unfortunately, this classification scheme failed to provide any documentation regarding the correlation of ankyloglossia class to clinical symptoms. Others [20,33] have used more detailed ratios of lingual dimensions to assess tongue-tie in the older child, and have documented a relationship between these measures and speech articulation. Owing to lack of cooperation and difficulties measuring this mobile muscle, however, these investigators resorted to the use of general anesthesia to accomplish this assessment in young children [20,33]. Williams and Waldron [27] proposed an alternate method, designed to measure lingual function, rather than a physical dimension. These authors recommended measuring the maximum ability of the patient to elevate the tongue, by asking the patient to touch the tip of his or her tongue to the upper teeth, and then open the mouth as widely as possible while maintaining contact. While the patient holds this position, the distance between the upper and lower central teeth, or interincisal distance, is recorded in millimeters (Fig. 5). The interincisal distance is thus used as an objective measure of tongue elevation ability; unfortunately, these authors [27] did not obtain normative data nor did they document clinical correlation. Despite the varied attempts listed above to quantify ankyloglossia severity, at present there is no way to predict—based on examination findings—which children are likely to have, or to develop, speech or mechanical symptoms related to their ankyloglossia. Ability to protrude the tongue past the lower lip has been used by some in the past as a quick rule of thumb to predict which patients will not require surgical repair [1]. In our personal experience with treating children with ankyloglossia, however, we have found that those with an associated articulation disorder (as documented on formal speech pathology assessment) will often be able to protrude the tongue 15 mm or more beyond the lower dentition [26].
Fig. 5. Interincisal distance. The tongue is placed at the back posterior edge of the upper teeth and the mouth is maximally opened. Measurement (in millimeters) is taken between the teeth. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:95; with permission.)
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In our practice, we measure lingual mobility in children who are able to cooperate for protrusion (measured in millimeters of tip extension past the lower dentition) (Fig. 6), and elevation (as measured by recording interincisal distance with the tongue tip maximally elevated and in contact with the upper teeth). Although these measurements may not predict necessarily the presence or absence of symptoms, they provide objective documentation of tongue mobility and help to define the degree of restriction. We have found that protrusion and elevation values are typically in the range of 15 mm or less in children with ankyloglossia, and 20 to 25 mm or greater in normal children [26]. Moreover, mobility measurements are a valuable tool in documenting change, particularly preoperatively and postoperatively.
Indications for intervention History and physical examination generally are sufficient to confirm the presence of ankyloglossia, and supplemental evaluations are not required to establish whether a child may be considered a candidate for surgery. When the presenting problem relates to breast-feeding difficulty, consultation with a lactation specialist
Fig. 6. Tongue protrusion. Measurement (in millimeters) taken from the lower incisors to the tongue tip. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:94; with permission.)
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is an option—particularly if the degree of restriction appears quite mild—to rule out other contributing factors. When the patient’s primary complaint is speech difficulty, a formal speech evaluation may be helpful if the relationship of the ankyloglossia to the speech disorder is in doubt. A failed trial of speech therapy is not a requirement before surgical repair is considered. Patients with ankyloglossia with obvious or definite indications for surgery include infants with associated breast-feeding difficulties, young children with characteristic articulation problems, and older children and adults adversely affected by the mechanical and social manifestations of this condition. There are other relative indications (discussed below); however, the timing of intervention and the benefit of surgery in these alternate clinical scenarios are more controversial. The optimal timing for surgery has not been determined. Some investigators have advocated surgical intervention prior to the development of speech difficulties [22,25], whereas others have mandated withholding surgery until age 4 or more, and then offering surgery only to those with a manifest speech problem [1,3,7]. Current opinion is no less varied; in a recent survey regarding ankyloglossia treatment, 34% of OTOs believed that surgery was inappropriate in children less than 1 year of age, 36% believed surgery could be performed at any age, and 42% believed surgery to be inappropriate (and perhaps less effective) for those 12 years of age or older [2]. In our view, given the minor nature of the surgery and the significant potential for speech difficulties and later social and mechanical problems, it may be appropriate to consider surgery for those children with significant ankyloglossia at any age, including infants and toddlers who have yet to demonstrate overt symptoms. When very young children are referred for evaluation, we inform parents that there is no way to predict which children will develop symptoms related to their condition, and which children may remain asymptomatic or later outgrow their condition. We do, however, present information regarding the potential early and late consequences of ankyloglossia and solicit parental opinion with respect to treatment preferences. Although early intervention in all children before the development of speech problems may be unwarranted, delaying intervention until obvious difficulties emerge may commit some children unnecessarily to a period of rehabilitative speech therapy or social embarrassment. One additional consideration when evaluating an infant with ankyloglossia is that up to several months of age, a frenotomy can be performed quickly in the clinic without requiring general anesthesia. In contrast, if surgery is deferred until the child is older than 1 year of age, general anesthesia in the operating room usually is required. Cooperative children older than 6 to 7 years of age usually can undergo repair under local anesthesia, but deferring surgery until this age can subject a child to an unnecessarily long period of symptoms or social concerns. Therefore, our practice is to discuss these issues with parents, who may elect early intervention at their discretion, or may prefer to wait and elect surgery only if and when symptoms occur. There are no absolute contraindications to surgery; however, as noted earlier, children with apparent speech delay should be approached with caution, and
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surgery should be deferred until the child has been evaluated and treated by other specialists as appropriate.
Alternatives to surgery Alternatives to surgery include observation or a trial of speech therapy. The wait-and-see approach may be considered for the young child with minimal symptoms, because it is possible that the child will outgrow the ankyloglossia, or compensate sufficiently so that future clinically significant speech or mechanical problems will not occur. In the child with speech difficulties, speech therapy may correct articulation errors successfully in some cases, although SPs vary in their degree of optimism for this approach [2]. One disadvantage of speech therapy is that it may require a lengthy period during which time the child may be self-conscious about his or her impairment. The benefit of speech therapy must be weighed against surgical treatment that, although invasive, is simple, quick, and effective. Another disadvantage is that although speech therapy may allow the child to improve his or her articulation over time by using various compensatory mechanisms, it may still leave him or her vulnerable to later manifesting mechanical problems associated with restricted lingual mobility.
Surgical procedures Frenotomy, or simple release of the frenulum, and frenuloplasty (release with plastic repair) are the two most commonly used surgical procedures in the treatment of ankyloglossia. As noted above, appropriate patient selection and ideal age for intervention have been topics of much disagreement. Past reports of complications and the lack of clearly agreed-on measures of success have contributed to the debate. Frenotomy Frenotomy (Fig. 7) also has been termed ‘‘clipping’’ of the frenulum, and is most appropriate for the treatment of ankyloglossia in infants (eg, for breastfeeding problems) because it is rapid and relatively easy to accomplish. The procedure may be performed at the bedside in the newborn nursery, or in the office with local or no anesthesia. Practitioners who typically perform frenotomy include OTOs, dentists, and PDs. Interestingly, 22% of a group of 425 US and Canadian PDs who responded to a survey for a recent paper on this topic indicated that they had performed frenotomy, although only 10% reported that they had been taught this skill in residency [2]. Although some practitioners use local anesthesia, in our experience, the discomfort associated with the release of thin and membranous frenula is brief and quite minor. The infant is positioned in front of the clinician with his or her
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Fig. 7. Frenotomy. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:95; with permission.)
head directed toward the clinician’s left (if the clinician is right handed). Two gloved fingers of the clinician’s left hand are placed below the tongue on either side of midline, to retract it upward toward the palate, exposing the frenulum. Small sterile scissors are used to release the frenulum, beginning at its free border and proceeding posteriorly directly adjacent to the tongue to avoid injury to the (more inferiorly placed) submandibular ducts in the floor of mouth. Occasionally, complete release may be accomplished with a single scissor cut, but more frequently, especially when the frenulum is quite tight, two or three sequential cuts are required; each cut provides some release, allowing improved retraction and visualization for subsequent cuts, if necessary. Bleeding is generally slight, although the amount of blood may appear greater due to mixing with saliva. If needed, bleeding can be controlled easily with a brief period of pressure applied with a 2 2-inch gauze. The incision is not sutured, and the infant is allowed to feed immediately following the procedure. Acetaminophen may be used for pain control, but often is not required. Antibiotic therapy is unnecessary. The patient may be seen in follow-up in 1 to 2 weeks as needed; generally by that time the incision has healed completely. Once the practitioner has sufficient experience with the procedure, telephone follow-up may be appropriate as long as the patient is doing well. Frenuloplasty Frenuloplasty (Fig. 8) is the preferred procedure for most patients greater than 1 to 2 years of age, because it allows for more complete release of the tongue-tie. In addition, because a plastic closure is performed, some surgeons believe that the chance of scarring and recurrent ankyloglossia is reduced. In young children, the
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Fig. 8 A-C. Horizontal to vertical frenuloplasty. (A) the incision line; (B) the resulting defect; and (C ) the closure line. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:96; with permission.)
procedure is performed using a brief general anesthetic, whereas in older children and adults, it can be accomplished easily in the clinic using a local anesthetic. The frenulum is released in a manner identical to that used for frenotomy. Occasionally, limited division of the genioglossus muscle may be required to effect adequate release. The resulting wound is closed with sutures. Some surgeons use a z-plasty flap closure, and others prefer to close the diamond-shaped defect created by the initial horizontally oriented release in a vertical manner (hori-
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zontal-to-vertical plasty). Both methods cover the exposed raw surface and result in additional lengthening of the frenulum. Frenuloplasty is only slightly more involved than is simple frenotomy, and requires only minutes to perform. Bleeding is generally minimal, and is controlled with pressure or cautery. Patients are advised to resume a normal diet as tolerated. As a rule, patients report little in the way of postoperative discomfort, and may use acetaminophen as needed for pain. Antibiotics are not required. When patients are old enough to comply, we ask them to perform a series of tongue exercises several times daily for the first 4 to 6 weeks postoperatively, with the expectation that this practice may enhance mobility, assist in retraining of tongue musculature, and reduce the potential for scarring.
Surgical outcome The results of frenotomy and frenuloplasty for ankyloglossia are highly favorable, provided that surgical indications are met and patients are appropriately selected for intervention. In children and adults, tongue mobility measures predictably improve in the first 1 to 3 months following surgery, and tend to remain stable after that time. Postoperatively, patients may be expected to gain 10 mm or more of tongue protrusion and tongue elevation on average, as compared with preoperative measures of mobility [26]. In addition, significant subjective gains may be anticipated as well; a majority of parents/patients report that they are highly satisfied with these procedures. Frenotomy has been reported to result in immediate improvement in problems related to breast-feeding in a majority of cases [6,9,30]. There are multiple anecdotal reports in the lactation literature describing rapid resolution of maternal nipple pain, better latching, and enhanced infant weight gain [10,11,23,24]. In our practice, it is not uncommon for mothers to note a marked difference in nursing mechanics as early as the first feeding after frenotomy. Frenotomy is not a panacea for all breast-feeding problems, however; when the degree of ankyloglossia is minimal, or there is doubt as to the contribution of tongue-tie to nursing problems, it may be wise to obtain a lactation consultation or to counsel the mother appropriately regarding expectations before proceeding with frenotomy. Speech results following frenuloplasty are good, assuming that patients who are chosen for surgery have articulation problems characteristic of tongue-tie, and not speech and language delay or an unrelated speech issue. More than 75% of such children will have demonstrable improvements in articulation as judged by an SP postoperatively as compared with preoperatively [26]. Supplemental speech therapy may be required in the remaining minority to effect retraining of tongue musculature and correction of preoperative compensatory strategies. Although surgery alone may not be fully corrective in all patients, it may allow those with a residual speech deficit to progress more quickly in a program of speech therapy. Because lingual mobility improves quite reliably, one additional
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benefit of surgery in this setting is that it presumably will reduce the chances of other, later-presenting mechanical and social problems as well. Frenuloplasty appears to be equally effective in the treatment of mechanical problems related to tongue-tie, even when surgery is not performed until late childhood [21]. Following frenuloplasty, patients frequently report greater facility with activities such as licking the lips, licking an ice cream cone, using the tongue to sweep the teeth free of debris, kissing, and playing a wind instrument, as well as a decrease in local discomfort and cuts beneath the tongue. Interestingly, patients often will note improvement in some of these activities even when they failed to perceive a limitation preoperatively. For example, a patient may be aware only in hindsight that he or she was unable to lick the lips, when comparing this newfound ability to his or her preoperative status; a patient who has never experienced normal lingual mobility may not fully recognize the extent of his or her limitations until presented with a new frame of reference. Approximately half of our own adolescent and adult patients with uncorrected ankyloglossia have commented that they wished their condition had been repaired earlier in childhood. Complications of frenotomy and frenuloplasty reported historically include infection, excessive bleeding, recurrent ankyloglossia due to excessive scarring, one case of a new speech disorder developing postoperatively, and ‘‘tongue swallowing’’ or glossoptosis due to excessive tongue mobility [1,7,8,29]. Contemporary literature suggests that complications of frenotomy and frenuloplasty are rare. In our experience, and that of others [2,30], frenotomy is quite safe when performed by those who are comfortable and familiar with the procedure. Similarly, frenuloplasty has had an excellent safety record in the current literature; no complications were recorded in a recent series of 158 procedures [21]. Among the potential complications of surgery for tongue-tie, recurrent ankyloglossia is the most common [2]. Some surgeons believe that recurrence is less likely with frenuloplasty as compared with simple frenotomy. When it occurs, recurrent ankyloglossia is generally less severe than at original presentation, and may respond favorably to revision surgery.
Summary Ankyloglossia is an uncommon oral anomaly that can cause difficulty with breast-feeding, speech articulation, and mechanical tasks such as licking the lips and kissing. For many years the subject of ankyloglossia has been controversial, with practitioners of many specialties having widely different views regarding its significance. In many children, ankyloglossia is asymptomatic; the condition may resolve spontaneously, or affected children may learn to compensate adequately for their decreased lingual mobility. Some children, however, benefit from surgical intervention (frenotomy or frenuloplasty) for their tongue-tie. Parents should be educated about the possible long-term effects of tongue-tie while their child is young (< 1 year of age), so that they may make an informed choice regarding possible therapy.
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References [1] Wallace AF. Tongue-tie. Lancet 1963;2:377 – 8. [2] Messner AH, Lalakea ML. Ankyloglossia: controversies in management. Int J Pediatr Otorhinolaryngol 2000;54:123 – 31. [3] Paradise JL. Evaluation and treatment for ankyloglossia. JAMA 1990;262:2371. [4] Ulshen M. Clinical manifestations of gastrointestinal disease. In: Behrman RE, Kliegman RM, Jenson HB, editors. Nelson textbook of pediatrics. 16th edition. Philadelphia: W.B. Saunders; 2000. p. 1102 – 8. [5] Catlin FI, De Haan V. Tongue-tie. Arch Otolaryngol 1971;94:548 – 57. [6] Marmet C, Shell E, Marmet R. Neonatal frenotomy may be necessary to correct breastfeeding problems. J Hum Lact 1990;6:117 – 21. [7] Horton CE, Crawford HH, Adamson JE, et al. Tongue-tie. Cleft Palate J 1969;6:8 – 23. [8] McEnery ET, Gaines FP. Tongue-tie in infants and children. J Pediatr 1941;18:252 – 5. [9] Notestine G. The importance of the identification of ankyloglossia (short lingual frenulum) as a cause of breastfeeding problems. J Hum Lact 1990;6:113 – 5. [10] Berg KL. Tongue-tie (ankyloglossia) and breastfeeding: a review. J Hum Lact 1990;6:109 – 12. [11] Nicholson WL. Tongue-tie (ankyloglossia) associated with breastfeeding problems. J Hum Lact 1991;7:82 – 4. [12] Jorgenson RJ, Shapiro SD, Salinas CF, et al. Intraoral findings and anomalies in neonates. Pediatrics 1982;69:577 – 82. [13] Friend GW, Harris EF, Mincer HH, et al. Oral anomalies in the neonate, by race and gender, in an urban setting. Pediatr Dent 1990;12:157 – 61. [14] Messner AH, Lalakea ML, Aby J, et al. Ankyloglossia: incidence and associated feeding difficulties. Arch Otolaryngol Head Neck Surg 2000;126:36 – 9. [15] Flinck A, Paludan A, Matsson L, et al. Oral findings in a group of newborn Swedish children. Int J Paediatr Dent 1994;4(2):67 – 73. [16] Harris EF, Friend GW, Tolley EA. Enhanced prevalence of ankyloglossia with maternal cocaine abuse. Cleft Palate Craniofac J 1992;29:72 – 6. [17] Brooks JK, Leonard CO, Coccaro PJ. Opitz (BBB/G) syndrome: oral manifestations. Am J Med Genet 1992;43:595 – 601. [18] Martinot VL, Manouvrier S, Anastassov Y, et al. Orodigitofacial syndromes type I and II: clinical and surgical studies. Cleft Palate Craniofac J 1994;31:401 – 8. [19] Gorski SM, Adams KJ, Birch PH, et al. Linkage analysis of X-linked cleft palate and ankyloglossia in Manitoba Mennonite and British Columbia native kindreds. Hum Genet 1994;94: 141 – 8. [20] Fletcher SG, Meldrum JR. Lingual function and relative length of the lingual frenulum. J Speech Lang Hear Res 1968;2:382 – 90. [21] Wright JE. Tongue-tie. J Paediatr Child Health 1995;31:276 – 8. [22] Gray SD, Parkin JL. Congenital malformations of the mouth and pharynx. In: Bluestone CD, Stool SE, Kenna MA, editors. Pediatric otolaryngology. 3rd edition. Philadelphia: W.B. Saunders; 1996. p. 989 – 91. [23] Wilton JM. Sore nipples and slow weight gain related to a short frenulum. J Hum Lact 1990;6: 122 – 3. [24] Huggins K. Ankyloglossia: one lactation consultant’s personal experience. J Hum Lact 1990;6: 123 – 4. [25] Zalzal GH, Cotton RT. Non-cleft disorders of the oral cavity and oropharynx. In: Cummings CW, Krause CJ, Schuller DE, Fredrickson JM, Harker LA, editors. Otolaryngology—head and neck surgery. 2nd edition. St. Louis: Mosby Yearbook; 1993. p. 1169 – 88. [26] Messner AH, Lalakea ML. The effect of ankyloglossia on speech in children. Otolaryngol Head Neck Surg 2002;127:539 – 45. [27] Williams WN, Waldron CM. Assessment of lingual function when ankyloglossia (tongue-tie) is suspected. J Am Dent Assoc 1985;110:353 – 6.
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[28] Kotlow LA. Ankyloglossia (tongue-tie): a diagnostic and treatment quandary. Quintessence Int 1999;30:259 – 62. [29] Fleiss PM, Burger M, Ramkumar H, et al. Ankyloglossia: a cause of breastfeeding problems? J Hum Lact 1990;6:128 – 9. [30] Masaitis NS, Kaempf JW. Developing a frenotomy policy at one medical center: a case study approach. J Hum Lact 1996;12:229 – 32. [31] Ketty N, Sciullo PA. Ankyloglossia with psychological implications. J Dent Child 1974;41:43 – 6. [32] Hazelbaker AK. The Assessment Tool for Lingual Frenulum Function [master’s thesis]. Pasadena (CA): Pacific Oaks College; 1993. [33] Fletcher SG, Daly DA. Sublingual dimensions in infants and young children. Arch Otolaryngol 1974;99:292 – 6.
Ankyloglossia: does it matter? M. Lauren Lalakea, MDa,b, Anna H. Messner, MDa,c,* a
Division of Surgery/Otolaryngology/Head & Neck Surgery, Stanford University, Stanford, CA 94305, USA b Division of Otolaryngology/Head & Neck Surgery, Santa Clara Valley Medical Center, 751 S. Bascom Avenue, San Jose, CA 95128, USA c Lucile Salter Packard Children’s Hospital, Stanford Pediatric Otolaryngology, 725 Welch Road, Palo Alto, CA 94304-5654, USA
An author nearly 40 years ago aptly commented that ‘‘much entertaining nonsense has been written about tongue-tie’’ [1]. Tongue-tie (more formally known as ankyloglossia) is a congenital anomaly characterized by an abnormally short lingual frenulum, which may restrict mobility of the tongue tip. The clinical significance of this anomaly and the best method of management have been the subject of debate for some time [2]. Much of the controversy about management of ankyloglossia probably is related to the paucity of relevant scientific data demonstrating efficacy of intervention. To quote a prominent pediatrician (PD), ‘‘much of the information needed for making rational treatment decisions in cases of tongue-tie is lacking’’ [3]. In a recent edition of a popular pediatric textbook, only one sentence is devoted to the discussion of ankyloglossia [4]. The purpose of this article is to summarize fact, fiction, and areas of controversy about ankyloglossia, to create a greater awareness and understanding of this condition.
Historical context Historical references to tongue-tie may be found beginning in biblical times: ‘‘. . .and the string of his tongue was loosened and he spoke plain,’’ (Mark 7:35). In the eighteenth century, several references, as cited by Catlin and De Haan [5] and Marmet et al [6], recommended clipping the frenulum in tongue-tied infants to facilitate breast-feeding. Horton et al [7] reported that it was the habit of midwives in this period to use their fingernails to divide the lingual frenulum of all infants. Since that time, support for tongue-tie as a cause of feeding or speech problems * Corresponding author. Division of Surgery/Otolaryngology/Head & Neck Surgery, Stanford University, Stanford, CA 94305, USA. E-mail address: [email protected] (A.H. Messner). 0031-3955/03/$ – see front matter D 2003, Elsevier Inc. All rights reserved. doi:10.1016/S0031-3955(03)00029-4
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has waxed and waned. By the early part of the twentieth century, strenuous opposition was raised to the practice of frenotomy [6,7]. Moreover, by the 1940s, some authors were quite vehement in their opinion that ankyloglossia should never be considered causally related to speech difficulties of any type [8]. Current opinion ranges from the belief that tongue-tie only rarely interferes with feeding or speech, and generally requires no treatment [3,4]; to enthusiastic support for frenotomy in the lactation literature [9 –11]. Illustrating this diversity of opinion, a recent article reported the results of a survey of hundreds of US and Canadian PDs, otolaryngologists (OTOs), lactation consultants (LCs), and speech pathologists (SPs), and concluded that there was little consensus among and within these groups with regard to the significance of ankyloglossia or its management [2].
Incidence and natural history Ankyloglossia is uncommon, but not rare. Incidence figures reported in the literature vary widely, ranging from 0.02% to 4.8% [5,12 – 15]. Tongue-tie occurs more commonly in males—with a male-to-female ratio on the order of 3 to 1, and shows no racial predilection [13,14]. The variation in reported incidence may be attributed, in part, to the lack of a uniform definition and objective grading system for tongue-tie. In addition, some of the variation may reflect age-related differences in the presence of this anomaly. Contemporary studies conducted in well-baby nurseries have yielded incidence figures for neonates in the range of 1.7% to 4.8% [12 – 15]. Tongue-tie is thought to be less common in adults, although there are no exact figures in this regard [3,5]. Ankyloglossia occurs most frequently as an isolated anatomic variation. Increased prevalence has been noted in infants with a history of maternal cocaine abuse (incidence = 10.4%, odds ratio = 3.5) [16]. Ankyloglossia also may occur with increased frequency in association with various congenital syndromes, including Opitz syndrome, orodigitofacial syndrome, and in association with X-linked cleft palate [17 – 19]. There is little definitive information on the natural history of untreated ankyloglossia. Certain authors [1,7] have postulated (but not substantiated) that the short frenulum can elongate spontaneously due to progressive stretching and thinning of the frenulum with age and use. This explanation might account for the general perception that this anomaly is more frequently seen in young children, as compared with adults. There is, however, no prospective longitudinal data on the fate of the congenitally short lingual frenulum. This lack of scientific data complicates clinical decision making with regard to the need for treatment of ankyloglossia, if any, and its timing.
Potential manifestations Opinions range widely regarding the clinical significance of ankyloglossia. Some authors feel that ankyloglossia is only rarely symptomatic [3,4], whereas
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others believe it may lead to a host of problems, including infant feeding difficulties, speech disorders, and various mechanical and social issues related to the inability of the tongue to protrude sufficiently [6,7,20 – 22]. The ankyloglossia survey study mentioned above [2] found that as a group, PDs were less likely than their OTO, SP, and LC colleagues to believe that this anomaly was associated with symptoms of any kind. Ninety percent of PDs surveyed indicated that ankyloglossia is never or rarely a cause of feeding dysfunction, whereas 69% of LCs stated that ankyloglossia was frequently or always associated with feeding difficulty. Seventy-seven percent of PDs noted that ankyloglossia is never or rarely a cause of speech dysfunction, whereas only 40% of OTOs believed similarly. Lastly, 67% of OTOs as compared with 21% of PDs believed that tongue-tie is at least sometimes associated with a variety of social and mechanical issues. Although some of the above differences may reflect referral biases, it is clear that beliefs about the significance of ankyloglossia are quite divergent. Information that is relevant to the potential clinical manifestations of tongue-tie is outlined below. Effect on feeding With the increasing popularity of breast-feeding in the past 2 decades, there has been a resurgence of interest in ankyloglossia as a cause of breast-feeding problems [6,9]. Although much of the current literature relating to this topic is in the form of case reports and small uncontrolled case series [6,11,23,24], many LCs and some physicians believe that ankyloglossia can make breast-feeding difficult for at least some infants with this condition [2,6,25]. Multiparous mothers who have breast-fed their unaffected infant(s) successfully in the past note an obvious difference when nursing a subsequent newborn with tongue-tie. Moreover, mothers of affected infants frequently report a marked improvement in breast-feeding after tongue-tie release. Problems reported previously include sore nipples, poor latching and sucking mechanics, poor infant weight gain, and early weaning. A recently published prospective study of infants with ankyloglossia has shed some light on this controversy [14]. Mothers of 36 infants with ankyloglossia noted at birth and mothers of a matched control group of unaffected newborns were followed for up to 6 months to determine the incidence of breast-feeding difficulties. There was no significant difference in the percentage of mothers with affected infants as compared with mothers in the control group who were able to successfully breast-feed for at least 2 months (83% and 92%, respectively). Breastfeeding problems, defined as nipple pain lasting longer than 6 weeks or infant difficulty latching onto the breast, occurred significantly more frequently in the ankyloglossia group, however (25%, versus 3% for controls). This study concluded that ankyloglossia may adversely affect breast-feeding in selected infants. There is general agreement that infants with ankyloglossia do not have trouble with bottle-feeding, nor with handling solid foods once they are introduced. Bottle-feeding, however, should not be proposed as a solution for ankyloglossiarelated problems in a mother who otherwise desires to breast-feed. The role of frenotomy in this situation is discussed later in the article.
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Effect on speech The effect of ankyloglossia on speech has not been defined clearly. Although speech problems historically have been viewed as the hallmark manifestation of symptomatic tongue-tie, particularly among the lay public, others in the literature have vociferously denied that any such relationship exists [8]. Current opinion among speech therapists is evenly divided; 49% of those responding to a survey on this topic believed that ankyloglossia was never or rarely associated with speech problems, and an equal number believed that it was sometimes or frequently associated with speech problems [2]. Anecdotal evidence indicates that some children with ankyloglossia develop normal speech, and compensate for limited tongue-tip mobility without surgical repair or need for speech therapy [1,7,21]. In at least some individuals, however, tongue-tie contributes to articulation errors or difficulty with the rate and range of articulation, thereby decreasing speech intelligibility [1,7,20,21,26]. Speech sounds that may be affected by impaired tongue-tip mobility include lingual sounds and sibilants such as T, D, Z, S, TH, N, and L [1,5,27]. The compensatory techniques used by children with ankyloglossia typically include restricted mouth opening while speaking, and alternate tongue placement for sounds requiring tongue-tip elevation. An excellent study by Fletcher and Meldrum [20] provided strong evidence regarding the relationship of tongue mobility to speech articulation. In this study, normal children, 11 to 12 years of age, underwent careful intraoral measurements of the relative lengths of the tongue and frenulum, to determine the ratio of ‘‘free’’ tongue to total tongue length. Participants were separated into ‘‘limited lingual freedom’’ and ‘‘greater lingual freedom’’ groups on this basis. These investigators found a highly significant increase in the number of articulation errors in the limited lingual freedom group as compared with the greater lingual freedom group, and concluded that these findings might be particularly pertinent to the entity of ankyloglossia. When present, the severity of the articulation problems in affected individuals may vary; problems may be so pronounced as to be evident at the single word level, or be mild enough so as to be noticeable only in connected speech. In our experience, up to one half of young children with ankyloglossia referred for otolaryngology evaluation will have articulation difficulties that may be detected in the context of a formal speech pathology consultation [26]. In addition, among adults with ankyloglossia, even those with grossly normal speech still may complain of speech problems, believing that their speech is more effortful than that of others. It is important to keep in mind that ankyloglossia is not a cause of speech delay. Children with ankyloglossia are expected to acquire speech and language at a normal rate, although some may experience articulation difficulties for certain speech sounds, as indicated above. Occasionally, parents and others who care for a child with speech delay may erroneously ascribe the delay to tonguetie, and demand surgical intervention in the hope that normal speech and
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language will result. In such a patient, potential causes of speech delay should be sought carefully, and the patient should be directed for other evaluations as appropriate (eg, audiologic, speech/language, or neurodevelopmental assessments). Surgical repair may be considered at a later time, once the child has been evaluated fully, and other issues have been addressed. Mechanical and social effects The potential mechanical and social consequences of ankyloglossia have received little attention in the medical literature. Many clinicians are unaware that tongue-tie may have consequences beyond those of speech and feeding difficulties [2]. Mechanical problems related to tongue-tie may include difficulty with intraoral toilet (licking the lips and sweeping the teeth free of food debris), local discomfort or cuts beneath the tongue, dental issues such as a diastasis between the lower central incisors due to pressure from a tight frenulum, and difficulty wearing dentures later in life due to poor fit. Other issues that may be associated with limited lingual range of motion may include decreased facility in playing a wind instrument, difficulty licking an ice cream cone, and difficulty with ‘‘french kissing’’ [1,21,28,29]. In addition, as might be expected, these types of symptoms may be accompanied by a sense of social embarrassment, due to teasing and ridicule from peers [30,31]. In our experience, approximately 50% of older children and adults with persistent ankyloglossia report one or several of the above complaints when their presence is sought in the course of obtaining a complete problem-directed history. These mechanical limitations and social issues may occur even in the absence of other ankyloglossia-related complaints (ie, without a history of feeding or speech problems). It is important to note that mechanical and social concerns may not manifest until later in childhood. Younger children indeed may have symptoms, but may not recognize or be able to report them. In addition, complaints related to activities such as playing a wind instrument and kissing, and issues relating to teasing and ridicule from peers, may not become apparent or relevant until later childhood and beyond. Finally, owing to a sense of embarrassment regarding their condition, patients may refrain from expressing or volunteering their concerns unless questioned directly.
Diagnosis Although the diagnosis of ankyloglossia may be suggested by history, it is confirmed by characteristic physical examination findings. The frenulum may be thick and fibrous or thin and membranous, and is abnormally short, inserting at or near the tongue tip (Figs. 1, 2). The tongue may have a notched or heart shape on protrusion, due to tethering by the frenulum (Fig. 3). Protrusion is limited, and in some cases, may fail to extend past the lower lip. The tongue may appear to roll or curl with attempted protrusion as the midportion of the tongue moves forward,
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Fig. 1. One-day-old male with mild ankyloglossia and difficulty latching onto the breast.
while the tip itself is drawn inferiorly by the frenulum with little forward extension. Affected individuals also characteristically will have difficulty lifting or elevating the tip of the tongue toward the upper dentition and upper lip. Patients may be completely unable to lift the tongue, or in those cases when they are able to elevate the tip to some degree, there is often dimpling noted behind the tongue tip due to tethering. Side-to-side motion of the tongue may be impaired as well. A space or diastasis between the lower teeth is occasionally present, due to the repeated thrusting action of the frenulum through the teeth with attempted tongue protrusion (Fig. 4).
Fig. 2. Preschooler with moderate ankyloglossia and articulation problems.
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Fig. 3. Heart-shaped tongue on protrusion.
A variety of schemas have been proposed to verify the presence of ankyloglossia and grade its severity. Hazelbaker [32], an LC, devised an ‘‘Assessment Tool for Lingual Frenulum Function’’ to be used with neonates. This tool evaluates seven tongue movements, including lateralization, lift, extension, spread of anterior tongue, cupping, peristalsis, and snap-back. Each movement is graded on a 0 to 2 scale; if an infant scores poorly, frenotomy is recommended.
Fig. 4. Four-year-old male with diastasis between his lower central incisors due to ankyloglossia.
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Kotlow [28] used a measurement termed ‘‘free’’ tongue (distance in mm from tongue tip to the insertion of the frenulum) to divide tongue-tie into four classes: mild, moderate, severe, and complete. Unfortunately, this classification scheme failed to provide any documentation regarding the correlation of ankyloglossia class to clinical symptoms. Others [20,33] have used more detailed ratios of lingual dimensions to assess tongue-tie in the older child, and have documented a relationship between these measures and speech articulation. Owing to lack of cooperation and difficulties measuring this mobile muscle, however, these investigators resorted to the use of general anesthesia to accomplish this assessment in young children [20,33]. Williams and Waldron [27] proposed an alternate method, designed to measure lingual function, rather than a physical dimension. These authors recommended measuring the maximum ability of the patient to elevate the tongue, by asking the patient to touch the tip of his or her tongue to the upper teeth, and then open the mouth as widely as possible while maintaining contact. While the patient holds this position, the distance between the upper and lower central teeth, or interincisal distance, is recorded in millimeters (Fig. 5). The interincisal distance is thus used as an objective measure of tongue elevation ability; unfortunately, these authors [27] did not obtain normative data nor did they document clinical correlation. Despite the varied attempts listed above to quantify ankyloglossia severity, at present there is no way to predict—based on examination findings—which children are likely to have, or to develop, speech or mechanical symptoms related to their ankyloglossia. Ability to protrude the tongue past the lower lip has been used by some in the past as a quick rule of thumb to predict which patients will not require surgical repair [1]. In our personal experience with treating children with ankyloglossia, however, we have found that those with an associated articulation disorder (as documented on formal speech pathology assessment) will often be able to protrude the tongue 15 mm or more beyond the lower dentition [26].
Fig. 5. Interincisal distance. The tongue is placed at the back posterior edge of the upper teeth and the mouth is maximally opened. Measurement (in millimeters) is taken between the teeth. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:95; with permission.)
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In our practice, we measure lingual mobility in children who are able to cooperate for protrusion (measured in millimeters of tip extension past the lower dentition) (Fig. 6), and elevation (as measured by recording interincisal distance with the tongue tip maximally elevated and in contact with the upper teeth). Although these measurements may not predict necessarily the presence or absence of symptoms, they provide objective documentation of tongue mobility and help to define the degree of restriction. We have found that protrusion and elevation values are typically in the range of 15 mm or less in children with ankyloglossia, and 20 to 25 mm or greater in normal children [26]. Moreover, mobility measurements are a valuable tool in documenting change, particularly preoperatively and postoperatively.
Indications for intervention History and physical examination generally are sufficient to confirm the presence of ankyloglossia, and supplemental evaluations are not required to establish whether a child may be considered a candidate for surgery. When the presenting problem relates to breast-feeding difficulty, consultation with a lactation specialist
Fig. 6. Tongue protrusion. Measurement (in millimeters) taken from the lower incisors to the tongue tip. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:94; with permission.)
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is an option—particularly if the degree of restriction appears quite mild—to rule out other contributing factors. When the patient’s primary complaint is speech difficulty, a formal speech evaluation may be helpful if the relationship of the ankyloglossia to the speech disorder is in doubt. A failed trial of speech therapy is not a requirement before surgical repair is considered. Patients with ankyloglossia with obvious or definite indications for surgery include infants with associated breast-feeding difficulties, young children with characteristic articulation problems, and older children and adults adversely affected by the mechanical and social manifestations of this condition. There are other relative indications (discussed below); however, the timing of intervention and the benefit of surgery in these alternate clinical scenarios are more controversial. The optimal timing for surgery has not been determined. Some investigators have advocated surgical intervention prior to the development of speech difficulties [22,25], whereas others have mandated withholding surgery until age 4 or more, and then offering surgery only to those with a manifest speech problem [1,3,7]. Current opinion is no less varied; in a recent survey regarding ankyloglossia treatment, 34% of OTOs believed that surgery was inappropriate in children less than 1 year of age, 36% believed surgery could be performed at any age, and 42% believed surgery to be inappropriate (and perhaps less effective) for those 12 years of age or older [2]. In our view, given the minor nature of the surgery and the significant potential for speech difficulties and later social and mechanical problems, it may be appropriate to consider surgery for those children with significant ankyloglossia at any age, including infants and toddlers who have yet to demonstrate overt symptoms. When very young children are referred for evaluation, we inform parents that there is no way to predict which children will develop symptoms related to their condition, and which children may remain asymptomatic or later outgrow their condition. We do, however, present information regarding the potential early and late consequences of ankyloglossia and solicit parental opinion with respect to treatment preferences. Although early intervention in all children before the development of speech problems may be unwarranted, delaying intervention until obvious difficulties emerge may commit some children unnecessarily to a period of rehabilitative speech therapy or social embarrassment. One additional consideration when evaluating an infant with ankyloglossia is that up to several months of age, a frenotomy can be performed quickly in the clinic without requiring general anesthesia. In contrast, if surgery is deferred until the child is older than 1 year of age, general anesthesia in the operating room usually is required. Cooperative children older than 6 to 7 years of age usually can undergo repair under local anesthesia, but deferring surgery until this age can subject a child to an unnecessarily long period of symptoms or social concerns. Therefore, our practice is to discuss these issues with parents, who may elect early intervention at their discretion, or may prefer to wait and elect surgery only if and when symptoms occur. There are no absolute contraindications to surgery; however, as noted earlier, children with apparent speech delay should be approached with caution, and
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surgery should be deferred until the child has been evaluated and treated by other specialists as appropriate.
Alternatives to surgery Alternatives to surgery include observation or a trial of speech therapy. The wait-and-see approach may be considered for the young child with minimal symptoms, because it is possible that the child will outgrow the ankyloglossia, or compensate sufficiently so that future clinically significant speech or mechanical problems will not occur. In the child with speech difficulties, speech therapy may correct articulation errors successfully in some cases, although SPs vary in their degree of optimism for this approach [2]. One disadvantage of speech therapy is that it may require a lengthy period during which time the child may be self-conscious about his or her impairment. The benefit of speech therapy must be weighed against surgical treatment that, although invasive, is simple, quick, and effective. Another disadvantage is that although speech therapy may allow the child to improve his or her articulation over time by using various compensatory mechanisms, it may still leave him or her vulnerable to later manifesting mechanical problems associated with restricted lingual mobility.
Surgical procedures Frenotomy, or simple release of the frenulum, and frenuloplasty (release with plastic repair) are the two most commonly used surgical procedures in the treatment of ankyloglossia. As noted above, appropriate patient selection and ideal age for intervention have been topics of much disagreement. Past reports of complications and the lack of clearly agreed-on measures of success have contributed to the debate. Frenotomy Frenotomy (Fig. 7) also has been termed ‘‘clipping’’ of the frenulum, and is most appropriate for the treatment of ankyloglossia in infants (eg, for breastfeeding problems) because it is rapid and relatively easy to accomplish. The procedure may be performed at the bedside in the newborn nursery, or in the office with local or no anesthesia. Practitioners who typically perform frenotomy include OTOs, dentists, and PDs. Interestingly, 22% of a group of 425 US and Canadian PDs who responded to a survey for a recent paper on this topic indicated that they had performed frenotomy, although only 10% reported that they had been taught this skill in residency [2]. Although some practitioners use local anesthesia, in our experience, the discomfort associated with the release of thin and membranous frenula is brief and quite minor. The infant is positioned in front of the clinician with his or her
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Fig. 7. Frenotomy. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:95; with permission.)
head directed toward the clinician’s left (if the clinician is right handed). Two gloved fingers of the clinician’s left hand are placed below the tongue on either side of midline, to retract it upward toward the palate, exposing the frenulum. Small sterile scissors are used to release the frenulum, beginning at its free border and proceeding posteriorly directly adjacent to the tongue to avoid injury to the (more inferiorly placed) submandibular ducts in the floor of mouth. Occasionally, complete release may be accomplished with a single scissor cut, but more frequently, especially when the frenulum is quite tight, two or three sequential cuts are required; each cut provides some release, allowing improved retraction and visualization for subsequent cuts, if necessary. Bleeding is generally slight, although the amount of blood may appear greater due to mixing with saliva. If needed, bleeding can be controlled easily with a brief period of pressure applied with a 2 2-inch gauze. The incision is not sutured, and the infant is allowed to feed immediately following the procedure. Acetaminophen may be used for pain control, but often is not required. Antibiotic therapy is unnecessary. The patient may be seen in follow-up in 1 to 2 weeks as needed; generally by that time the incision has healed completely. Once the practitioner has sufficient experience with the procedure, telephone follow-up may be appropriate as long as the patient is doing well. Frenuloplasty Frenuloplasty (Fig. 8) is the preferred procedure for most patients greater than 1 to 2 years of age, because it allows for more complete release of the tongue-tie. In addition, because a plastic closure is performed, some surgeons believe that the chance of scarring and recurrent ankyloglossia is reduced. In young children, the
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Fig. 8 A-C. Horizontal to vertical frenuloplasty. (A) the incision line; (B) the resulting defect; and (C ) the closure line. (From Lalakea ML, Messner AH. Frenotomy and frenuloplasty: if, when, and how. Op Tech Otolaryngol 2002;13:96; with permission.)
procedure is performed using a brief general anesthetic, whereas in older children and adults, it can be accomplished easily in the clinic using a local anesthetic. The frenulum is released in a manner identical to that used for frenotomy. Occasionally, limited division of the genioglossus muscle may be required to effect adequate release. The resulting wound is closed with sutures. Some surgeons use a z-plasty flap closure, and others prefer to close the diamond-shaped defect created by the initial horizontally oriented release in a vertical manner (hori-
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zontal-to-vertical plasty). Both methods cover the exposed raw surface and result in additional lengthening of the frenulum. Frenuloplasty is only slightly more involved than is simple frenotomy, and requires only minutes to perform. Bleeding is generally minimal, and is controlled with pressure or cautery. Patients are advised to resume a normal diet as tolerated. As a rule, patients report little in the way of postoperative discomfort, and may use acetaminophen as needed for pain. Antibiotics are not required. When patients are old enough to comply, we ask them to perform a series of tongue exercises several times daily for the first 4 to 6 weeks postoperatively, with the expectation that this practice may enhance mobility, assist in retraining of tongue musculature, and reduce the potential for scarring.
Surgical outcome The results of frenotomy and frenuloplasty for ankyloglossia are highly favorable, provided that surgical indications are met and patients are appropriately selected for intervention. In children and adults, tongue mobility measures predictably improve in the first 1 to 3 months following surgery, and tend to remain stable after that time. Postoperatively, patients may be expected to gain 10 mm or more of tongue protrusion and tongue elevation on average, as compared with preoperative measures of mobility [26]. In addition, significant subjective gains may be anticipated as well; a majority of parents/patients report that they are highly satisfied with these procedures. Frenotomy has been reported to result in immediate improvement in problems related to breast-feeding in a majority of cases [6,9,30]. There are multiple anecdotal reports in the lactation literature describing rapid resolution of maternal nipple pain, better latching, and enhanced infant weight gain [10,11,23,24]. In our practice, it is not uncommon for mothers to note a marked difference in nursing mechanics as early as the first feeding after frenotomy. Frenotomy is not a panacea for all breast-feeding problems, however; when the degree of ankyloglossia is minimal, or there is doubt as to the contribution of tongue-tie to nursing problems, it may be wise to obtain a lactation consultation or to counsel the mother appropriately regarding expectations before proceeding with frenotomy. Speech results following frenuloplasty are good, assuming that patients who are chosen for surgery have articulation problems characteristic of tongue-tie, and not speech and language delay or an unrelated speech issue. More than 75% of such children will have demonstrable improvements in articulation as judged by an SP postoperatively as compared with preoperatively [26]. Supplemental speech therapy may be required in the remaining minority to effect retraining of tongue musculature and correction of preoperative compensatory strategies. Although surgery alone may not be fully corrective in all patients, it may allow those with a residual speech deficit to progress more quickly in a program of speech therapy. Because lingual mobility improves quite reliably, one additional
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benefit of surgery in this setting is that it presumably will reduce the chances of other, later-presenting mechanical and social problems as well. Frenuloplasty appears to be equally effective in the treatment of mechanical problems related to tongue-tie, even when surgery is not performed until late childhood [21]. Following frenuloplasty, patients frequently report greater facility with activities such as licking the lips, licking an ice cream cone, using the tongue to sweep the teeth free of debris, kissing, and playing a wind instrument, as well as a decrease in local discomfort and cuts beneath the tongue. Interestingly, patients often will note improvement in some of these activities even when they failed to perceive a limitation preoperatively. For example, a patient may be aware only in hindsight that he or she was unable to lick the lips, when comparing this newfound ability to his or her preoperative status; a patient who has never experienced normal lingual mobility may not fully recognize the extent of his or her limitations until presented with a new frame of reference. Approximately half of our own adolescent and adult patients with uncorrected ankyloglossia have commented that they wished their condition had been repaired earlier in childhood. Complications of frenotomy and frenuloplasty reported historically include infection, excessive bleeding, recurrent ankyloglossia due to excessive scarring, one case of a new speech disorder developing postoperatively, and ‘‘tongue swallowing’’ or glossoptosis due to excessive tongue mobility [1,7,8,29]. Contemporary literature suggests that complications of frenotomy and frenuloplasty are rare. In our experience, and that of others [2,30], frenotomy is quite safe when performed by those who are comfortable and familiar with the procedure. Similarly, frenuloplasty has had an excellent safety record in the current literature; no complications were recorded in a recent series of 158 procedures [21]. Among the potential complications of surgery for tongue-tie, recurrent ankyloglossia is the most common [2]. Some surgeons believe that recurrence is less likely with frenuloplasty as compared with simple frenotomy. When it occurs, recurrent ankyloglossia is generally less severe than at original presentation, and may respond favorably to revision surgery.
Summary Ankyloglossia is an uncommon oral anomaly that can cause difficulty with breast-feeding, speech articulation, and mechanical tasks such as licking the lips and kissing. For many years the subject of ankyloglossia has been controversial, with practitioners of many specialties having widely different views regarding its significance. In many children, ankyloglossia is asymptomatic; the condition may resolve spontaneously, or affected children may learn to compensate adequately for their decreased lingual mobility. Some children, however, benefit from surgical intervention (frenotomy or frenuloplasty) for their tongue-tie. Parents should be educated about the possible long-term effects of tongue-tie while their child is young (< 1 year of age), so that they may make an informed choice regarding possible therapy.
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