U.S. flag

An official website of the United States government

NCBI Bookshelf. A service of the National Library of Medicine, National Institutes of Health.

Francis DO, Chinnadurai S, Morad A, et al. Treatments for Ankyloglossia and Ankyloglossia With Concomitant Lip-Tie [Internet]. Rockville (MD): Agency for Healthcare Research and Quality (US); 2015 May. (Comparative Effectiveness Reviews, No. 149.)

Cover of Treatments for Ankyloglossia and Ankyloglossia With Concomitant Lip-Tie

Treatments for Ankyloglossia and Ankyloglossia With Concomitant Lip-Tie [Internet].

Show details


Results of Literature Searches

We identified 1,626 nonduplicative titles or abstracts with potential relevance, with 244 proceeding to full text review (Figure 3). We excluded 187 studies at full-text review, which yielded 57 published studies included in the review. We also included one unpublished thesis in our results, thus the report summarizes data from 58 unique publications.

This figure outlines the disposition of studies identified for the review. We identified 1626 nonduplicative titles or abstracts with potential relevance, with 244 proceeding to full text review (1382 excluded at abstract review). We excluded 187 studies at full text review, which yielded 57 published studies included in the review. We also included one unpublished thesis in our results, thus the report summarizes data from 58 unique publications.

Figure 3

Disposition of articles identified by the search strategy. * Articles may be excluded for multiple reasons † Includes 15 case reports of harms. We also included data from 1 unpublished thesis.

Description of Included Studies

The 58 unique publications included in the review comprise six randomized controlled trials (RCTs), three assessed as good quality7, 8, 20 for outcomes related to breastfeeding effectiveness and maternal pain related to breastfeeding. One RCT was rated as poor quality for breastfeeding effectiveness and pain outcomes.21 One RCT was of poor quality for outcomes of tongue protrusion, frenulum length, and articulation/intelligibility,22 and we rated one RCT as fair quality for measures of breast and bottle feeding.23 The literature also includes three cohort studies (all poor quality24-26), 33 case series,3, 6, 27-57 and 15 case reports (one of which reports two cases and one of which reports five cases).58-72 We also included one unpublished thesis (not quality rated). Table 6 outlines study characteristics.

Table 6. Overview of comparative studies included.

Table 6

Overview of comparative studies included.

Because case series do not include comparison groups, they do not provide comparative effectiveness data but were read to determine if they generally provided support for comparative data and as an additional source of harms. We used case reports to seek harms data only. We considered all comparative studies (RCTs and cohort studies) as poor quality for harms outcomes. We considered the quality for harms outcomes as good in four case series49-52 and poor in 26.3, 6, 27-48, 54, 56

KQ (Key Question) 1. Benefits of Interventions To Improve Breastfeeding Outcomes

Key Points

  • Results for reduction in nipple pain immediately after surgery were inconsistent, and potentially associated with how early after birth surgery occurred, with the one good quality study with positive results including the youngest infants.
  • Frenotomy was associated with significantly improved maternally reported breastfeeding effectiveness immediately post-procedure compared with sham in two RCTs7, 20, but inconsistent evidence that it improved infant's latch and breastfeeding effectiveness compared with no intervention. Results on whether frenotomy prolonged duration of breastfeeding were unclear and not consistent.
  • No comparative study identified expressly evaluated the role of non-surgical interventions in improving breastfeeding effectiveness.

Overview of the Literature

Twenty-nine studies provided data on breastfeeding outcomes after surgical treatments for ankyloglossia. Only six included a comparison group and could provide information on comparative effectiveness. These studies included five randomized controlled trials conducted either in the United Kingdom (n=3),8, 20, 23United States (n=1),7 or Israel (n=1)21 and one retrospective cohort study conducted in the United States.25 We rated three RCTs as good quality for outcomes related to breastfeeding effectiveness and pain related to breastfeeding.7, 8, 20 One RCT was rated as fair23 and one as poor quality for breastfeeding effectiveness and pain outcomes,21 and we rated the cohort study as poor quality. The remainder of the studies were case series and therefore used to identify harms (n=23). Case series were conducted in the United Kingdom (n=11),28, 29, 32, 35, 36, 39, 41, 49, 50, 53, 57United States (n=5),3, 31, 44, 45, 56Australia (n=3),30, 38, 40Finland (n=1),48 Israel (n=2),52, 55 and Canada (n=1).27

In the studies that provided breastfeeding outcomes, ankyloglossia was only identified in the presence of breastfeeding difficulties. It was diagnosed by clinician examination in all comparative studies but using different methods. In three studies, clinicians diagnosed it from exam without defining clear diagnostic criteria.20, 23, 25 In others, ankyloglossia was defined as breastfeeding difficulties combined with either 1) Hazelbaker Assessment Tool of Lingual Frenulum Function (HATLFF) score between 6 and 12 and Latch, Audible swallowing, Type of nipple, Comfort, Hold (LATCH) score ≤88, or 2) abnormal HATLFF (cut-off not defined).7

Two RCTs compared frenotomy to sham surgery,7, 20 one to usual care,8 one to intensive lactation consultation,23and one used a crossover design to compare frenotomy followed by sham surgery to sham surgery followed by frenotomy with assessment of breastfeeding after each order of intervention (i.e., frenotomy and sham).21 Similarly, the retrospective cohort study compared frenotomy to usual care.25 The frenotomy procedure was explicitly described by three of five RCTs and the cohort study. In all descriptions, the frenulum was divided with straight scissors: straight iris (1),25 blunt tipped (2),20, 23 unspecified (1).7 Two RCTs mentioned frenotomy without specifying how it was technically performed.8, 21 The cohort study was the only comparative study that described systematic use of anesthetic (i.e., viscous lidocaine) prior to ankyloglossia division;25 however, when case series were considered, a total of four of 25 studies reported use of some anesthetic before surgery.3, 25, 31, 49 In the sham procedure, infants were removed from their parents to a separate room for the same amount of time as those receiving the procedure.

Detailed Analysis

Overview by Study Design for All Breastfeeding Outcomes

Randomized Controlled Studies

Five RCTs addressed the benefits of treating ankyloglossia with frenotomy on breastfeeding outcomes among neonates and infants who had breastfeeding difficulties (Table 7). The first good quality RCT was single-blinded and randomly assigned infants causing maternally reported nipple pain or difficulty breastfeeding with concomitant and significant ankyloglossia diagnosed by lactation consultant based on HATLFF criteria to frenotomy (n=30) or a sham procedure (n=28).7 Infants in this study were young (mean 6.0 ± 6.9 days), and had a gender distribution of approximately 2:1 male: female in both treatment groups. Primary outcomes were 1) nipple pain assessed using the Montreal Pain Questionnaire (MPQ-SF); 2) objective breastfeeding effectiveness using Infant Breastfeeding Assessment Tool (IBFAT); and 3) lingual frenulum function via the HATLFF appearance and function scores. Mothers assessed pain outcomes and were blinded to their infant's treatment group.

Table 7. Breastfeeding effectiveness following surgical procedures.

Table 7

Breastfeeding effectiveness following surgical procedures.

Mothers whose infants had frenotomy reported significantly less nipple pain immediately following the procedure (mean MQP-SF: 4.9 ± 1.46 vs. 13.5 ± 1.5, p<0.001), which remained significantly less than the sham group until the 4-week assessment. Moreover, the mean IBFAT score was higher among frenotomized infants than those undergoing the sham procedure (11.6 ± 0.81 vs. 8.07 ± 0.86, p=0.026) immediately post-procedure, but was no different from the sham group at 2-week postoperative evaluation.

A second good quality RCT randomized infants less than 4 months of age with breastfeeding problems and ankyloglossia to either frenotomy (n=30) or sham procedure (n=30). There was nearly identical distribution of males and females (∼2:1) and mean ages between groups (33 vs. 28 days).20 The primary outcome was objectively observed improvement in breastfeeding effectiveness using a score adapted from LATCH and IBFAT, and the secondary outcome was maternally reported improvement in breastfeeding immediately after intervention. Treatment allocation was blinded to both the parents and independent outcome assessor.

No difference in breastfeeding improvement was reported by trained objective observers immediately following intervention (50% [13/26] vs. 40% [12/30]). In contrast, mothers whose infants had frenotomy reported significantly improved breastfeeding compared with those in the sham group (78% [21/27] vs. 47% [14/30] p<0.02). There was no immediate difference in the reduction in maternal reported pain scores between the frenotomy and sham groups (mean -2.5 ± 1.9 and -1.3 ± 1.5, p=0.13). Although the study reports that they re-assessed outcomes at 3 months, the data are not provided by treatment group.

A third good quality RCT randomized term infants with breastfeeding difficulties and ankyloglossia (HATLFF score between 6 – 12 and LATCH score ≤ 8) to either frenotomy (n=55) or no intervention (n=52).8 All dyads consulted with a lactation consultant prior to randomization. Infants with severe ankyloglossia (defined as HATLFF < 6) were excluded and offered immediate frenotomy. At randomization, the median age was 11 days (IQR 8 – 14) and 11 days (IQR 8 – 16) in the frenotomy and control groups, respectively (p=0.94). This study did not report on gender of enrolled infants, but matched infants on age and birth order. Primary outcomes assessed 5-days and 8 weeks post-procedure included 1) change in maternal pain using VAS and 2) LATCH score. Secondary outcomes were method of feeding (i.e., bottle vs. breast), percent breastfeeding, and Breastfeeding Self-Efficacy Scale-Short Form (BSES-SF) score. Independent researchers collecting outcomes, but not mothers, were blinded to infant group assignment and performed assessment at the 5-day follow-up visit. The 8-week assessment was limited since 35 of 52 in the comparison group requested frenotomy before that follow-up date due to continued breastfeeding problems. Therefore, the 8-week comparison was between 52 of 55 of the frenotomized infants, and 50 of 52 in the “no intervention” group of whom only eight of 50 (15%) had not had frenotomy at the time of this follow-up assessment.

Five days after the procedure, reductions in pain scores were not significantly greater among mothers whose infants had a frenotomy (median -2 [IQR -3 to 0.4] vs. -1 [-13.5 to 1]). Of note, 17 percent randomized to usual care did not wait 5 days before getting a frenotomy due to painful breastfeeding. Similarly, no significant improvement in median maternal pain was reported 8 weeks post-procedure (median -2 [IQR -3 to -1] vs. -2 [-3.5 to -0.6], p=0.83). Infant outcomes showed no differential median improvement between frenotomy and control group at 5-days for LATCH score (median 1 [IQR 0 – 2] vs. 1 [0 – 2], p=0.52) or IBFAT score (median 0 [IQR -1.8 to 1.0] vs. 0 [IQR 0 – 1]), p=0.36).

In contrast, compared with controls, there was improvement in both median BSES-SF score (median 9 [IQR 1.8 – 12.3] vs. 1 [-4 to 7.5] p=0.0002) and HATLFF score (4.5 [IQR 3.3 – 6] vs. 0 [0 – 2.3], p<0.001) 5-days post-intervention in the frenotomy group. Between 5-days and 8 weeks post-intervention, there was less improvement in the median BSES-SF score among frenotomy infants compared with those in the control group, but this difference was not statistically significant (3 [IQR 0 – 13] vs. 10 [2 – 18], p=0.082). The BSES-SF improvement occurred more rapidly after frenotomy in the surgery group than in the control group, but by 8-weeks both groups were nearly equivalent in overall improvement (5-day median + 8-week median: frenotomy 9 + 3=12 vs. control 1 + 10=11). However, this comparison is difficult to interpret because so many control infants underwent frenotomy between the 5- and 8-week assessments. Crossover to frenotomy may also explain the equivalence of exclusive breastfeeding rates between groups at the 8-week assessment (intervention 82.7% vs. 80%, p=0.73).

A fair quality RCT randomized infants born with ankyloglossia diagnosed within the first 5 months with feeding problems to either frenotomy (n=28) or a control group who had intensive support, advice and help from lactation consultants (n=29).23 The percentage of the tongue attached by the frenulum was gauged by clinician visualization to be between 0 percent (i.e., none) and 100 percent (i.e., to the tongue tip). This was judged to be 25 percent in six patients, 50 percent in 13, 75 percent in 15, and 100 percent in 23. Infants in both the frenotomy and control group had similar ages (20 vs. 18 days), but gender distribution was only recorded for the frenotomy group where there was a 1:1 ratio of males to females. The primary outcome was maternally reported improvement in breastfeeding. Most (96%) of frenotomized infants had improved feeding with 48 hours compared with 3 percent in the control group. The study was, however, entirely unblinded and all outcomes were by maternal report.

The final poor quality trial randomized full-term healthy for gestational age infants, ages 1 to 21 days, who were referred to a lactation clinic due to maternal nipple pain, and diagnosed with ankyloglossia by a neonatologist to either frenotomy followed by sham procedure (n=15) or vice versa (n=11) with assessment of breastfeeding after each intervention type in both arms.21 Neither infant ages nor gender distribution was reported. The study's primary outcomes were maternal breastfeeding pain or nipple trauma measured by a standard Visual Analog Scale (VAS) and breastfeeding LATCH scores. Main outcome assessors were the mothers who were blinded to infant treatment group. Comparative group results were not reported, therefore preventing comparative analysis in this review.

Cohort Studies

A single poor quality retrospective cohort study compared frenotomy to no intervention.25 It included 367 infants with feeding or latching difficulties that caused maternal pain when breastfeeding, 302 of whom underwent frenotomy. In this cohort, 58.6 percent of infants were male, mean age at ankyloglossia diagnosis was 18 days, and the majority of patients were either Caucasian (70.3%) or African American (15.5%). Ankyloglossia grade was recorded using Coryllos et al. system.73 Overall, 17.4 percent had type I, 45.5 percent type II, 25.3 percent type III, 18 percent type IV, and 5.8 percent indeterminate. Outcomes were only assessed in the 91 mothers (24.9%) who agreed to participate in a follow-up survey (82 had frenotomy, 9 no intervention), thus limiting its generalizability. Nonetheless, 80.4 percent of interviewed mothers whose infant had undergone frenotomy felt it had benefited their child's ability to feed. Breastfeeding was continued in 82.9 percent of 82 frenotomized infants for a mean 7.09 months total compared with 66.7 percent of nine infants not treated who breastfed a mean 6.28 months total. In all, 17.1 percent and 33.3 percent in the frenotomy and no intervention group stopped breastfeeding due to difficulty or pain due to ankyloglossia. Having a frenotomy in the first week of life versus later did not affect the total months of breastfeeding (mean: ≤7 days 7.11 vs. >7 days 7.06 months; p<0.9).

Case Series

We identified 23 case series that addressed treatments for ankyloglossia on effectiveness of breastfeeding. All studies focused on surgical treatments, which included frenotomy, frenulotomy, or frenuloplasty. None explicitly evaluated non-surgical interventions. By design, none included a comparison group, thereby eliminating the ability to assess comparative effectiveness of surgical approaches, although the studies typically reported improvements in breastfeeding effectiveness after surgery. Harms reported in case series are included in KQ5.

Analysis of Breastfeeding Effectiveness

Immediate Outcomes

Breastfeeding effectiveness was evaluated in four of five RCTs (Table 8).7, 8, 20, 23 We rated two RCTs as good quality for these outcomes7, 20 and two as fair quality.8, 23 Among the three RCTs that used a blinded independent reviewer to assess effectiveness,7, 8, 20 one reported objective improvement in breastfeeding effectiveness based on IBFAT score immediately postfrenotomy compared with sham treatment (mean 11.6 ± 0.81 vs. 8.07 ± 0.86; p=0.026).7 In contrast, in two of the three RCTs, the independent blinded observers did not detect a difference in breastfeeding improvement. Outcomes that failed to show a difference in these two RCTs included percent improvement (50% vs. 40%) immediately after intervention20and LATCH and IBFAT change 5-days post-intervention (LATCH change: median 1 [IQR 0 – 2] vs. median 1 [ IQR 0 – 2], p=0.52 and IBFAT change: 0 [IQR -1.8 to 1.0] vs. 0 [IQR 0 – 1], p=0.36).8

Table 8. Breastfeeding-associated pain scores after surgical procedures.

Table 8

Breastfeeding-associated pain scores after surgical procedures.

Three of four RCTs with usable data used maternally reported improvement in breastfeeding as an outcome,8, 20, 23 and in one, it was the primary outcome measure of effectiveness.23 Maternally reported outcomes differed from objective independent assessment reported above. For example, in one RCT, mothers self-reported improved breastfeeding among infants immediately after frenotomy (78% in the treated group vs. 47% in the comparison group, p<0.02).20 Similarly, another trial using non-blinded maternally assessed breastfeeding effectiveness reported that 96 percent of frenotomized infants had improved feeding with 48 hours compared with 3 percent in a control group who had intensive lactation consultant support.23 Finally, one RCT used the BSES-SF as a secondary outcome and found that mothers whose infants had had frenotomy had significantly improved scores 5 days after intervention (median BSES-SF =9 [IQR 1.8 – 12.3] vs. 1 [IQR -4 to 7.5], p=0.0002).8

Longer Term Outcomes

Three RCTs7, 8, 20and the retrospective cohort study25 followed up dyads during the first postoperative year. One RCT contacted mothers 3 months after frenotomy, but did not stratify results by treatment group.20 Overall, 92 percent (54/59) of all patients reported improved feeding, with 56 percent reporting full resolution of breastfeeding difficulties. Moreover, 65 percent (38/59) of infants were being breastfed at 3 months of age, whereas 51 percent (30/59) were continuing to breastfeed at second outcome assessment (4.5 months). The second RCT evaluated results 2-weeks post-operatively and found no difference between those who underwent frenotomy or sham treatment.7 A third RCT found no difference in breastfeeding effectiveness between groups as measured by LATCH score at an 8-week follow-up survey, but mothers did report nonsignificantly improved BSES-SF scores among frenotomized infants.8 Of note, 35 of 52 children assigned to the control arm had undergone frenotomy after 5 days. Seventeen of 35 had not had surgery, and two additional infants were lost to followup at 8 weeks.

The retrospective cohort reported that breastfeeding was continued in 82.9 percent of frenotomized infants for a mean 7.09 months total compared with 66.7 percent of infants not treated who breastfed a mean 6.28 months total. In all, 17.1 percent in the frenotomy and 33.3 percent in the no intervention group stopped breastfeeding due to difficulty or pain due to ankyloglossia. Having had frenotomy in the first week of life versus later did not affect the total months of breastfeeding (mean: ≤7 days 7.11 vs. >7 days 7.06 months; p<0.90).

Maternal Pain Outcomes

Among comparative studies, three RCTs, rated as good7, 8, 20 for pain outcomes, reported on maternal nipple pain outcomes. Of these, one reported significant and immediate improvement in maternally reported nipple pain among mothers of frenotomized infants compared with sham treatment.7 Both remaining RCTs found nonsignificant reductions in maternally reported nipple pain between the frenotomy and sham groups at immediate20 and 5-day8 post-procedure assessments. Of note, 17 percent of infants randomized to no intervention in the study that followed patients out five days8 requested and received early frenotomy before the data were collected.

KQ2a. Benefits of Treatments To Mitigate Feeding Sequelae

Key Points

  • Existing data are insufficient to draw conclusions about the benefits of surgical interventions for infants and children with ankyloglossia on medium- and long-term feeding outcomes other than breastfeeding. The studies used different populations and measured different outcomes.

Overview of the Literature

We identified three studies examining medium- and long-term benefits related to feeding outcomes and sequelae of various interventions for infants and children with ankyloglossia (Table 9).23, 24, 35 One was an RCT23 (fair quality for feeding outcomes) and one was a poor quality retrospective cohort study24; the remaining study was a case series.35 All studies were single center or single surgeon studies. Two studies were conducted in the United Kingdom23, 35 and one study in the United States.24

Table 9. Feeding sequelae.

Table 9

Feeding sequelae.

Detailed Analysis

Comparative data were included in two studies.23, 24 A detailed description of the included fair quality RCT study design and population are reported in the detailed analysis for KQ1. In summary, the study23 randomized infants born with ankyloglossia and diagnosed within the first 5 months with feeding problems to either frenotomy (n=28) or a control group who had intensive support, advice and help from lactation consultants (n=29). Outcomes were based solely on maternal-report within 48-hours of randomization. However, in the RCT the control group was offered – and the majority elected to receive –frenotomy within 48 hours of randomization to the comparison group, so the outcomes do not reflect “medium to long term” feeding outcomes. This study was included herein, because it includes data on bottle-feeding efficiency. Outcomes related directly to breastfeeding are presented in KQ1.

Among pre-treatment bottle fed infants, 76 percent had major problems with dribbling, and 71 percent had “excess wind” (gas). Mothers reported significant improvement in feeding in all eight who received the frenotomy and in none who did not. The interval to ascertainment of outcomes was not specifically reported, but outcomes were obtained within the first 4 weeks of life.

The retrospective cohort study compared parent-reported (typically maternal) outcomes at age 3 years for children born in 2010 who 1) received frenotomy for tongue-tie (n=71; frenotomy group), 2) were offered but declined frenotomy for tongue-tie (n=15; no frenotomy group), and 3) children without ankyloglossia (n=18; control group).24 Three questions rated on a 5-point Likert scale were used to assess a child's difficulty (a) cleaning his or her teeth with the tongue, (b) licking the outside of his or her lips, and (c) eating ice cream. With respect to answers on each of the questions, the frenotomy group performed better than the no frenotomy group at age 3 years and did not differ significantly from the comparison group without ankyloglossia. P-values were presented without reporting the central tendency (e.g., median, mean) or variance (IQR, SD) from which they were calculated. Therefore, further comparative description or analysis was not possible.

In the case series of 62 infants, 51 had complete outcome data (11 lost to follow-up).35 Of these, infant ages ranged from 12 to 35 days at time of referral for frenulotomy by plastic surgeon, and outcomes were assessed prospectively over an 8-month period, on the day of frenulotomy, and at 2-weeks post-procedure at outpatient appointment. Over this period, the number of breastfeeding sessions decreased from 10 ± 0.7 pre-frenulotomy to 7 ± 0.5 postfrenulotomy (p<0.0001) and bottle feeding supplementary sessions per day were reduced from nine to two at 2-week follow-up (p<0.0001). The authors suggest that this reflects longer-term improvement in feeding efficiency.

KQ2b. Benefits of Treatments To Prevent Other Sequelae

Key Points

  • Two studies reported better articulation among children who had received ankyloglossia treatment compared to those who had not, but results related to word, sentence, and fluent speech were inconsistent.
  • Results in two studies comparing children with ankyloglossia who received treatment to children without a history of ankyloglossia were inconsistent.
  • One small, poor quality RCT compared two surgical methods and reported that children in a four-flap Z-frenuloplasty group had greater articulation gains than those in the horizontal-to-vertical frenuloplasty group.
  • Although a number of case series report positive outcomes related to speech after treating ankyloglossia, most discussed modalities, with safety, feasibility or utility as the main outcome, rather than speech itself.

Overview of the Literature

Ten studies addressed ankyloglossia treatment in children with speech and articulation concerns. One RCT22 rated as poor quality comparing two different surgical techniques and one poor quality cohort study24 were conducted in the United States. An additional poor quality retrospective cohort study was conducted in Israel (Table 10).26 Of seven case series addressing this question, two were conducted in the United States,42, 43 one each from the United Kingdom,51 China,37 India,47 Japan,54 and Korea.34 No study addressed the effect of ankyloglossia on sleep disordered breathing, dental/occlusal issues, or dysphagia.

Table 10. Comparative studies with speech outcomes.

Table 10

Comparative studies with speech outcomes.

Among the comparative studies identified, two of three had speech and articulation assessed by speech-language pathologists,22, 26 while the third relied on parental report.24 Professional assessment was performed by speech-language pathologists using the Articulation and Naming Test26 in one of two studies in which they were the outcome assessors and with the other using consensus between speech-language pathologists.22 The third study used a non-validated parental survey to determine parent perception of the severity of the child's speech misarticulations.24

Detailed Analysis

Cohort Studies

One poor quality retrospective cohort study24 compared three treatment groups of children who were three years old in 2010 who had: (1) ankyloglossia and frenotomy within the first month of life (n=71), (2) ankyloglossia and whose parents declined frenotomy during the same period (n=21), and (3) a control group of randomly selected 3-year old patients with no history of ankyloglossia (n=18). Three-year old subjects were chosen because that is the age that speech and articulation abnormalities typically present. Pediatric otolaryngologists assessed ankyloglossia using Coryllos criteria in the postpartum ward or during outpatient clinical examination. Parents of all identified patients were then contacted for a telephone survey that consisted of nine questions related to the health care provider who identified restriction, recommendations for surgery, intelligibility of speech to parent(s), impaired speech sounds, deficiencies in oral motor activities, and perceived need for speech therapy. Speech intelligibility was graded on a 5-point Likert scale (1=poor to 5=well-developed).

Overall, 36 of 86 with treated or untreated ankyloglossia had parent-identified speech difficulties. Three-way comparison found statistically improved speech scores among treated versus untreated groups (mean 4.52 ± 0.61 vs. 3.60 ±0.63, p<0.0001) and between the control and untreated groups (mean 4.33 ± 0.77 vs. 3.60 ±0.63, p=0.01). No difference was found between the treatment and non-ankyloglossia control arms. The authors suggest that these results indicate that frenotomy can improve speech, and that speech outcomes for children after frenulum release are on par with those of children who never had ankyloglossia. However, little information is provided about why children in the untreated group did not receive frenotomy or why certain children were treated, nor were parents unaware of the treatment their child had received making recall bias a clear possibility.

A second poor quality retrospective cohort study recruited children who underwent frenotomy for ankyloglossia between ages of 2 days and 4 weeks and who were 4 to 8 years of age at the time of the study.26 These children were age-matched to children with untreated ankyloglossia whose parents reported a history of breastfeeding difficulties (nipple pain and/or latching difficulties) and to children with no history of ankyloglossia. All patients were administered the Articulation and Naming Test74 by two speech-language pathologists who were blinded to the group assignment. Each child's oral anatomy was systematically assessed from a standard oral motor evaluation test and scored.

In all, 23 children (17 males, 6 females) were divided into age-matched groups based on treatment status: treated (n=8; mean age 6.2 ± 1.8), untreated (n=7; mean age 6.2 ± 1.9), and controls (n=8; mean age 5.8 ± 1.9). All were found to have normal oral anatomy on examination. No significant differences were detected between treated and control patients in word, sentence, and fluent speech intelligibility. In contrast, children with untreated ankyloglossia had more articulatory errors than those who had been treated (14.5 ± 10 errors vs. 6.0 ± 4.2 errors).

Relevant case series examined different treatment methods including simple division with scalpel, scissors, and CO2 laser,51 frenuloplasty,42, 43, 54 and the addition of genioglossus myotomy.34 All studies reported positive outcomes and none reported significant harms, but as noted, these studies provide no comparative effectiveness data.

Comparison of Surgical Approaches

One RCT randomized children presenting to a cleft lip and palate-craniofacial clinic between 1999 and 2003 with a tight frenulum (<15 mm), an articulation or speech problem related to tongue tie, and/or age greater than 3 years to four-flap Z-frenuloplasty or horizontal-to-vertical frenuloplasty.22 Technical aspects of both surgical procedures were well described. Primary outcomes were changes from pre-operative to follow-up (>10 months) in frenulum length, tongue-protrusion measurements, and speech assessment. Both frenulum length and tongue protrusion were measured pre- and post-operatively by trained independent raters. Each patient had speech evaluations performed by two independent speech-language pathologists.

The study included 16 children with articulation problems, of whom 11 underwent four-flap Z-frenuloplasty (7 male, 4 female) and the remainder (2 male, 3 females) horizontal-to-vertical frenuloplasty. Ages were similar between treatment groups (Z-frenuloplasty: mean 5.7 ± 2.14 vs. horizontal-to-vertical: mean 5.56 ± 1.52). Pre-operatively, children in the Z-frenuloplasty arm had articulation difficulties rated as severe in six (55%) and moderate in five by the speech-language pathologists. Of the five patients in the horizontal-to-vertical frenuloplasty group, three (60%) were rated as severe and two (40%) as moderate. Ten of eleven children in the Z-plasty arm had two orders of magnitude improvement (i.e., severe to mild) and seven had complete resolution of articulation problems. In contrast, no patients in the horizontal-to-vertical group had two order of magnitude improvement or complete resolution. Two had one level improvement in articulation and three had none. Table 11 reports key outcomes in comparative studies.

Table 11. Comparison of surgical approaches.

Table 11

Comparison of surgical approaches.

KQ3. Benefits of Treatments To Prevent Social Concerns Related to Tongue Mobility

Key Points

  • Evidence is insufficient to assess the effects of intervention on social concerns related to tongue mobility.
  • Studies assessed different surgical interventions and different patient populations with widely varying age ranges.

Overview of the Literature

We identified nine studies that addressed either social concerns24, 33, 42, 51 and/or tongue mobility.6, 22, 26, 42, 43, 46, 51 Studies related to the effect of ankyloglossia on social concerns included one poor quality retrospective cohort24 and three case series33, 42, 51 that included outcome data for social concerns (e.g., drooling, embarrassment, kissing). The retrospective cohort was conducted in the United States24 and case series in the United Kingdom,51United States,42 and Brazil.33 None reported objective measurements of social concerns; instead each used parent- or patient-report to measure improvement. Subject age ranges varied significantly with the cohort study concentrating on 3 year old children24and case series including wider age ranges.33, 42, 51 The studies employed different surgical techniques and used different terminology without technical explanation: laser excision,6, 51 frenotomy,24, 33, 34 frenectomy,6, 33 and horizontal-to-vertical frenuloplasty.42, 43 Two studies described novel approaches to ankyloglossia repair, frenuloplasty with buccal mucosal graft,46 and four flap Z-frenuloplasty.22

Studies assessing the effect of ankyloglossia treatment on tongue mobility included a single RCT from the United States (rated as poor quality for outcomes related to tongue mobility),22a poor quality retrospective cohort study26 from Israel, and five case series: three from the United States42, 43, 46 and one each from the United Kingdom,51 and Brazil.6 One of two comparative studies objectively measured frenulum length and tongue protrusion,22 while the other used speech-language pathologists to rate children's tongue movement.26

Detailed Analysis

Social Concerns

One comparative study addressed the effect of ankyloglossia treatment on social concerns unrelated to speech.24 This retrospective cohort study enrolled 3-year old patients who received a frenotomy in infancy (n =71) and age- matched children with untreated ankyloglossia (n=15) and a control group of children without ankyloglossia (n=18). This study design and patient population is described in detail in KQ2 as it relates to feeding outcomes and in KQ2b with respect to speech outcomes. In short, parents were contacted in a telephone survey developed by a speech-language pathologist using a Likert scale to detect improvement in 1) difficult cleaning teeth with tongue, 2) difficulty licking outside of lips, and 3) difficulty eating ice cream.

Compared with individuals with non-treated ankyloglossia, those that were treated had significantly less difficulty cleaning the teeth with the tongue (p = 0.0006), licking the outside of their lips (p <0.0001) and eating ice cream (p= 0.0003). Similarly, control patients had significantly less difficulties with these tasks compared with untreated children (p<0.05). Unfortunately, the central tendency and variance from which these p-values were derived were not presented in the manuscript. Because this study was retrospective and included only parent report, both recall bias and confounding by indication are likely.

In one case series of older patients (mean age 29.8 ± 10.0 years), pre- and post-procedure patient survey was used to determine improvement.42 Seven of 15 participants reported embarrassment due to their ankyloglossia. In the six patients who elected to undergo frenuloplasty (mean age 17.3 ± 3.2 years), all reported improvement in tongue function in at least three of six areas which included: licking ice cream, licking lips, cleaning teeth, kissing, and playing a wind instrument. Another case series reported subjective improvement in oral hygiene (n=18/21) after laser frenectomy.51 Limiting these findings was the absence of pre-procedure status of these patients in these domains and how each was assessed. In addition to not including a comparison group of any type, case series are strongly affected by selection bias and are, by nature, not comparative studies.

Tongue Mobility

We identified two comparative studies that provided data on tongue mobility (Table 12).22, 26 One RCT enrolled 16 children (mean age 5.7±2.14) randomized to either four-flap Z-frenuloplasty or horizontal-to-vertical frenuloplasty.22 A thorough review of its study design is described in KQ2b in relation to speech outcomes. Authors measured frenulum length and tongue protrusion using a string to record the distance from the lower dentition to tongue tip during maximum protrusion of the tongue. The string was then transferred to a ruler for measurement in millimeters (mm). Three trained raters measured each patient's tongue protrusion.

Table 12. Outcomes of interventions for social concerns related to tongue mobility.

Table 12

Outcomes of interventions for social concerns related to tongue mobility.

The study reported improved tongue tip mobility in all 11 patients who underwent Z-frenuloplasty. The mean frenulum length in this group was 49.4 ± 16.6mm, which was significantly longer than pre-operatively (11.9 ± 6.1 mm, p<0.001). Thus, the mean gain in length was 37.5 ± 13.5 mm. In contrast, mean frenulum length for horizontal-to-vertical frenuloplasty was 22.6 ± 7.02 from 11.4 ± 3.36 mm, which was significantly longer, but less so than in the comparison group. Both groups were able to protrude the tongue past the inferior dentition. Mean gains in tongue protrusion for Z-frenuloplasty and horizontal-to-vertical frenoplasty were 36.2 ± 7.6 mm and 13.2 ± 2.6 mm, respectively. Measurements in both groups were significantly improved from baseline (p values <0.01).

The retrospective cohort study compared outcomes among children with ankyloglossia that was treated with frenotomy (n=8), untreated children with ankyloglossia (n=7) and a control group without a history of ankyloglossia (n=8). Design of this cohort is summarized as part of KQ2b in relation to speech outcomes. In terms of tongue mobility, speech-language pathologists examined each child's oral anatomy and tongue movements by performing 10 different exercises as part of a standardized oral motor evaluation test: protrusion, elevation, left and right movements, licking of lower and upper lips, clicking, touching hard palate, elevation of mid-tongue toward the hard palate). Each task was scored from 0 (normal) to 1 (for distorted movement or inability to perform task). Untreated individuals had more difficulties in tasks of tongue movement (11.4 ± 7.6 uncompleted tasks) compared with treated children (3.7 ± 4.2). Children with no history of tongue-tie had the lowest rate of uncompleted tasks (1.2 ± 1.6).

Five case series reported improvements in mobility and elevation.6, 42, 43, 46, 51 Two case series assessing the safety of CO2 laser (total n=36) concluded that it was safe and effective alternative to conventional release.6, 51 Both studies reported improvement in tongue mobility after repair but one6 described greater improvement if the patient received speech therapy prior to release. A third case series in participants (mean age 8 at surgery, 15 with ankyloglossia and two with short labial frenulums) reported improvements in tongue mobility in the 3-4 months following surgery in an unspecified number of participants.46 For most of these studies there was minimal explanation of expectations for normal tongue mobility. For the few studies with objective measurements, the total sample size (n= 52) was too small and the ages too varied to establish normative data.

KQ4. Benefits of Simultaneously Treating Ankyloglossia and Concomitant Lip-Tie

We identified no studies that presented outcomes specifically for infants or children treated simultaneously for ankyloglossia and lip tie. One study reported that some of the participants also had lip-tie, but the outcomes were not presented separately for this subset.31

KQ5. Harms of Treatments for Ankyloglossia or Ankyloglossia With Concomitant Lip-Tie in Neonates, Infants, and Children Through Age 18

Key Points

  • Most studies that reported harms information explicitly noted that no significant harms were observed (n=17) or reported minimal harms, most commonly self-limited bleeding, which would be expected with oral surgery.

Overview of the Literature

We identified 46 studies addressing harms (31 RCTs, cohort studies, or case series and 15 case reports). One RCT conducted in the United Kingdom reported minor harms of surgery and need for reoperation.8 A single retrospective cohort study conducted in the United States reported harms (scarring).25. Twelve of 33 case series reported minor harms: four from the United States,3, 31, 42, 46 four from the United Kingdom,29, 49-51 one from Brazil,6 one from Finland,48 one from Israel,52and one from China37 Seventeen studies (13 case series, four RCTs) specifically noted that no harms were observed. We included case reports specifically to address harms; details of the 15 case reports yielding harms data are in Appendix G.

Detailed Analysis

Data on harms were only available for studies of surgical interventions. Given the paucity of comparative data on this topic, we also sought case series and case reports to ensure that we captured possible evidence of harms associated with treatment. Of six RCTs, four reported that there were no harms, one was silent on the subject, and one study reported that 64 percent of participants had a small white patch at the base of the frenulum (likely healing slough) that took approximately 7 days to heal and four of 99 (4%) required a reoperation.8 Among the three cohort studies, two did not address harms. In the one cohort study that reported harms, eight of 302 (2.6%) participants had a recurrence due to scarring or incomplete clipping that required reoperation.25 Harms were described in 11 of 33 case series. Minor bleeding occurred in six and infant distress/pain was described as affecting 2 of 36 infants (5.6%) in another.49 Rates of reoperation ranged from 0.1 percent37 to 27 percent31, with a need for reoperation occurring in a total of five case series. One case series reported mild wound cicatrization following frenuloplasty involving use of buccal mucosa grafts.46 Another case series reported no complications after CO2 laser excision, but in patient surveys two of 21 disagreed with the statement “no pain” and one of 21 disagreed with the statement “no blood.”51

To ensure that we did not miss potential harms of surgical intervention, we searched for case reports of harms and identified 15,58-72 details of which are presented in Appendix G. Among 15 case reports (two of which reported multiple cases58, 72), there were two cases of surgical site infection, three cases of reoperation and four reports of swelling and pain. One case reported post-surgical mucocele in a 12-year-old patient.59 Only two cases, in Nigeria, sustained harms to the degree that they were hospitalized for bleeding; in these cases, the authors indicated that the procedure was done by inexperienced clinicians and that this likely accounted for the excessive bleeding.60

Gray Literature

Conference Abstracts

We searched for conference paper and poster abstracts from recent national and international societies and associations related to pediatrics, nursing, breastfeeding medicine, lactation, otolaryngology, dentistry, orthodontics, speech and hearing. Conference abstracts predominantly addressed prevalence of ankyloglossia, investigation into incidence of anterior versus posterior rates of tongue-tie, rates of surgical treatment interventions, and case reports of successful surgical interventions to address breastfeeding issues. Results reported in abstracts generally aligned with our findings, with abstracts noting maternally reported improvements in breastfeeding effectiveness and nipple pain (Appendix H).

Dissertations and Theses

Although we did not identify any relevant dissertations in our search, one TEP member who recently completed a master's degree at the University of Liverpool allowed us to use findings from her unpublished thesis. She conducted a retrospective survey of parents in the United States of children who had had frenotomy for ankyloglossia either before or after age 12 weeks (Table 13).75 The survey included questions related to breastfeeding effectiveness and pain, supplemental bottle feeding, feeding with solid food, knowing and pronouncing words, and oral hygiene and was sent to parents of children treated between 2006 and 2011 at a single institution. Findings supported the published literature in reporting improvements after frenotomy in maternally reported outcomes. This study adds to the published literature in assessing early versus late outcomes, finding improved outcomes associated with early treatment. Because it is not a published study, we did not include it in our strength of evidence assessment but provide the results here.

Table 13. Outcomes reported in unpublished thesis.

Table 13

Outcomes reported in unpublished thesis.

Findings included data from 125 children with ankyloglossia who received frenotomy, 51 of whom were treated before 12 weeks of age (early treatment) and 74 who were treated after (late treatment). All children in the early treatment group were diagnosed within 90 days of birth, while 43 of the late treatment arm were diagnosed by 90 days, eight by 180 to 365 days, and 15 at >365 days of age.

Breastfeeding Outcomes

Children in the early treatment group had a longer duration of breastfeeding compared with the later treatment group. Within the early treatment group, about a third either did not have a latch issue or it was resolved prior to frenotomy, while in 45 percent of the cases the issue was resolved with frenotomy. Nonetheless, in almost a quarter (23.5%), latch issues led to abandonment of breastfeeding. In the late treatment group, however, most (82%) either never had a latch issue or it resolved before the frenotomy, with only 1.4 percent having latch resolved via frenotomy. Pain was resolved after frenotomy in about a third (33.3%) of the early treatment group, whereas about half either did not have pain or it had resolved prior to frenotomy in this group. Among infants diagnosed and treated late, mothers reported that most (89%) did not have pain or that it resolved prior to frenotomy.

Other Feeding Outcomes

In terms of latching to a bottle, in the early treatment group, 75.5 percent either had no issue or had it resolve prior to treatment. Twenty-four percent had problems with latch to a bottle resolved with frenotomy.

Speech Outcomes

Speech issues were unique (as expected) to children with a later treatment. Among these children, pronunciation issues were resolved in in 43.1 percent (n=31/72) of the cases.

Other Outcomes

In this study, no children in the early frenotomy group had oral hygiene issues, compared to 15 in late treatment arm. Issues resolved with frenotomy in 18.1 percent (n=13/122) of children in this group.