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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].

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Discussion

We identified 57 published studies for this review, six of which were randomized controlled trials (RCTs), three were cohort studies, and the remainder case series (n=33) and case reports (n=15). The analysis and discussion concentrate on comparative studies (RCTs and cohorts), as these studies were used for strength of evidence assessment. Case series were included in the results only to ensure that the full range of available literature is made available to the end users of this report. Harms were reported from all included studies as well as a specific search for case reports.

Three RCTs were assessed as good7, 8, 20 and one as fair23 quality for outcomes related to breastfeeding effectiveness and associated maternal pain. One RCT was rated as poor quality for breastfeeding effectiveness and pain outcomes.21 One RCT addressing tongue protrusion, frenulum length, and speech outcomes was rated as poor quality for those outcomes,22 and we rated one RCT as fair quality for measures of bottle feeding.23 We rated all three cohort studies as poor quality.24-26

We assessed the quality of harms reporting in RCTs and cohort studies as poor and as good in four case series49-52 and poor in 23.3, 6, 27-29, 31, 32, 34, 35, 37-40, 42-45, 48-51, 54, 56 We also included data from one unpublished thesis (not quality scored).

Key Findings and Strength of Evidence

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

Key Findings

Overall, three good7, 8, 20 and one fair23 quality RCTs assessed whether treatment of ankyloglossia improved breastfeeding effectiveness. While only one of three RCTs that used blinded independent observers found significantly improved breastfeeding effectiveness among frenotomized infants immediately post-procedure,7 maternally reported breastfeeding effectiveness was significantly improved in the treated group compared with untreated in two of two RCTs that evaluated it either as a primary23 or secondary20 outcome. A third RCT evaluated the mother's breastfeeding self-efficacy and found a significant improvement from baseline in the frenotomy group 5-days post-procedure.8 In all, there is some evidence that maternally reported breastfeeding outcomes improve. Comparative data are lacking to assess the durability of effects.

These same studies had disparate findings about whether frenotomy decreased maternal nipple pain during breastfeeding. Only the RCT performed on infants at 6 days of age showed a significant reduction in maternal pain.7 Those performed on infants a few weeks older did not report either an immediate20 or 5-day8 reduction in pain. The difference between earlier frenotomy and later frenotomy on nipple pain may relate to cumulative trauma on the breast from several additional weeks with inefficient latch from tongue-tied infants.

Strength of the Evidence

Few comparative studies have addressed the effectiveness of surgical interventions to improve breastfeeding outcomes. Mothers consistently reported improved breastfeeding effectiveness, but outcome measures were heterogeneous and most were short term. Future studies could provide additional data to confirm or change the measure of effectiveness; thus we consider the strength of the evidence (confidence in the estimate of effect) to be low at this time.

We also considered the strength of the evidence to be low for an immediate reduction in nipple pain. Improvements were reported in the current studies, but additional studies are needed to confirm and support these results. Only one poor quality cohort study addressed effects on the length of breastfeeding; thus, we considered the strength of the evidence to be insufficient (Table 14).

Table 14. Strength of the evidence for studies addressing surgical approaches for ankyloglossia and breastfeeding outcomes.

Table 14

Strength of the evidence for studies addressing surgical approaches for ankyloglossia and breastfeeding outcomes.

KQ2a. Benefits of Treatments To Mitigate Feeding Sequelae

Key Findings

We identified three studies examining feeding outcomes other than breastfeeding: one RCT,23 one poor quality retrospective cohort study,24 and one case series.35 All three studies were single center or single surgeon studies. Bottle feeding and ability to use the tongue to eat ice cream and clean the mouth improved more in treatment groups in comparative studies. Supplementary bottle feedings decreased over time in the case series.

Strength of the Evidence

With only two comparative studies, both with significant study limitations, 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. The studies used different populations and measured different outcomes (Table 15).

Table 15. Strength of the evidence for studies addressing surgical approaches and feeding outcomes.

Table 15

Strength of the evidence for studies addressing surgical approaches and feeding outcomes.

KQ2b. Benefits of Treatments To Prevent Other Sequelae

Key Findings

Speech concerns were the second most prevalent topic in the ankyloglossia literature, after breastfeeding. A speech-language pathologist measured speech outcomes in two studies22, 26 with the third using parent report.24 No studies included data related to sleep disordered breathing, occlusal issues and dysphagia in the non-breastfeeding child. Two cohort studies attempted to assess the effectiveness of frenotomy, 24, 26 and one compared two surgical approaches to frenotomy. 22

Two poor quality cohort studies24, 26 reported an improvement in articulation and intelligibility with ankyloglossia treatment, but benefits in word, sentence and fluent speech were unclear. The one poor quality RCT reported improved articulation in patients treated with Z-frenuloplasty compared to horizontal-to-vertical frenuloplasty.22 Numerous non-comparative studies reported a speech benefit after treating ankyloglossia; however these studies primarily discussed modalities, with safety, feasibility or utility as the main outcome, rather than speech itself.33, 34, 37, 42, 43, 47, 48, 51

Strength of the Evidence

Given the lack of good quality studies and limitations in the measurement of outcomes, we considered the strength of the evidence for the effect of surgical interventions to improve speech and articulation to be insufficient (Table 16).

Table 16. Strength of the evidence for studies addressing surgical approaches and other outcomes.

Table 16

Strength of the evidence for studies addressing surgical approaches and other outcomes.

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

Key Findings

Only one poor quality comparative, retrospective cohort study assessed outcomes related to social concerns other than speech.24 It reported significantly improved ability to clean teeth with tongue, licking outside of lips, and eating ice cream in the treatment group compared with untreated participants. The intermediate outcome of improved tongue movement or mobility after ankyloglossia repair was assessed in two comparative studies—one poor quality RCT22 and one poor quality cohort study.26 The RCT assessed tongue mobility using two different surgical techniques for treating ankyloglossia and found that both approaches significantly improved tongue mobility, but that Z-frenuloplasty was superior.22 In the cohort study, individuals with untreated ankyloglossia had the worst tongue mobility followed in order by children with treated ankyloglossia, and those with no history of ankyloglossia.26

Strength of the Evidence

With only one poor quality comparative study, strength of the evidence related to the ability of treatment for ankyloglossia to alleviate social concerns is currently insufficient. Also, with only three comparative studies with small sizes and limitations in the measurement of outcomes related to tongue mobility, we considered the strength of the evidence for the effect of surgical interventions to improve the short-term outcome of mobility to be insufficient (Table 17).

Table 17. Strength of the evidence for studies addressing surgical approaches and social concerns related to tongue mobility.

Table 17

Strength of the evidence for studies addressing surgical approaches and social concerns related to tongue mobility.

KQ4. Benefits of Simultaneously Treating Ankyloglossia and Lip-Tie

We did not identify any studies addressing this question.

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

Key Findings

We identified all possible harms reported within comparative studies and case series that potentially provided effectiveness data. We also sought case reports of harms. With this approach, we reported harms from 51 studies that reported that they had looked for harms, either reporting actual harms or specifically indicating that they found none. These included five RCTs, one cohort study, 28 case series, and 15 case reports. 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 24.3, 6, 27-48Most studies that reported harms information explicitly noted that no significant harms were observed (n=18) or reported minimal harms. Among studies reporting harms, bleeding and the need for reoperation were most frequently reported. Bleeding was typically described as minor and limited. Few studies described what specific methods they used to collect harms data.

Strength of the Evidence

We considered the strength of the evidence for minimal and short-lived bleeding as a harm of surgical interventions as moderate based on an expanded search for harms reports in addition to the comparative data. We considered the strength of the evidence for reoperation and pain as harms to be insufficient given the small number of studies that included these outcomes (Table 18).

Table 18. Strength of the evidence for studies addressing harms of surgical approaches.

Table 18

Strength of the evidence for studies addressing harms of surgical approaches.

Findings in Relationship to What Is Already Known

Few recent reviews assessed outcomes of ankyloglossia treatment,2, 5, 76, 77 and our findings generally align with those prior reviews, concluding that current evidence is drawn from a small literature base with inconsistent findings related to the benefits of ankyloglossia treatments for increasing breastfeeding effectiveness or reducing maternally reported nipple pain. In a review focused solely on frenotomy and breastfeeding, the authors rated most of the seven studies evaluating frenotomy as poor quality (mean score of 24.4, range 9-40 on a 47-point scale).76 Studies included one RCT, and all used different outcome measures to assess effects of frenotomy. Outcomes (breastfeeding mechanics, nipple pain, rate of breastfeeding, sucking, weight gain) all improved post-procedure, and no studies reported significant adverse effects. Another review and meta-analysis addressing frenotomy and breastfeeding included four RCTs and 12 observational studies and concluded that moderate quality evidence supports the effectiveness of frenotomy for improving latching and nipple pain.77 The risk ratio for overall improvement in latching was 2.88 (95% confidence interval [CI]: 1.82 to 4.57) in meta-analysis of four RCTs, and the mean difference in pain scores was -5.10 (95% CI: -5.60 to -4.59) in meta-analysis of three RCTs. The review noted that no major complications were reported in the studies analyzed.

In a review addressing diagnosis and treatment and including 10 studies assessing effects of treatment on breastfeeding outcomes, breastfeeding mechanics and related outcomes typically improved.2 Four studies of tongue mobility and three of speech problems also reported improvement. The review notes insufficient evidence related to choice of procedure, timing of procedure, or surgical versus conservative management; however, the investigators did not include any quality metrics for included studies.

A fourth recent review assessed outcomes related to breastfeeding and speech.5 The 20 studies included ranged from level 4 case series to randomized controlled trials, and concluded that there is both objective and subjective evidence that frenotomy benefits breastfeeding (facilitated breastfeeding, enhanced milk transfer to the infant, and contributed to protecting maternal nipple and breast health), but tempered this by recognizing that there were a limited number of studies available with high quality evidence. Outcomes in four studies addressing speech articulation reported few definitive improvements following treatment. This review did not evaluate non-surgical management or broader outcomes.

Applicability

We set inclusion criteria intended to identify studies with applicability to newborns, infants, and children with ankyloglossia. Studies differed in terms of study population and outcome measures. Most studies were non-comparative, and lack of direct comparisons of treatment options further hinders the ability to understand what findings will best extrapolate to a specific newborn or infant or decisions about care protocols. Overall the data on breastfeeding and maternal breast pain that are available may be applicable to newborns with ankyloglossia with concomitant feeding problems. There is no evidence to suggest that the data would be applicable to infants with ankyloglossia who do not present with feeding problems. Appendix I contains applicability tables for individual KQs.

Applicability of Studies With Breastfeeding Outcomes

Newborns referred for treatment of ankyloglossia were born primarily at tertiary care centers and recognized as having difficulty with breastfeeding concomitant with ankyloglossia. The frenotomy procedure itself is not technically difficult and is likely performed similarly across birthing sites; what is less clear is whether the diagnostic criteria by which the decision is made to perform the procedure are similar across practice settings. Moreover, newborns of mothers not choosing to breastfeed may not be recognized as having and/or diagnosed with ankyloglosssia as breastfeeding difficulties were used as an indicator to evaluate for ankyloglossia. Interestingly, two studies7, 8 reported that all patients had lactation consultation prior to enrollment without significant improvement in feeding. Arguably, this limits the applicability of their results to newborns that had failed to improve adequately with such consultation.

In these studies, various clinicians were involved in making the ankyloglossia diagnoses; however, assessment of breastfeeding difficulty and diagnostic criteria for ankyloglossia were not universally described. Lack of a consistent objective measure to define and classify this condition may limit the reproducibility of findings. Furthermore, patients in these studies were between a median 6 days of age7 and up to a mean 33 days of age (range 6 to 115) in another study.20 Applicability to findings in older infants cannot be gleaned from these data; nor can durability of results.

Frenotomy was the only intervention employed in the good quality RCTs.7, 8, 20 However, the specifics of the procedure were variably reported. As such the degree of posterior extension of the frenulum incision was not clearly defined and appears to be at the discretion and clinical expertise of the clinician. Also, the severity of the ankyloglossia was inconsistently reported, making inter-study generalizations difficult and, more importantly, limiting the broader applicability of findings.

The comparators used were sham surgery7, 20 and usual care.8 These outcomes are identical except in regards to blinding and outcome assessment. Both no intervention and sham surgery are perhaps misnomers, however, since these infant-mother dyads underwent usual care, which could include, but is not limited to, lactation consultation, supportive care, and bottle-feeding advice. Finally, there is insufficient evidence from available literature to assess the applicability of frenotomy on durability of breastfeeding.

Applicability of Studies With Other Feeding Outcomes

Only one study with comparative poor quality retrospective cohort data addressed other feeding outcomes.24 The study's intervention group received frenotomy for ankyloglossia, which was identified within the first month of life, and was compared to dyads who were also offered, but declined, frenotomy for the same indication in the same time period. Although this is a common decisional dilemma for parents of infants with congenital ankyloglossia, in usual clinical care, surgical intervention is not considered unless congenital ankyloglossia co-occurs with breast- or other feeding problems. Furthermore, there are several biases inherent in this treatment decision. First, those with “worse” ankyloglossia are more likely to get treated. Second, mothers who more strongly want to breastfeed may opt for division. Mothers who would rather pump or bottle feed with formula would more likely chose observation. Third, practitioners' presentation of the evidence may sway the decision, thus perpetuating their personal bias about effectiveness of frenotomy on improving breastfeeding and reducing maternal pain. Additionally, the study was conducted in an academic medical center in large, urban area with ankyloglossia severity graded by pediatric otolaryngologists. Therefore, applicability of its findings and observations may not translate to other care environments (i.e. community hospital, rural) and many usual clinical care settings may not include practitioners from this sub-specialty, instead relying more on pediatricians, lactation consultants, family practitioners, or dentists.

Applicability of Studies With Speech Outcomes

Comparative studies providing data on speech outcomes were all rated as poor quality and included a randomized controlled trial22 and two retrospective cohort studies.24, 26 The RCT compared two different frenuloplasty approaches for treatment of children of a mean age of approximately 6 years with a tight frenulum effecting articulation or intelligibility22 and found that children treated with either four-flap Z-frenuloplasty and horizontal-to-vertical frenuloplasty had significant improvement in articulation as judged by trained speech-language pathologists. Applicability of these findings is limited due to the small sample size, inadequate characterization of candidate children, and that specialist pediatric craniofacial surgeons performed these surgeries at an urban tertiary care center. “Usual sites” where ankyloglossia is diagnosed and treated would have a difficult time extrapolating these findings considering the limitations.

Similarly, the cohort studies were performed solely in urban tertiary care centers. One assessed outcomes on 3-year old children treated for ankyloglossia as neonates compared to those who had untreated ankyloglossia, and a control group without a history of ankyloglossia.24 Pediatric otolaryngologists made the diagnosis using standardized diagnostic criteria. The reason that infants presented for treatment of ankyloglossia was not identified. Further limiting the applicability is that these patients were all cared for at a tertiary care facility and outcomes were assessed using a non-validated parent reported telephone survey. Thus, there was no objective evaluation of speech. Parents of children with ankyloglossia would have a higher index of concern for speech issues than those whose children never had been diagnosed with tongue mobility restriction. The second poor quality retrospective cohort with a relatively small sample size (n=23) of children a mean of roughly 6 years of age that were similarly divided into those with treated ankyloglossia, untreated ankyloglossia, and a control group.26 It was performed at a tertiary care facility in an Israeli urban center. Unfortunately, its applicability is limited similarly to that previously described except that speech-language pathologists objectively assessed speech using a standardized assessment tool. Both retrospective studies lacked explanations about the rationale for initial surgical intervention or reason parent chose not to intervene.

Applicability of Studies With Social Outcomes

The population studied in the question of benefit of ankyloglossia repair for social concerns included children and adults with wide variation in ages. Studies were rated as poor quality, were retrospective, and few in number. Outcomes in one were assessed by parental report and subject to recall bias24 and social outcomes assessed were limited to licking lips, cleaning teeth with tongue and eating ice cream. Thus, the social concerns or implications of these issues are unclear. No other comparative study considered social concerns. In addition, at least two case series did consider the impact of ankyloglossia on kissing and playing a wind instrument42 and drooling and oral hygiene.33 Limiting these findings was the absence of preprocedure 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. Moreover, patients were selected either by retrospective chart review or as they presented to otolaryngology clinics. Only surgical interventions were studied and no two studies measured the same outcomes. Typically, social concerns were measured as a secondary outcome. The setting was typically the outpatient setting, within academic medical centers.

Implications for Clinical and Policy Decision Making

A small body of evidence suggests that frenotomy may be associated with mother-reported improvements in breastfeeding and possibly reduction in nipple pain, when feeding difficulties are present. At this point, the evidence is fairly inconclusive on effectiveness for most outcomes. However, there does seem to be stronger evidence that harms are minimal to none, Thus, given the mixed evidence, clinicians and families will likely need to make individual decisions about pursuing intervention for ankyloglossia-related feeding and speech production difficulties. Importantly, no research evidence exists to assess any non-surgical interventions, so clinical and policy decision making will necessarily occur in the absence of evidence for nonsurgical interventions.

Limitations of the Comparative Effectiveness Review Process

This review included only studies published in English. However, our scan and review of non-English references revealed that high percentage of non-eligible items. Specifically, we determined that 502 of the 520 foreign language references identified in MEDLINE (search conducted in February 2014) would be excluded based on our criteria. Of the 18 potential includes, six appeared, from the information in the abstract and/or title to be eligible for inclusion; 12 did not include abstracts or sufficient information from the title to make an inclusion decision. Two of these appeared to be case reports and neither gave clear indications on whether harms of interventions were addressed. Given the high percentage of non-eligible items in this scan (97%), we feel that excluding non-English studies did not introduce significant bias into the review.

While we focused the review on comparative studies (studies including an intervention and a comparison group), we provide summaries of case series data to supplement the comparative findings given the small number of studies addressing ankyloglossia interventions. We further specifically sought case reports of any harms associated with ankyloglossia intervention. This approach may provide particularly useful information about harms as we found little evidence of serious harm of surgical interventions, though harms reporting was limited.

Limitations of the Evidence Base

The evidence base for the benefits of treatment in ankyloglossia is very limited. Overall, the evidence base consists of a few small studies that use varied outcomes and provide little information to adequately characterize participants. Infants vary in age at treatment from 6 to 33 days and in reasons for presentation. Studies are focused on neonates and infants who present because of breastfeeding difficulties, and while improving breastfeeding success is an important goal, by definition, this means data are unavailable on infants with ankyloglossia but without feeding difficulties in infancy. The degree to which these infants are likely to go on to develop either feeding, speech or social impediments is inadequately understood. No study effectively assessed mid- and long-term comparative outcomes of frenotomy making it difficult to predict whether mother-reported improvements early in infancy led to longer term breastfeeding. In particular, there is a lack of evidence on significant long-term outcomes such as exclusive breast-feeding at six month of age or at one year of age, growth and other measures of health outcomes. Furthermore, studies are entirely lacking that compare surgical intervention to well-described skilled lactation consultation and other breastfeeding report. Although complementary and alternative methods of care are used in some practices, no studies are available. In addition, the literature base may be subject to publication bias. Most controlled studies reported only positive outcomes, and we identified no negative trials.

Finally, we found no comparative effectiveness data on nonsurgical interventions, although they are in use in clinical care, and in surgical studies, case series predominated, providing little comparative data.

Research Gaps

Breastfeeding Outcomes

Future studies should consider direct comparisons of alternative treatments as currently available literature only addressed the comparison of frenotomy to sham. In order to conduct these studies, it would be helpful if the field could agree upon on standardized approach to identifying and classifying ankyloglossia; this would also improve our ability to synthesize the data across studies.

A critical unknown at this point is a good description of the natural history of ankyloglossia by severity, including long term risk of feeding, social and speech production difficulties. Studies should also consistently report measures of severity.

Given variation in outcomes that may be associated with earlier versus later frenotomy, future studies should assess timing of frenotomy to determine whether more significant reduction in maternal pain is achievable by earlier treatment and whether mothers are more apt to breastfeed longer if done earlier.

A final gap in research is in understanding the durability of outcomes. Good quality comparative studies evaluated breastfeeding effectiveness immediately7, 20 or within 5 days of frenotomy.8 However, none adequately assessed whether effectiveness and other outcomes (e.g., changes in maternal nipple pain) were maintained months or, if appropriate, years later. Longer term follow up of both treated infants and controls is needed.

Other Feeding Outcomes

Because there is such a paucity of available data on other feeding outcomes, this entire research question represents a gap and a potential area for future research.

Speech and Other Outcomes

Similarly, substantially more research is needed to consider whether treatment of ankyloglossia in infancy prevents future speech production difficulties as well as whether treatment later in life with frenotomy leads to improvement when speech problems arise. To conduct this research effectively, methods for evaluating risk and presence of speech production difficulties will need to be standardized, and outcomes agreed upon. Understanding of the natural history of speech concerns in children with ankyloglossia is lacking as are comparative studies that utilize standardized measurement tools for speech outcomes.

Social Concerns Related to Tongue Mobility

No standard definitions of tongue mobility or established norms for mobility exist, and further research is needed to determine such parameters. Social concerns are difficult to measure objectively so there will likely always be a subjective component to social outcomes. Larger studies that assess both treated and untreated individuals could provide useful data to minimize the potential bias found in the existing literature. Similarly, future research in objective measurement tools, or validated self-report tools, is needed.

Harms Reporting

Few studies prespecified harms or provided details of harms collection. Harms were not systematically reported, and therefore there may be substantial underreporting. Minor, limited bleeding and need for re-operation were reported in some studies, but methods for collecting harms in studies overall were poorly reported. Future studies would benefit from explicit description of methods for harms collection, including estimating blood loss, and assessment and explicit reporting.

Conclusions

A small body of evidence suggests that frenotomy may be associated with improvements in breastfeeding as reported by mothers, and potentially in nipple pain, but with small studies, inconsistently conducted, strength of the evidence is low to insufficient, preventing us from drawing firm conclusions at this time. Research is lacking on nonsurgical interventions as well as on outcomes other than breastfeeding, particularly speech and dental outcomes. In particular, there is a lack of evidence on significant long-term outcomes such as exclusive breast-feeding at six month of age or at one year of age, growth and other measures of health outcomes. Harms are minimal and rare; the most commonly reported harm is self-limited bleeding. Future research is needed on a range of issues, including prevalence and incidence of ankyloglossia and problems with the condition. The field is currently challenged by a lack of standardized approaches to assessing and studying the problems of infants with ankyloglossia.

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