Untreated Ankyloglossia: A Broader Perspective

In this issue of Pediatrics, Raol et al1 report on a prospective mixed-methods study conducted at a large urban tertiary care hospital in Hyderabad, India. Their study enrolled 476 maternal-infant dyads within 48 hours of birth and compared breastfeeding outcomes and perceived breastfeeding difficulties among 366 infants who did not have ankyloglossia (“tongue-tie”) and 110 who did, based on clinical assessment with the Bristol Tongue Assessment Tool (BTAT).2 Their study is unique in that none of the infants had a frenotomy or other surgical treatment of their ankyloglossia, and exclusive breastfeeding was assessed at 2–4 weeks, 3 months, and 6 months after delivery. Surprisingly, there were no differences in rates of exclusive breastfeeding at any time point, including at 6 months (82.3% [no ankyloglossia] vs 73.5% [assessed with ankyloglossia]; P = .25), and no differences in infant growth velocity at any time point. The only factor associated with lower odds of exclusive breastfeeding at 6 months after delivery was maternal concern about milk supply at the 2–4 weeks phone call (odds ratio, 0.22; P < .003). In structured interviews of 36 mothers with a spectrum of experiences in this study, reports of nipple pain (commonly associated with ankyloglossia3) did not differ between those with and without either early breastfeeding cessation or ankyloglossia.To provide context for these intriguing study results, the American Academy of Pediatrics clinical report, the Academy of Breastfeeding Medicine position statement, and the American Academy of Otolaryngology–Head and Neck Surgery Foundation clinical consensus statement provide helpful insight.4–6 These current reports agree that evidence is very limited with respect to assessment and treatment of ankyloglossia. Although the frequency of ankyloglossia diagnosis has increased in the United States and globally,3,5 none of the numerous assessment tools are validated,4 published rates vary widely (0.02% to 20%) and by country,7 and disagreements about surgical vs expectant management and the optimal type of surgical intervention persist, mainly because of a paucity of high-quality evidence.4–6The study by Raol et al is notable because it addresses weaknesses of prior work. Not only was it conducted in a setting in which breastfeeding is the norm (evidenced by high rates of exclusive breastfeeding), but the study enrolled a large number of dyads, assessed exclusive breastfeeding, included long-term follow-up (through 6 months), and did not include a surgical treatment group, permitting a long view of ankyloglossia natural history that is missing from most studies.Neither we nor the authors are suggesting that infants with ankyloglossia should never be treated with frenotomy, and some key study strengths and limitations help frame interpretation of the results. A notable study strength was the breastfeeding environment, in which study mothers achieved high rates of exclusive breastfeeding at 6 months, suggesting societal and family support were optimal; for comparison, just 27.2% of US mothers are exclusively breastfeeding at 6 months.8 This difference illuminates the potential impact of cultural context on “biological” results.The main study limitations are (1) how the diagnosis of ankyloglossia was made and (2) the range of ankyloglossia severity in the diagnosed group, which limits comparisons with control infants. Additionally, although study families experiencing feeding difficulties received lactation counseling from a breastfeeding specialist, a comprehensive assessment with a functional breastfeeding evaluation was not completed for each ankyloglossia-diagnosed infant or, if completed, was not documented. Given the lack of high-level evidence, there is no agreement on a standardized tool for tongue-tie identification,4–6,9 and the tool and cutoff value selected by Raol et al may have led to a higher-than-expected ankyloglossia rate.10 The ankyloglossia rate in this study was 23%, which is meaningfully higher than the expected rate of approximately 2.8%-10.7%.6 Lower scores on the BTAT indicate greater severity of ankyloglossia; hence, in the context of this study, a BTAT cutoff of no more than 3 or 5 (vs <8) might have been more clinically useful and consistent with other studies using the BTAT.11,12In the study by Raol et al, just 2 of 110 study infants (1.8%) had “severe” ankyloglossia (BTAT scores 0–3), and 33 (30%) had BTAT scores less than or equal to 5, described as “impairment of tongue function” (this includes the 2 infants with scores 0–3). The remaining 77 infants (70%) diagnosed with ankyloglossia with BTAT scores 6–7 would likely be expected to breastfeed well with time and support. Thus, in the study by Raol et al a more informative comparison group for the infants without ankyloglossia might be the 33 infants (7%) with BTAT less than or equal to 5, aligning with prior work.11,12 This study therefore highlights the need for larger-scale, adequately powered studies to provide the necessary number of patients to fully evaluate the implications of untreated severe ankyloglossia.Additional clinical factors identified through a full breastfeeding history and feeding evaluation are important to consider when making decisions on ankyloglossia treatment and outcomes.4,5 Raol et al identified no difference in exclusive breastfeeding rates or growth trajectory, yet a full breastfeeding evaluation provides context not available with BTAT screening. Clinical observations find that some babies with ankyloglossia will feed more frequently, perhaps to counteract the fatigue of the tongue restriction, and gain weight appropriately. That increased feeding frequency may work for a parent accepting a frequent feeding pattern and if it is culturally appropriate for the family. In the setting of scheduled, less frequent feeds, the same infant may not gain appropriately. Infants with restricted tongue movement and ankyloglossia may have trouble maintaining latch. Sometimes the breastfeeding parent of an infant with ankyloglossia will hold the breast while feeding so the baby is less likely to slide off, and the latch adjustments help compensate for the ankyloglossia. This clinical context highlights the importance of breastfeeding support and may inform management. Either of these modifications, or others, might or might not have helped the 2 infants in this study identified as having “severe” ankyloglossia; follow-up with clinical detail for these infants could complement the qualitative aspects of this study.Integrated, multidisciplinary assessment and care remain important as we work to expand research on the evaluation and management of ankyloglossia. Raol et al reference several studies demonstrating decreases in frenotomy rates when a multidisciplinary team is involved in the care of infants with ankyloglossia.11,13,14 We agree with the study authors that, “…when focus is only placed on ankyloglossia, a host of other causes for early breastfeeding cessation may be missed…” and that multidisciplinary management helps “focus on all potential causes and not only on ankyloglossia.”There are several important messages here. For infants with ankyloglossia, more breastfeeding support is essential for the dyad, with a full breastfeeding evaluation and functional assessment, followed by shared decision-making that incorporates available evidence and close clinical follow-up. For clinician scientists, key research can focus on better evaluation and comparison of tools for identifying ankyloglossia,9 integration of a full clinical breastfeeding assessment into affected infants’ care, and a continued focus on long-term follow-up.Until more is known, a 2- to 3-week period of watchful waiting with medical and lactation support, remaining mindful of the increased risk of weaning if difficulties persist,15 may serve to “slow the trains” and give each dyad the advantage of time revealed in this thought-provoking study by Raol et al.