Ankyloglossia is a congenital condition characterized by an abnormally short, thickened, or tight lingual frenulum that restricts tongue mobility. The objective of this study was to systematically review literature on surgical and nonsurgical treatments for infants with ankyloglossia.
Medline, PsycINFO, Cumulative Index of Nursing and Allied Health Literature, and Embase were searched up to August 2014. Two reviewers independently assessed studies against predetermined inclusion/exclusion criteria. Two reviewers independently extracted data regarding participant and intervention characteristics and outcomes and assigned quality and strength-of-evidence ratings.
Twenty-nine studies reported breastfeeding effectiveness outcomes (5 randomized controlled trials [RCTs], 1 retrospective cohort, and 23 case series). Four RCTs reported improvements in breastfeeding efficacy by using either maternally reported or observer ratings, whereas 2 RCTs found no improvement with observer ratings. Although mothers consistently reported improved effectiveness after frenotomy, outcome measures were heterogeneous and short-term. Based on current literature, the strength of the evidence (confidence in the estimate of effect) for this issue is low. We included comparative studies published in English. The evidence base is limited, consisting of small studies, short-term outcomes, and little information to characterize participants adequately. No studies addressed nonsurgical interventions, longer-term breastfeeding or growth outcomes, or surgical intervention compared with other approaches to improve breastfeeding, such as lactation consultation.
A small body of evidence suggests that frenotomy may be associated with mother-reported improvements in breastfeeding, and potentially in nipple pain, but with small, short-term studies with inconsistent methodology, strength of the evidence is low to insufficient.
Comments
Re:Letter to the editor regarding Treatment of ankyloglossia and breastfeeding outcomes
Dear Dr. Bakshi,
Thank you for your comments. This is indeed a controversial and important topic. There is a significant literature on the benefits of breastfeeding and therefore understanding factors that impede successful breastfeeding is an important area of research. Our study was designed to assess the effectiveness of frenotomy for ankyloglossia as it relates to clinical outcomes as noted in the paper and accompanying report. We performed an exhaustive and systematic review of the literature with strict inclusion criteria. One inclusion criterion was that effectiveness studies should have a comparison group, as is necessary to establish comparative effectiveness. Therefore, case series like those listed (i.e., Geddes et al. 2008, Riskin et al. 2014, Toner et al. 2014) were excluded from assessment of effectiveness.
We did identify the referenced studies evaluating ultrasound in relation to tongue mobility in our search. These studies were case series and therefore did not meet study design criteria and measured tongue mobility, which we considered an intermediate outcome. The comparison of treatment data against normal controls is not the same as comparing treatment versus no treatment in similar populations, which is the focus of comparative effectiveness research. Importantly, these types of studies by their nature cannot conclusively demonstrate comparative clinical advantage to frenotomy in neonates and children with ankyloglossia.
We agree that there are substantial limitations to the current literature on ankyloglossia and the outcomes of its treatment. Even basic epidemiological concepts such as its incidence, grading, and prevalence are not clear. This may be in part due to the lack of a consensus on its definition and severity grading system. However, the letter's statement that "there is enough evidence to establish the efficacy of frenotomy in higher grades of ankyloglossia or neonates with feeding problems" cannot be supported by the findings from our systematic review.
Nonetheless, our study identified a small body of evidence that may suggest that frenotomy may be associated with mother-reported improvement in breastfeeding, and potentially in nipple pain. However, the studies had small sample sizes, short follow-up, and inconsistent methodologies. The fact that strength of evidence was insufficient to low does not necessarily indicate that frenotomy does not provide benefit; rather this strength of evidence means that currently available data do not demonstrate consistent effectiveness. Further comparative effectiveness research is necessary with better and consistent characterization of the tongue-tie, longer follow-up, and improved outcome ascertainment.
Sincerely, David O. Francis, MD, MS Melissa L. McPheeters, PhD, MPH
Conflict of Interest:
None declared
Letter to the editor regarding Treatment of ankyloglossia and breastfeeding outcomes
Dear editor,
I am writing to you in reference to a very thought provoking article titled 'Treatment of ankyloglossia and breastfeeding outcomes: a systematic review' by Francis DO ET al1. The article sheds light on a very controversial subject and I would like to appreciate the efforts put in by the authors. However I beg to differ from the author's conclusion and would like to put forth my argument through your esteemed journal.
There have been many trials and studies2, 3 which have proven the benefit of frenotomy in breastfeeding. There have been few interesting scientific studies using ultrasound to image the oral cavity3, which have conclusively demonstrated that the ease and ability to breastfeed does improve after frenotomy.
Breastfeeding is a complex and delicate process and can be affected by multiple factors, pain being one of them. Pain can affect the mother psychologically thereby reduce the motivation to breast feed and may also reduce the overall milk production. There have been many studies which have demonstrated that there is a reduction in pain and improvement in the mothers comfort while breastfeeding after frenotomy4.
The main problem with most of the previous studies has been the reported prevalence and grading of ankyloglossia, which varies from author to author, even till now no definite consensus has been established. The importance of breastfeeding has been universally accepted; also it has been proven that frenotomy is a simple and safe procedure and can be carried out on an outpatient basis with minimal cost5. Therefore even if it offers mild benefit to the mothers for breastfeeding, it should be offered as an option to them.
In the end I would like to stress that although I agree that presently, the benefit of frenotomy in milder grades of ankyloglossia is doubtful and cannot be recommended. But there is enough evidence to establish the efficacy of frenotomy in higher grades of ankyloglossia or neonates with feeding problems and therefore it should be recommended to mothers as an option to improve the outcome of breastfeeding.
REFERENCES
1. Francis DO, Krishnaswami S, McPheeters M. Treatment of ankyloglossia and breastfeeding outcomes: a systematic review. Pediatrics. 2015; 135:e1458-66. doi: 10.1542/peds.2015-0658.
2. Buryk M, Bloom D, Shope T. Efficacy of neonatal release of ankyloglossia: a randomized trial. Pediatrics. 2011; 128:280-8. doi: 10.1542/peds.2011-0077.
3. Geddes DT, Langton DB, Gollow I, Jacobs LA, Hartmann PE, Simmer K. Frenulotomy for breastfeeding infants with ankyloglossia: effect on milk removal and sucking mechanism as imaged by ultrasound. Pediatrics. 2008; 122:e188-94. doi: 10.1542/peds.2007-2553.
4. Riskin A, Mansovsky M, Coler-Botzer T, Kugelman A, Shaoul R, Hemo M, Wolff L, Harpaz S, Olchov Z, Bader D. Tongue-tie and breastfeeding in newborns-mothers' perspective. Breastfeed Med. 2014; 9:430-7. doi: 10.1089/bfm.2014.0072.
5. Toner D, Giordano T, Handler SD. Office frenotomy for neonates: resolving dysphagia, parental satisfaction and cost-effectiveness. ORL Head Neck Nurs. 2014; 32:6-7.
Conflict of Interest:
None declared