We systematically reviewed the literature on surgical and nonsurgical treatments for infants and children with ankyloglossia and ankyloglossia with concomitant lip-tie.
We searched MEDLINE® (PubMed®), PsycINFO®, Cumulative Index of Nursing and Allied Health Literature (CINAHL®) and Embase (Excerpta Medica Database), as well as the reference lists of included studies and recent systematic reviews. We conducted the searches between September 2013 and August 2014.
We included studies of interventions for ankyloglossia published in English. Two investigators independently screened studies against predetermined inclusion criteria and independently rated the quality of included studies. We extracted data into evidence tables and summarized them qualitatively.
We included 58 unique studies comprising 6 randomized controlled trials (RCTs) (3 good, 1 fair, 2 poor quality), 3 cohort studies (all poor quality), 33 case series, 15 case reports, and 1 unpublished thesis. Most studies assessed the effects of frenotomy (a procedure in which the lingual frenulum is divided) on breastfeeding-related outcomes. Four RCTs reported improvements in breastfeeding efficacy using either maternally reported or observer ratings, while two RCTs using observer ratings found no improvement. Mothers consistently reported improved breastfeeding effectiveness after frenotomy, but outcome measures were heterogeneous and short term. Future studies could provide additional data to confirm or change the measure of effectiveness; thus, we consider the strength of evidence (SOE; confidence in the estimate of effect) to be low at this time. Furthermore, this literature is characterized by (1) a lack of details about the surgical procedure, (2) cointerventions allowed variably in control groups, and (3) diversity of provider settings. Pain outcomes improved for mothers of frenotomized infants compared with control in one study of 6-day old infants but not in studies of infants a few weeks older. Given these inconsistencies and the small number of comparative studies and participants, the SOE is low for an immediate reduction in nipple pain. Three studies with significant limitations reported improvements in other feeding outcomes with frenotomy, and four poor-quality studies reported some improvements in speech articulation but mixed results related to overall speech sound production. Three poor-quality comparative studies noted some improvements in social concerns and gains in tongue mobility in treated participants. SOE for all of these outcomes is insufficient. SOE is moderate for minor and short-term bleeding following surgery and insufficient for other harms (reoperation, pain).
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 short-term studies, inconsistently conducted, SOE is generally low to insufficient. Comparative studies reported improvements in some measures of speech, but assessment of outcomes was inconsistent. Few studies addressed tongue mobility and self-esteem issues. Research is lacking on nonsurgical interventions, as well as on outcomes other than breastfeeding.