Authors' objectives: Tongue-tie can be diagnosed in 3–11% of babies, with some studies reporting almost universal breastfeeding difficulties, and others reporting very few feeding difficulties that relate to the tongue-tie itself, instead noting that incorrect positioning and attachment are the primary reasons behind the observed breastfeeding difficulties and not the tongue-tie itself. The only existing trials of frenotomy are small and underpowered and/or include only very short-term or subjective outcomes. To investigate whether frenotomy is clinically and cost-effective to promote continuation of breastfeeding at 3 months in infants with breastfeeding difficulties diagnosed with tongue-tie. Breastfeeding difficulties have been associated with many factors, from a societal to an individual level. Tongue-tie can be diagnosed in 3–11% of babies, with the variation in reported prevalence thought to relate to the use of different diagnostic or severity criteria. Up to half of babies with tongue-tie are reported to have breastfeeding difficulties, but the reported proportion is highly variable. Some studies report almost universal difficulties, and others report very few feeding difficulties that relate to the tongue-tie itself, instead noting that incorrect positioning and attachment are the primary reasons behind the observed breastfeeding difficulties and not the tongue-tie itself. In a UK survey, it was noted that management of tongue-tie in infants with breastfeeding difficulties was therefore highly variable across the country. This is coupled with highly variable provision of breastfeeding support, which can range from minimal to expert and intensive, and using a variety of different models including peer supporter, midwife and health visitor. A Cochrane review identified five prior randomised controlled trials (RCTs) of frenotomy including a total of only 302 infants. The trials are small and underpowered and/or include only very short-term or subjective outcomes, suggesting further robust evidence is needed. Hence there is considerable controversy regarding, not only the diagnosis and clinical significance, but also the management of tongue-tie. Current National Institute for Health and Care Excellence (NICE) guidance allows for the procedure, based on lack of safety concerns, but notes very limited evidence of efficacy. There is therefore a clear need for an assessment of the clinical- and cost-effectiveness of frenotomy for babies diagnosed with tongue-tie in the form of an adequately powered, pragmatic RCT, taking into account the diagnostic controversy and variation in practice. To investigate whether frenotomy is clinically- and cost-effective to promote continuation of breastfeeding at 3 months in infants with breastfeeding difficulties diagnosed with tongue-tie.

Authors' results and conclusions: Between March 2019 and November 2020, 169 infants were randomised, 80 to the frenotomy with breastfeeding support arm and 89 to the breastfeeding support arm from a planned sample size of 870 infants. The trial was stopped in the context of the COVID-19 pandemic due to withdrawal of breastfeeding support services, slow recruitment and crossover between arms. In the frenotomy with breastfeeding support arm 74/80 infants (93%) received their allocated intervention, compared to 23/89 (26%) in the breastfeeding support arm. Primary outcome data were available for 163/169 infants (96%). There was no evidence of a difference between the arms in the rate of breastmilk feeding at 3 months, which was high in both groups (67/76, 88% vs. 75/87, 86%; adjusted risk ratio 1.02, 95% confidence interval 0.90 to 1.16). Adverse events were reported for three infants after surgery [bleeding (n = 1), salivary duct damage (n = 1), accidental cut to the tongue and salivary duct damage (n = 1)]. Cost-effectiveness could not be determined with the information available. This trial does not provide sufficient information to assess whether frenotomy in addition to breastfeeding support improves breastfeeding rates in infants diagnosed with tongue-tie. Between March 2019 and November 2020, 169 infants were randomised, 80 to the frenotomy with breastfeeding support arm and 89 to the breastfeeding support arm from a planned sample size of 870 infants. The trial was stopped in the context of the ongoing COVID-19 pandemic due to withdrawal of breastfeeding support services, slow recruitment and crossover between arms. In the frenotomy with breastfeeding support arm 74/80 infants (93%) received their allocated intervention, compared to 23/89 (26%) in the breastfeeding support arm. Characteristics of participants were similar between the two trial arms. Infants had a mean age of 3 weeks, 87% were born at ≥38 weeks’ gestation, and they had a mean birthweight of 3439g. Overall 33% of infants had a BTAT score of 4 or less, 66% had exclusive breastmilk feeding in the previous 24 hours, and 40% had exclusive direct breastmilk feeding. Thirty-four per cent of infants had also received formula milk in the previous 24 hours. Mothers were a mean of 32 years old, 94% were of white ethnicity, and 48% had a previous live birth. Only 8% were resident in the most deprived quintile of areas. Mothers reported a mean pain score of 4 out of 10 while feeding during the previous 24 hours and 42% had some anxiety or depression. More than half of women recruited to the trial believed a frenotomy would help their baby. Primary outcome Primary outcome data were available for 163/169 infants (96%). There was no evidence of a difference between the arms in the rate of breastmilk feeding at 3 months, which was high in both groups [67/76, 88% vs. 75/87, 86%; adjusted risk ratio (aRR) 1.02, 95% CI 0.90 to 1.16]. The statistical power of the analysis was extremely limited due to not achieving the target sample size because of the early cessation of the trial and the high proportion of infants in the breastfeeding support arm who underwent frenotomy. There was no evidence of differences between trial arms in any outcomes. Rates of continued breastmilk feeding were high at 3 months in both the frenotomy with breastfeeding support and breastfeeding support groups. Complications of the procedure were not uncommon, occurring in around 1 in 50 infants. Most infants in the control groups of the five previous trials identified in a previous Cochrane review also underwent frenotomy (77–100%). On this basis all five trials were considered of low quality and at high risk of bias. The 73% frenotomy rate in the breastfeeding support arm that we observed in FROSTTIE is comparable, but on this basis it must also be regarded as at high risk of bias. This trial does not therefore provide sufficient information to assess whether frenotomy in addition to breastfeeding support improves breastfeeding rates in infants diagnosed with tongue-tie. The effectiveness and cost-effectiveness of the procedure still need to be established. Other study designs will need to be considered to address this objective.

Authors' methods: A multicentre, unblinded, randomised, parallel group controlled trial. Twelve infant feeding services in the UK. Infants aged up to 10 weeks referred to an infant feeding service (by a parent, midwife or other breastfeeding support service) with breastfeeding difficulties and judged to have tongue-tie. Infants were randomly allocated to frenotomy with standard breastfeeding support or standard breastfeeding support without frenotomy. Primary outcome was any breastmilk feeding at 3 months according to maternal self-report. Secondary outcomes included mother’s pain, exclusive breastmilk feeding, exclusive direct breastfeeding, frenotomy, adverse events, maternal anxiety and depression, maternal and infant NHS health-care resource use, cost-effectiveness, and any breastmilk feeding at 6 months of age. The statistical power of the analysis was extremely limited due to not achieving the target sample size and the high proportion of infants in the breastfeeding support arm who underwent frenotomy. Study design The FROSTTIE trial was a multicentre, RCT conducted in 12 infant feeding services in England. Inclusion criteria Any infant aged

Project Status: Completed

URL for project: https://www.journalslibrary.nihr.ac.uk/programmes/hta/16/143/01

Year Published: 2023

URL for published report: https://www.journalslibrary.nihr.ac.uk/hta/WBBW2302

URL for additional information: English

English language abstract: An English language summary is available

Publication Type: Full HTA

Country: England, United Kingdom

DOI: 10.3310/WBBW2302

Organisation Name: NIHR Health Technology Assessment programme

Contact Address: NIHR Journals Library, National Institute for Health and Care Research, Evaluation, Trials and Studies Coordinating Centre, Alpha House, University of Southampton Science Park, Southampton SO16 7NS, UK

Contact Name: journals.library@nihr.ac.uk

Contact Email: journals.library@nihr.ac.uk