Tongue-tie (ankyloglossia) is a common condition with a prevalence between 0.2 and 10.7% (Segal et al, 2007; Francis et al, 2015; Power and Murphy, 2015), and is defined as an embryological remnant of tissue between the under-surface of the tongue and the floor of the mouth that can restrict tongue movement. This is an important condition for primary care because it can cause breastfeeding difficulty for the mother and infant, including nipple pain, difficult attachment and increased bottle feeding rates (Segal et al, 2007; Suter and Bornstein, 2009; Edmunds et al, 2011). Mothers have described an ‘anticipatory dread’ towards breastfeeding an infant with tongue-tie (Edmunds et al, 2013). Tongue-tie can be obvious, such as with a restricting frenulum or heart-shaped tongue, but some infants may only be diagnosed after assessment of breastfeeding difficulties, positioning, attachment, tongue appearance and function, by practitioners with the appropriate expertise (Figure 1) (Hill and Johnson, 2007). Other causes of difficult feeding such colic, reflux, poor attachment, positioning, and inverted nipples would need to be considered (Amir, 2014). Assessment for tongue-tie is not part of routine UK neonatal examination and is usually identified by GPs, midwives or health visitors in the context of emerging breastfeeding problems. Tongue-tie, if it is symptomatic, can be treated with a frenulotomy, which involves snipping the tongue with sharp, round scissors. Bleeding is stopped by allowing the baby to feed, which compresses the wound (Edmunds et al, 2011). In 2005, UK guidelines from the National Institute of Health and Clinical Excellence (NICE) (2005) concluded that limited evidence suggested that when tongue-tie is thought to be a problem, division is both safe and able to improve breastfeeding where conservative measures such as frenulum massage and breastfeeding support have failed, although there is no published research to recommend frenulum massage.
Literature review
Eight systematic reviews (Segal et al, 2007; Suter and Bornstein, 2009; Edmunds et al, 2011; Finigan and Long, 2013; Webb et al, 2013; Ito, 2014; Francis et al, 2015; Power and Murphy, 2015) have been undertaken in this area, highlighting the clinical uncertainty surrounding management of tongue-tie. Five reviews reported that, compared to routine breastfeeding support, frenulotomy could improve breastfeeding difficulties caused by tongue-tie (Finigan and Long, 2013; Webb et al, 2013; Ito, 2014; Power and Murphy, 2015). Finigan and Long (2013) concluded that there was evidence for improvement in more than 50% of the cases and Edmunds et al (2011) reported that it would be unethical not to provide treatment where benefit had been shown. Two reviews drew more tentative conclusions that there was a small body of evidence to suggest that frenulotomy could offer improvement, and stated that confidence in that evidence was low (Segal et al, 2007; Francis et al, 2015). Suter and Bornstein (2009) wrote that, while breastfeeding problems could be associated with tongue-tie in 25-80% of cases, due to the impossibility of making comparisons between studies, they could not draw conclusions to resolve controversy in this area. Ambivalent conclusions were universally due to the heterogeneous nature of the research, use of diverse diagnostic and outcome measures; inadequate comparison of conservative management and lack of robust study design.
Although not overwhelming, there is a growing body of research that suggests that some infants with tongue-tie need additional breastfeeding support with or without frenulotomy, but there is evidence of a reluctance to refer or of a lack of knowledge about frenulotomy. This is understandable given the limited evidence, but this may have an impact on the quality of care for these infants. Two studies have identified diverse and contrasting views between paediatricians, otolaryngologists and surgeons for frenulotomy (Messner and Lalakea, 2000; Brinkmann et al, 2004) and a third study described mothers encountering professionals who rarely identified tongue-tie as a problem, or who gave conflicting advice (Edmunds et al, 2013). In addition, referral routes for frenulotomy vary around the UK and can cause confusion for parents and referrers: procedures are performed by otolaryngologists, maxillofacial surgeons, paediatric surgeons, midwives, health visitors or lactation consultants. Treatment is also widely available through private tongue-tie practitioners. It is unclear how soon is ideal to perform a frenulotomy to support the establishment of breastfeeding; however, there is evidence of a substantial wait for treatment within the UK: a freedom of information request to NHS Trusts found that the longest wait for frenulotomy, after diagnosis, was 84 days and the average 21.6 days (Boffey, 2014).
Objectives
Overall, the literature provides evidence that tongue-tie can cause breastfeeding problems that may be improved by frenulotomy, but that this is often not reflected in practice and can lead to diagnostic and treatment delay. This study aimed to explore the experience of parents and primary care professionals, exploring knowledge and opinions on tongue-tie in breastfeeding and their experience of the diagnostic and treatment pathway in the UK, with the hope of understanding how treatment pathways for infants with tongue-tie can be improved, even in the context of clinical uncertainty.
Methods
A qualitative case study approach was used with 23 participants; this sample size is in keeping with wider qualitative research practice as it was felt that data saturation had been reached and further interviews would not elicit further information (Mason, 2010). Semi-structured telephone interviews were conducted with five GPs, four midwives and five health visitors and a focus group with nine mothers. The women were recruited from one of three local free-to-attend breastfeeding clinics and NHS professionals were recruited from 25 training general practices in Cambridgeshire, where the lead researcher was based. All research was performed in compliance with University of East Anglia guidelines and approval was received from the Faculty of Medicine and Health Sciences Research Ethics Committee.
As large scale random sampling to achieve generalisability is not possible with such intensive research methods, purposive sampling was undertaken to attempt to gain transferability to the wider population (Silverman, 2005). The inclusion criteria were women with breastfed infants aged 7 months or younger. In order to achieve a mixed sample, participants were stratified to reflect rural and urban backgrounds and different pathways to clinic. Women who had breastfed older children with tongue-tie were not included in the study, nor mothers of babies born prematurely or with significant health problems, or who were not confident to communicate in English. A mixed gender sample was selected of three female and two male GPs, and all female midwife and health visitor participants. Professionals were selected from a mixture of urban and rural settings with a range of years practising. No professionals with specific training in tongue-tie were included in the study.
Data were collected over a 4-month period from June to October 2015. The focus group was conducted over 60 minutes with a facilitator and observer. A topic guide was used covering four areas: knowledge about tongue-tie, diagnosis, clinical assessment of tongue-tie, and referral practices. Practitioners were interviewed by telephone; questions explored experience of tongue-tie and breastfeeding, thoughts on the impact of tongue-tie and frenulotomy, their examination and referral practices, and their knowledge and opinions. The interviews were recorded digitally and transcribed verbatim. Data were analysed in Microsoft Word and Excel 2013 using thematic analysis based on a grounded theory approach (Charmaz, 2006). Codes were assigned to each line, grouped into categories and links made between codes and categories. A sample of initial codes were derived separately by two of the authors to avoid bias and discrepancies in approach to coding were resolved. Pseudonyms were used to preserve anonymity.
Nine mothers participated; for eight, this was their first child. All the women in this study had wanted to breastfeed but had universally experienced difficulties in feeding. They described vulnerabilities, by recounting feeling ‘out of it’ postpartum; receiving sometimes conflicting advice on tongue-tie and breastfeeding; and feeling ‘desperate’ to have a frenulotomy. One health visitor, Kacy, described some of the mothers she has seen having problems with tongue-tie:
‘They are pretty desperate because of their wish to breastfeed, the pain and also desperately trying to do what they feel is the right thing for their baby and them just not managing. And the babies tend to be irritable and grizzly and you know not having a good time of it either.’
For the infants of the mother participants, the average age of tongue-tie diagnosis was 18.4 days (range 0-70), the average age of treatment was 25.3 days (range 5-70). All infants received a frenulotomy and six of the nine were done privately. Mothers discussed at length how they felt that they were ‘lucky’ to see the right person and examples were given of health professionals who seemed to have, ‘no idea’ about tongue-tie or where tongue-tie diagnoses were missed. Mothers described consulting ‘multiple’ clinics and professionals for diagnosis. For some, the diagnoses were made at breastfeeding clinics, refuted by physicians and then later reconfirmed. Wendy talks of her distress when seeking advice about tongue-tie,
‘I went to the GP, they said they don't know anything about tongue-tie […] you don't know where to go. You're so alone in there.’
In contrast, many participant practitioners demonstrated a breadth of knowledge about tongue-tie and discussed the need to examine the tongue in certain situations. Five mothers said that they had received their baby's tongue-tie diagnosis within 3 days of birth, and mothers also talked about the helpful advice they had received about attachment and positioning.
Mothers discussed their surprise that many practitioners did not examine their babies' mouths, and were frustrated at it not being part of the routine check and with healthcare practitioners' lack of skills in this area. Many described themselves as ‘lucky’ to see the right practitioner. Esther compared her experience when a practitioner made the diagnosis to her previous encounter with professionals, saying:
‘No one else, I don't think, listened to the whole story. So they won't latch, she won't latch … But also, no one listened to the “and they're feeding for hours and there's this and there's that.” They just wanted to look at a little bit of the picture.’
Aside from knowledge, attitudes towards frenulotomy among practitioners also varied: two GPs felt they did not have enough information to comment; many practitioners emphasised that the need for it depended on the clinical picture, and several practitioners emphasised how important they felt frenulotomy could be. In contrast, there were some more negative views about breastfeeding and the diagnosis of tongue-tie, with some professionals dismissing its existence or significance, and others referring to it as a ‘hobby horse’ of the midwives or a ‘trend term’. Mothers described feeling dismissed; noticing negative remarks and body language; receiving unhelpful or conflicting advice; and being told to ‘toughen up’, that they were ‘doing something wrong’ or that they had a ‘lazy baby’. Louise discussed an encounter with a paediatrician after her tongue-tie diagnosis was refuted, saying:
‘She was like, “If it's really bad, you can put him on formula.” And we both looked at each other, [and said] “But that's not an option”. She stood on the side, leaning with her arms crossed and just didn't answer. So we just got up and walked out. I was so upset.’
Professional knowledge on breastfeeding was in the form of embodied knowledge, which is subjective knowledge gained through personal experience of breastfeeding a baby; vicarious or cultural knowledge, through observing a breastfeeding experience; practice-based knowledge gained through clinical experience; and formal knowledge from structured learning opportunities, literature and web based sources. Among practitioners there was varied knowledge of national guidelines and all expressed some lack of confidence regarding their knowledge base or ability to diagnose tongue-tie. Although the health visitors and midwives had received professional training on tongue-tie in recent years, it was something that all professionals reported was mostly absent from their previous training. While clinical experience seemed to be the key source of knowledge, several healthcare professionals had personal experience of tongue-tie, which clearly influenced their attitudes. This was seen in comments beginning ‘based on my own personal experience…’, or in their reported observations of others: midwife Shellie discussed a paediatrician who she felt ‘knows the evidence’ but ‘because his wife successfully breastfed their baby with a tongue-tie he sort of won't address it’. Issues of multidisciplinary dialogue were also raised, with evidence of limited discussion about tongue-tie between GPs and midwives or health visitors. It was clear that there were differing epistemological approaches to understanding tongue-tie, with each of these disciplines finding greater value in theoretical, embodied or vicarious knowledge. There was also a lesser emphasis on the promotion of breastfeeding from GPs; two noted that they felt that, as GPs, they needed to put on less pressure to breastfeed. One midwife, Shellie, commented,
‘You know, I still think there is a feeling among some people, “Oh, if you don't breastfeed it is okay, you just formula feed don't you?” … It is deemed to be an equal alternative.’
Most participants raised concerns that frenulotomies were often performed within the private sector, and many expressed unease or confusion about how to share information about private practice with mothers when this was not their usual practice. Others raised questions about a financial conflict of interest, that follow-up may be inadequate or costly, or that this took advantage of vulnerable mothers who were in a ‘desperate’ situation and would ‘pay anything’. In contrast, professionals and mothers also talked about tongue-tie practitioners who provided a wealth of experience, usually as ex-midwives or health visitors; prompt assessment; adequate time to fully listen and examine; and good follow-up care. Mothers discussed with some passion that they felt lucky that they could afford private treatment, but felt strongly that this was ‘far too much money’ and unfair for those less fortunate, something that was also discussed by practitioners.
Strengths and limitations
The research had a strong insider perspective, with the lead researcher, a GP and mother, having had experience of breastfeeding an infant with tongue-tie, which enhanced the research process. The interview technique used was designed to be facilitatory rather than conversational, and established data analysis techniques and triangulation of data from a range of participants were used to attempt to mitigate bias from the insider perspective. The sample size was small and from one geographic area but included a range of practitioners and mothers who had had a variety of referral routes to the breastfeeding clinic. Eight participants were from Caucasian background and only one did not hold a higher degree; however, this fits with known breastfeeding patterns, where a higher proportion of breastfeeding mothers in the UK are Caucasian and from professional and intermediate occupations (McAndrew et al, 2012). Six participants had their frenulotomy performed privately and had a shorter wait to treatment. As the prevalence of private frenulotomies within the UK is undocumented, it is unclear as to whether this is very different from wider practice. In addition, due to recruitment problems, many of the mothers were first time mothers and thus their problems may be amplified. However, the majority of study participants were health professionals and thus brought a breadth of experience that offered some balance to the selection bias seen within the mother participants.
Comparison with existing literature
Although there were some design limitations, many of these findings are supported by previous research. The experience of mothers in our study is akin to the difficult journey illustrated by Edmunds et al (2013), although in this study group there was a shorter wait between diagnosis and treatment (6.9 days, range 0-24) compared to a UK average of 21.6 identified by Boffey (2014). Variability in knowledge regarding tongue-tie has also been previously demonstrated (Messner and Lalakea, 2000) as has a lack of knowledge and confidence towards breastfeeding in general (Smale et al, 2006). Smale et al (2006) document the lack of prioritisation of breastfeeding education, which is mirrored by lack of tongue-tie in training in this study. The inconsistent approaches towards tongue-tie discussed here have also been seen by Dykes (2006) in relation to wider breastfeeding care as well as to differing paradigmatic stances between professionals. It was clear in this study that different disciplines had different epistemological stances, stemming from the extent of their different sources of breastfeeding knowledge (theoretical, embodied and vicarious) but also due to the impact that different types of knowledge had on their opinion.
Conclusion
Summary
This study painted a picture of vulnerable patients, who reported experiencing breastfeeding difficulties while meeting the challenges of a new baby—in a system that has resource limitations, organisational difficulties and a private–NHS interface. This is in the context of variable professional knowledge and opinions, conflicting advice and diagnostic conclusions. This led to a difficult patient pathway, and mothers told a common story of having to push for support, attending multiple appointments, diagnostic and treatment delay and ongoing distress. They described feeling ‘lucky’ to receive a diagnosis, after having sometimes been lost in the system, frustrated and desperate.
Implications for research and practice
The authors support the need for further investigation into the impact of tongue-tie and the effectiveness of frenulotomy, including studies with newly developed tools, such as the Bristol Tongue Assessment Tool (Ingram et al, 2015).
With regards to education, an increased emphasis on breastfeeding training is recommended, including discussion around tongue-tie and the referral routes for management; this would be included within undergraduate and pre-registration training and as part of continuing professional development. Undergraduate training could be through the Baby Friendly Initiative University Standards Programme (UNICEF, 2014), which provides learning outcomes to be met by midwives and health visitors, who can then provide basic breastfeeding knowledge. Importantly, this includes drawing on an understanding of wider social, cultural and political influences that affect breastfeeding, essential to providing holistic support of breastfeeding. In addition, training also needs to develop skills to learn from formal, embodied and vicarious knowledge, especially within the context of clinical uncertainty. Key practitioners such as midwives, health visitors and lactation consultants should also be adequately trained to be able to diagnose tongue-tie.
Practice-based issues have also been partly described in relation to wider breastfeeding care. In existing pathways, there may be potential to improve diagnosis of tongue-tie. There is not enough evidence on the benefits and potential harms from frenulotomy and other treatments for tongue-tie to support screening for this condition; however, guidelines on tongue-tie, such as those from NICE (2005), could be extended from suggesting that it is a safe procedure to encompassing guidance on education, consideration of tongue-tie diagnosis when evaluating feeding problems, and universal provision of the frenulotomy when clinically indicated. The role of clinicians without expertise in tongue-tie diagnosis would be to refer mothers for further evaluation by someone with skills in breastfeeding and tongue-tie assessment. Issues of discordance of approach and inadequate dialogue between the multiprofessional team need to be considered in education and clinical pathway design.
Finally, there have been ethical and professional issues raised concerning a national health service system with vulnerable parents making desperate decisions about whether to pay for private treatment. In a public health system such as the NHS, treatment pathways need to be clear, timely and accessible.