Diagnostic et traitement de l’ankyloglossie au Canada : analyse de l’environnement

Contexte

Santé Canada et l’Organisation mondiale de la santé encouragent fortement l’allaitement maternel au cours des six premiers mois de la vie^1,2. En 2011-2012, l’Enquête sur la santé dans les collectivités canadiennes (ESCC) indique que 89 % des mères ont allaité, ce qui représente une légère augmentation lorsque l’on compare à 85 % en 2003³. Le pourcentage des mères ayant recours à l’allaitement exclusif pour six mois ou plus a aussi augmenté, passant de 17 % en 2003 à 26 % en 2011-2012⁴. Alors que les organismes professionnels appuient de plus en plus l’allaitement comme le choix optimal en nutrition pour les nouveau-nés et les nourrissons⁵, il semble y avoir une pression croissante pour déterminer quels sont les obstacles à la réussite de l’allaitement.

L’ESCC souligne que l’une des raisons les plus souvent utilisées pour justifier l’arrêt de l’allaitement avant six mois est « la difficulté associée à la technique d’allaitement⁴». L’ankyloglossie est l’une des conditions qui peuvent affecter l’allaitement. L’ankyloglossie se caractérise par un frein lingual anormalement court. Le frein lingual est cette membrane muqueuse située sous la langue⁶. Lorsqu’elle est trop courte, le mouvement de la langue est restreint. En plus d’avoir des répercussions fonctionnelles liées à la parole ou à l’hygiène buccale^8,9, l’ankyloglossie a une incidence sur la capacité de l’enfant à s’arrimer correctement sur le sein de la mère et cause une réduction de l’afflux de lait maternel. Plus spécifiquement en ce qui concerne l’allaitement, l’ankyloglossie peut mener à un apport de lait maternel insuffisant, à la prolongation de la durée de l’allaitement et à l’irritation du mamelon de la mère pouvant aller jusqu’à des saignements⁸. Le taux de diagnostics d’ankyloglossie à l’hôpital au Canada est de 22,6 par 1 000 naissances¹⁰, ce qui pourrait représenter une sous-évaluation du nombre de cas réels parce que plusieurs cas sont diagnostiqués ailleurs qu’à l’hôpital. Des études américaines au sujet de la prévalence ont présenté des estimations allant de 4,2 % à 10,7 % des enfants.

Des données probantes suggèrent que la frénectomie pourrait améliorer les résultats touchant aux enjeux de l’allaitement pour les nouveau-nés^5,11,12. La frénectomie, aussi appelée frénotomie, est une intervention consistant en une scission du frein lingual en utilisant un ciseau ou un scalpel stériles ou, parfois, des techniques au laser^8,13. Les effets secondaires sont rares, mais comprennent des saignements, de l’infection et des dommages à la langue ou aux glandes salivaires^5,13. Il peut aussi arriver que le frein se rattache à la base de langue et qu’une nouvelle intervention soit nécessaire¹³. Dans certains cas, la plastie du frein, une procédure plus complexe, peut être effectuée, habituellement sous anesthésie et avec des outils chirurgicaux¹³. Dans ce cas, la fermeture de la plaie est achevée selon un modèle particulier visant à allonger le frein lingual, alors que la frénectomie n’est qu’un simple relâchement sans suture^13,14. Étant donné qu’il n’existe pas de définition mondialement acceptée de l’ankyloglossie, on ne s’entend pas sur le moment opportun d’avoir recours à la frénectomie.

Les autorités sanitaires canadiennes ont noté une augmentation perceptible du taux de frénectomie, attribuable partiellement aux pressions des mères qui commencent l’allaitement avant de prendre leur congé de l’hôpital, bien que les facteurs influençant cette tendance restent à déterminer¹⁶. Certains croient que des lignes directrices de pratique plus précises sont nécessaires si l’on veut s’assurer que les nourrissons ayant des problèmes à l’allaitement en raison d’une ankyloglossie soient traités adéquatement¹⁶.

(La suite de ce texte est en anglais.)

Objectives

CADTH conducted an Environmental Scan to gather jurisdictional perspectives on ankyloglossia diagnosis and treatment in Canada. The key objectives of this Environmental Scan were:

to describe current practices for the assessment and diagnosis of ankyloglossia in Canadian jurisdictions
to describe current practices for patient selection for frenectomy (tongue-tie release, also referred to as frenotomy) procedures in Canadian jurisdictions
to describe and compare temporal trends related to the use of frenectomy to treat ankyloglossia across Canadian jurisdictions.

Methods

Approach

Information was retrieved from a limited literature search and a survey, both informed by the components outlined in Table 1. The survey questionnaire was distributed to key jurisdictional informants and stakeholders. Findings from the literature search were used to supplement the information retrieved from the surveys.

Table 1: Components and Information-Gathering Approach

		Inclusion
Components	Population	Pediatric patients with suspected ankyloglossia
	Intervention	Strategies for the assessment and diagnosis of ankyloglossia Frenectomy as a form of treatment
	Settings	Any Canadian health care setting (e.g,. urban, rural and remote settings, primary and secondary care, private facilities)
	Outcomes	Current practices for: the assessment and diagnosis of ankyloglossia patient selection for frenectomies including : capacity (i.e., eligibility criteria and referral process) location and setting for delivering care (i.e., province or territory; urban, rural, or remote; within a hospital, clinic, or home; remotely delivered) services offered to patients (i.e., types of interventions) Temporal trends related to the use of frenectomy Canadian guidance on the assessment, diagnosis, and treatment of ankyloglossia Barriers to and facilitators of optimal diagnosis and treatment of ankyloglossia
Information- gathering approach	Consultation ☐
	Survey ☒
	Literature search: ☒

Literature Search

A limited literature search was conducted using the following bibliographic databases: MEDLINE, PubMed, Embase, Cumulative Index to Nursing and Allied Health Literature (CINAHL) via EBSCO, the Cochrane Library, and University of York Centre for Reviews and Dissemination (CRD) databases. Grey literature was identified by searching relevant sections of the Grey Matters checklist (https://www.cadth.ca/grey-matters). No methodological filters were applied. The search was also not limited to any language or publication date.

Research Questions

The literature review component of this Environmental Scan aimed to address the following questions:

What are the Canadian policies, frameworks, guidelines, and other guidance documents related to the assessment and diagnosis of ankyloglossia in Canada?
What are the barriers to optimal assessment and diagnosis of ankyloglossia in Canadian jurisdictions?
What are the facilitators of optimal assessment and diagnosis of ankyloglossia in Canadian jurisdictions?
What are the Canadian policies, frameworks, guidelines, and other guidance documents related to the patient selection for frenectomy procedures (tongue-tie release, also referred to as frenotomy) in Canada?
What are the barriers to appropriate patient selection for frenectomies in Canadian jurisdictions?
What are the facilitators of appropriate patient selection for frenectomies in Canadian jurisdictions?
What are the temporal trends related to the use of frenectomy to treat ankyloglossia across Canadian jurisdictions?

Screening and Study Selection

A single reviewer screened articles identified through the literature search for selection. Those that met the inclusion criteria (Table 1) were summarized in the report. Only English-language publications were selected for inclusion.

Survey

The survey questionnaire was developed to address the key objectives and included a combination of dichotomous (i.e., yes/no), ordinal, and nominal scales, and open-ended questions (Appendix 1). The survey questionnaire was peer-reviewed by eight expert stakeholders involved in the request prior to distribution.

The survey was distributed electronically, using Hosted in Canada Surveys,¹⁷ to key jurisdictional informants and stakeholders involved in planning, decision-making, management, and care provision related to assessing, diagnosing, and treating ankyloglossia. Attempts were made to capture responses from each province or territory, including respondents working in rural, remote, and urban health care settings. Survey respondents agreed to the reporting of the information they provided by electronically providing their consent.

The survey targeted the following viewpoints:

clinicians (including pediatricians and obstetrician-gynecologists)
ministry-level decision-makers
regional health authorities
breastfeeding specialists (including midwives)
lactation consultants
breastfeeding clinics
specialized hospitals, facilities, or clinics with an emphasis on maternal/newborn/children health
speech-language pathologists
dental associations and practitioners (including pediatric dentists)
public health professionals.

Respondents were identified through CADTH’s Implementation Support and Liaison Officer team, existing CADTH networks, and via stakeholder and expert referrals.

Synthesis Approach

Feedback from respondents who provided consent to use their survey information was included in the report. Quantitative survey questionnaire responses were summarized by question and presented according to the objectives of the report. Feedback from open-ended survey questions was also incorporated. Information identified through the literature search was organized by objective and summarized within relevant sections of the report.

Stakeholder feedback was solicited by posting a draft version of the report on CADTH’s website and by emails to subscribers to CADTH’s mailing lists. Survey questionnaire respondents and key informants involved in refining the project were also asked to provide feedback.

Findings

Quantity of Research and Summary of Study Characteristics

A total of 24 citations were identified in the literature search. No additional articles were retrieved from the grey literature search. Following screening of titles and abstracts, 15 articles were excluded and nine were selected for full-text review. Of the nine potentially relevant articles, three were selected for inclusion in the report.^10,16,18 This was supplemented by two documents identified through additional handsearching.^5,19 No additional literature was identified during search updates.

These final five studies included three observational studies,^10,16,18 one position statement,⁵ and one guideline.¹⁹ All studies were conducted in Canada, and guidance and position statements were issued by Canadian organizations.

Summary of Survey Respondent Characteristics

Overall, 36 individuals responded to the survey questionnaire. At least one response was received from all jurisdictions, excluding New Brunswick, the Northwest Territories, and the Yukon. A range of occupations were represented, including pediatricians and pediatric specialists, pediatric dentists, public health nurses and registered nurses, midwives, clinic directors, dietitians, and lactation consultants. Occupational settings included regional health authorities, general hospitals, stand-alone facilities, or clinics; specialized hospitals, stand-alone facilities, or clinics with an emphasis on maternal, newborn, or child health; and breastfeeding clinics, general or pediatric dental clinics, community care settings, home care settings, midwifery clinics, public health offices, education centres, and private practice. Most respondents reported working in urban settings, although 16 of 36 (44%) reported also working in rural settings and a minority (two of 36 [6%]) of respondents indicated working in remote settings or having remote management capacity. Details about survey respondent characteristics are presented in Appendix 2.

Current Practices for the Assessment and Diagnosis of Ankyloglossia in Canadian Jurisdictions

Guidance for Assessment and Diagnosis of Ankyloglossia

The approach used to diagnose ankyloglossia varies depending on the information source. One guideline from British Columbia (Perinatal Services BC Health Promotion Guideline Breastfeeding Healthy Term Infants) states that ankyloglossia is present when the infant is unable to adequately extend or elevate the tongue due to a short lingual frenulum attached to the tip of the tongue that restricts movement.¹⁹ The British Columbia guideline¹⁹ recommends that if feeding problems persist, the infant should be referred to a physician for further assessment and a possible frenectomy, as outlined in the American Academy of Breastfeeding Guidelines in Protocol #11.¹² The Protocol #11¹² guidelines state that when breastfeeding is difficult and a short or tight sublingual frenulum is noted, the appearance and function of the tongue may be assessed using a scoring system like the Hazelbaker scale.²⁰

The survey questionnaire asked respondents if there are any policies, frameworks, guidelines, or other guidance documents in use in their jurisdictions to guide the assessment and diagnosis of ankyloglossia. Of the respondents, 12 of 36 (33%) said that they use a form of policy, framework, or guideline to diagnose ankyloglossia. The identified guidance documents noted to be in use by survey respondents are summarized in Table 2.

Table 2: Guidance Documents for the Assessment and Diagnosis of Ankyloglossia

Guidance Document	Description
Frenotomy Decision Tool for Breastfeeding Dyads developed by Carole Dobrich^21a	This decision tool is split into two main parts: Part one includes five questions regarding breastfeeding outcomes. Part two includes four questions related to the examination of the anatomy and function of the infant’s tongue and frenulum. The two parts are then reviewed and, if a certain score is achieved, a frenectomy is recommended. The tool also includes other indicators of ankyloglossia.
The Hazelbaker Assessment Tool for Lingual Frenulum Function^20b	This tool includes two main assessment domains: appearance items and function items. Appearance items include the appearance of the tongue when lifted, elasticity of frenulum, length of lingual frenulum when the tongue is lifted, attachment of the lingual frenulum to the tongue, and the attachment of the lingual frenulum to the inferior alveolar ridge. Function items include lateralization, lift of tongue, extension of tongue, spread of anterior tongue, cupping, peristalsis, and snapback. Post-assessment, a score is tallied and ankyloglossia status is determined based on the score assigned by the tool.
The Canadian Paediatric Society position statement on ankyloglossia and breastfeeding^5c	The position statement includes a definition of ankyloglossia: There is neither a universally accepted definition of ankyloglossia nor practical objective criteria for diagnosingthis condition. Historically, definitions have been based oneither anatomical characteristics of the lingual frenulum (ie,the degree of fusion between the child’s tongue and thefloor of the mouth) or on functional impairment (ie, aninability to protrude the tongue past the incisal edge of thelower gingiva and other signs of decreased tonguemobility).⁵ The statement also presents information on the prevalence, pathophysiology, and management of ankyloglossia; the frenectomy procedure; and recommendations on examination, diagnosis, and treatment.
The Goldfarb Breastfeeding Clinic patient handout from the Herzl Family Practice Centre^22d	The patient handout contains a brief explanation of ankyloglossia and a detailed explanation of the frenectomy procedure. It also provides significant detail on how to care for the infant post-procedure.

^a Two respondents from Ontario, one respondent from Nova Scotia, two respondents from Quebec.
^b Three respondents from Ontario.
^c One respondent from Nunavut, one respondent from Ontario.
^d One respondent from Quebec.

Two of 36 (6%) survey respondents reported the use of custom assessment tools developed and created based on existing tools, local expertise, and incorporating existing information from resources such as the Kotlow classification,²³ the Martinelli-developed Lingual Frenulum Protocol with Scores for Infants,²⁴ and the Hazelbaker Assessment Tool for Lingual Frenulum Function (HATLFF).²⁰ One (3%) respondent of 36 noted the use of a patient-oriented resource²⁰ available on the International Breastfeeding Centre website.

Criteria for Eligibility for Ankyloglossia Assessment and Diagnosis

The majority (25 of 36, or 69%) of respondents said there are specific criteria that a patient should meet in order to be assessed and diagnosed for ankyloglossia. Examples provided included:

The infant should be assessed by a public health professional, family physician, and/or lactation consultant either at birth or shortly thereafter; the assessment may be required for referral (responses from Nova Scotia, Prince Edward Island, and Ontario).
The infant should be examined to see if there is a physical or functional deficit caused by a restrictive lingual frenulum (responses from Quebec and Ontario).
The infant and mother require a full breastfeeding assessment, especially if and to assess if there are persistent breastfeeding difficulties (e.g., poor transfer, decreased supply, risk of discontinuing, pain) and the infant is experiencing a slow weight gain (responses from Quebec and Ontario).
There is an established family history of ankyloglossia (response from Newfoundland).

It was noted that The HATLFF²⁰ and the Frenotomy Decision Tool for Breastfeeding Dyads²¹ (developed by Carole Dobrich) might be used to inform specific criteria for eligibility. Several respondents (four of 36, or 11%) suggested that every infant should be assessed for ankyloglossia, regardless of symptoms. One respondent indicated that assessment should be up to clinician discretion.

Current Practices for Patient Selection for Frenectomy Procedures in Canadian Jurisdictions

Guidance for Patient Selection for Frenectomy

Several Canadian guidance documents for patient selection were identified. The Canadian Paediatric Society position statement states that when ankyloglossia contributes to substantial breastfeeding difficulties, frenectomy should be performed by an experienced clinician.⁵ A guideline established by perinatal services in British Columbia¹⁹ states that treatment is not necessary if breastfeeding proceeds successfully; however, if feeding problems persist, the infant should be referred to a physician for further assessment and possible treatment, as outlined in Academy of Breastfeeding Medicine guideline Protocol #11¹² and in Lawrence and Lawrence’s²⁵ guide for the management of breastfeeding for medical professionals to improve breastfeeding effectiveness.

The survey respondents were asked if there are any policies, frameworks, guidelines, or other guidance documents in use in their jurisdiction to guide the patient selection for frenectomy procedures. In the survey, seven of 36 (19%) respondents (from Ontario, Quebec, and Newfoundland) indicated that they use a form of policy, framework, guideline, or another guidance document for the selection of patients for frenectomies. The guidance documents referenced were exclusively the same ones used for ankyloglossia diagnosis, including the Canadian Paediatric Society position statement,⁵ the Frenotomy Decision Tool for Breastfeeding Dyads developed by Carole Dobrich,²¹ and the HATLFF.²⁰

Eligibility Criteria for Frenectomy

The majority of the survey respondents (26 of 36, or 72%) reported that there are specific criteria that a patient must meet in order to be referred for a frenectomy. Eight responses (22%) stated that the infant must be seen by a primary care physician, midwife, public health nurse, or lactation consultant to obtain a referral to a specialist for the procedure. It was noted that some dentists self-refer and that this may be the case for other practitioners, while other dentists require a physician referral. Three (8%) responses indicated that, to be eligible for frenectomy, the mother must be attempting to breastfeed. Specific physical criteria considered by assessors for referring a patient for frenectomy include a restrictive lingual frenulum causing restricted tongue mobility that interferes with feeding resulting in feeding issues (including nipple pain, inability to maintain latch, inadequate milk transfer, inability to main milk supply, and/or digestive problems). Two (6%) respondents indicated that the patient must be formally diagnosed with ankyloglossia to be eligible for the procedure.

Criteria That Exclude Patients From Frenectomy

Most of the survey respondents (23 of 36, or 64%) reported that there are criteria that would exclude patients from receiving a frenectomy procedure in their jurisdictions. One respondent indicated that procedures are not typically conducted until the infant has had feeding issues for four months. On the contrary, other respondents (six of 36, or 17%) explained that, if the child is older than a few months, local physicians may be hesitant to perform a frenectomy. These patients may receive a subsequent referral to a specialized physician, which may involve longer wait times. Objective conditions such as the risk of bleeding, poor infant development, medical instability, health conditions preventing performing the procedure, and certain types of malocclusion that could be misdiagnosed as ankyloglossia may be incompatible with performing a frenectomy. Further considerations that may preclude frenectomy include an inability to understand the conduct or outcomes of the procedure, a previous frenectomy, the absence of an assessment score or outcome indicative of ankyloglossia, and the determination of an underlying cause of breastfeeding issues not related to ankyloglossia.

Qualifications Required to Perform Frenectomy

In Canada, physicians (including dentists) perform frenectomy procedures. The Canadian Paediatric Society position statement notes that a referral to an ear, nose, and throat specialist or a physician with experience performing frenotomies should be made.⁵ One study from Nova Scotia reported that the most common surgical procedure performed by a dentist is a frenectomy (29.4% of dentists reported they performed this procedure).¹⁸ No literature was identified on the involvement of other health care practitioners in conducting frenectomies. Two (6%) respondents indicated a desire to allow non-physicians to conduct the procedure, but no information was shared regarding the current involvement of other types of practitioners in performing frenectomies, although one respondent indicated that lactation consultants may assist.

Perceived Barriers to and Facilitators of the Optimal Diagnosis and Treatment of Ankyloglossia

No literature on barriers or facilitators to the optimal assessment and diagnosis of ankyloglossia or appropriate patient selection for frenectomy was identified. Survey questionnaire responses are summarized by barriers and facilitators below. Factors impacting assessment and diagnosis, and treatment, are presented together.

Barriers

Survey respondents were asked what barriers to the optimal diagnosis and treatment of ankyloglossia were present in their jurisdiction. Table 3 summarizes the responses. A substantial proportion of respondents (44% to 83%) indicated that each of the pre-specified factors were considered to be relevant barriers in their jurisdictions.

Table 3: Barriers to the Optimal Diagnosis and Treatment of Ankyloglossia

Barrier	Proportion of “Yes” Responses^a(%)
No consensus across clinical specialties regarding how to manage patients with ankyloglossia	30/36 (83%)
Lack of guidelines on how to assess and diagnose ankyloglossia	29/36 (81%)
Lack of guidelines on the treatment of ankyloglossia	28/36 (78%)
Lack of funding	25/36 (69%)
Lack of access to medical expertise (e.g., breastfeeding specialists, lactation consultants)	25/36 (69%)
Lack of dedicated facilities for newborn and pediatric care, and breastfeeding care	18/36 (50%)
Lack of rural and/or remote care	16/36 (44%)

^a To survey question: “What are the barriers to the optimal diagnosis and treatment of ankyloglossia?”

Additional barriers noted by respondents included the perception that some practitioners may consider tongue-tie as clinically irrelevant, resulting in lack of access to care. Lack of awareness among practitioners about ankyloglossia was highlighted by survey respondents, regarding how to diagnose it, and how to provide breastfeeding support and follow-up lactation care (to support continuity of care). Lack of collaboration among practitioners assessing patients and those performing the procedure, and a general lack of interdisciplinary care for ankyloglossia, were also perceived barriers. Survey respondents also noted that many families do not have a regular family doctor and must visit a walk-in clinic to get a referral, which may take extra time and delay treatment. For patients in rural and remote areas, they may have to travel to an urban area to receive treatment. According to one survey respondent, some patients are being referred out of province for care. If the patient cannot afford the cost of travel, they may not have access to optimal treatment. Further, in some jurisdictions, frenectomies may be primarily performed by dentists and must be paid for out of pocket unless the patient has private insurance coverage. Another barrier noted by respondents is potentially prohibitive wait times associated with seeking specialist care. Lack of education on breastfeeding for health care practitioners was also a noted obstacle to optimal care.

Facilitators

In contrast to barriers, survey respondents were asked what facilitators of the optimal diagnosis and treatment of ankyloglossia were present in their jurisdiction. The responses are summarized in Table 4. A substantial proportion of respondents (47% to 86%) indicated that each of the pre-specified factors were considered to be relevant facilitators in their jurisdictions.

Table 4: Facilitators of the Optimal Diagnosis and Treatment of Ankyloglossia

Facilitator	Proportion of “Yes” Responses^a
Availability of dedicated facilities for newborn and pediatric care, and breastfeeding care	17/36 (47%)
Availability of care in rural and remote health care settings	17/36 (47%)
Adherence to guidelines on ankyloglossia treatment	22/36 (61%)
Adherence to guidelines on how to assess and diagnose ankyloglossia	23/36 (64%)
Availability of funding (e.g., frenectomies performed in private dental offices and covered by public funding)	24/36 (66%)
Availability of specialized medical expertise (e.g., breastfeeding specialists, lactation consultants)	27/36 (75%)
Awareness of guidelines on ankyloglossia treatment	28/36 (78%)
Awareness of guidelines on how to assess and diagnose ankyloglossia	31/36 (86%)

^a To survey question: “What are the barriers to the optimal diagnosis and treatment of ankyloglossia?”

Other facilitators noted by survey respondents included the ability of non-physicians to refer patients for the procedure and specialized care, and for non-physicians to perform the frenectomy procedure. As well, the availability of follow-up breastfeeding care was perceived to support optimal care. Lastly, the provision of education on ankyloglossia for health care practitioners involved in the management of ankyloglossia was suggested as a facilitator by respondents.

Temporal Trends Related to the Use of Frenectomy to Treat Ankyloglossia Across Canadian Jurisdictions

Population-based data and anecdotal accounts of the current landscape for the diagnosis and treatment of ankyloglossia from survey respondents suggest a temporal increase in the rate of diagnosis of ankyloglossia and performance of frenectomy procedures.

One population-based study in British Columbia reported an increase in the rate of ankyloglossia from 5.0 per 1,000 live births in 2004 to 8.4 per 1,000 live births in 2013.¹⁶ The rate of frenectomies also increased from 2.8 per 1,000 live births to 5.3 per 1,000 live births.¹⁶ The study attributed the increase in the diagnosis of ankyloglossia to an increased surveillance secondary to the focus on breastfeeding initiation.

In follow-up to the British Columbia study,¹⁶ an analysis of all hospital-based live births in all Canadian jurisdictions, excluding Quebec, was conducted using Canadian Institute for Health Information data.¹⁰ The study reported that rates of diagnosed ankyloglossia increased in Canada from 6.86 per 1,000 live births in 2002 to 22.6 per 100 live births in 2014. The study also observed an increase in frenectomy rates for infants diagnosed with ankyloglossia from 54.7% in 2002 to 63.9% in 2014.¹⁰ The study compared jurisdictional rates of diagnosis to those from British Columbia, noting three-fold higher rates of ankyloglossia in Saskatchewan, Alberta, and the Yukon, and three- to four-fold higher rates of frenectomy in the Yukon, Alberta, and Saskatchewan.¹⁰ The lowest rates of ankyloglossia were observed in British Columbia, with similar rates in Nunavut, Newfoundland and Labrador, and Manitoba. Similarly, British Columbia had the lowest rates of frenectomy procedures, with the exception of Newfoundland and Labrador, and Nunavut. Overall, the study authors describe a rapid temporal increase in ankyloglossia and frenectomy rates in the observation period, and noted substantial regional variation in the rates of diagnosis and treatment of ankyloglossia. They suggest that the change could be attributed to an increased emphasis on breastfeeding initiation before hospital discharge.¹⁰ Notably, the study does not capture births outside of the hospital, or ankyloglossia diagnosis and frenectomy procedures conducted after hospital discharge.¹⁰

Although based on anecdotal evidence, 22 of 36 (61%) survey respondents from jurisdictions including Nova Scotia, Ontario, Prince Edward Island, Nunavut, Newfoundland, Saskatchewan, Quebec, and Manitoba reported a noticeable increase in the uptake of frenectomies in their jurisdictions. However, all of the respondents said they could not confidently quantify the change in the number of frenectomy procedures performed in their jurisdictions.

Survey observations about the drivers behind increased rates of ankyloglossia and frenectomy included:

an increased quantity of referrals from lactation consultants
increased provider and parental awareness about the impact of ankyloglossia on breastfeeding
the increased importance placed on breastfeeding, and the motivation to initiate and continue breastfeeding through challenges
motivation to seek care to resolve breastfeeding issues
an increase in the number of providers offering the procedure
increased networking and dissemination of knowledge (e.g., national conferences) among health care professionals managing ankyloglossia
the proliferation of private practices offering laser-based releases
patient self-referral and self-diagnosis resulting from increased patient awareness might contribute to increased rates, in contradiction to the aforementioned expectation that referral must come from a certified practitioner.

Conversely, referral to musculoskeletal care or other physical interventions, when indicated, is perceived to have reduced the number of procedures.

As far as trends related to conducting frenectomy, multiple survey (nine of 36, or 25%) respondents observed that later-born children in families with another child with ankyloglossia are often brought in for assessment because of perceptions around the heritability of the condition, and that they are more likely to receive early assessment and undergo the procedure. Two of 36 (6%) respondents noted that there has been an increase in inter-professional collaboration among health professionals providing care such as lactation consultants and family doctors. Some (11 of 36, or 31%) observed that the procedure is rarely performed on non-breastfeeding infants, and that perception of the necessity of frenectomy and perceived eligible time frame (e.g., prior to hospital discharge) may vary by practitioner. Two respondents (6%) suggested that frenectomy is increasingly performed by dentists versus other practitioners, often necessitating patient out-of-pocket payment for the procedure. It was also indicated by five respondents (14%) that there might be a trend toward over-performance of frenectomy, as well as re-surgery for unsuccessful procedures, given the simplicity of the procedure compared to other strategies to support breastfeeding. Others (2 of 36, or 6%) observed increased utilization of laser-based techniques versus scissor or scalpel.

Limitations

This Environmental Scan presents an overview of current practices for ankyloglossia diagnosis and frenectomy procedures in Canada.

An assessment of treatment effectiveness or outcomes was not within the scope of this report. In addition, the guidance documents identified and summarized in this report were not subject to quality appraisal. The objective was to understand current practices, so no restrictions were placed on the types of guidance documents summarized in the report. Thus, their inclusion is for information purposes regarding current practices, and we are unable to comment on the quality of these resources. This report does not address potential differences in practices to address anterior versus posterior ankyloglossia.

The findings of this report are based on a limited literature search and survey responses from Canadian jurisdictions. Although most Canadian provinces and territories were represented, there were no informants from New Brunswick, Yukon, and the Northwest Territories. In addition, with the exception of Ontario, less than 10 responses were received from the other jurisdictions that participated. Findings reflect the individual perspectives of the survey respondents and literature identified and may not represent all Canadian or local contexts or perspectives.

There was a lack of data available regarding temporal trends related to the use of frenectomy to treat ankyloglossia outside of the hospital setting. As a result, statements on changes in the rate of procedures may not be generalizable to other settings and may underestimate the use of the procedure given the volume conducted outside of the hospital.

Conclusions

This Environmental Scan set out to capture perspectives on current practices related to the assessment and diagnosis of ankyloglossia and the use of frenectomy procedures from Canadian jurisdictions.

Some respondents reported that they use specific guidance and criteria to help diagnose ankyloglossia. Several resources were highlighted, but other tools commonly cited in the literature, such as the Coryllos grading tool and the Bristol Tongue Assessment Tool, were not acknowledged.^26,27 The varied approach to diagnosis indicated by this Environmental Scan’s findings is consistent with conclusions of an earlier review. The review summarized articles regarding the diagnostic criteria used for ankyloglossia and the methods used for diagnosis in studies of ankyloglossia prevalence, and reported substantial variation in diagnostic criteria and the age of assessment used.²⁸ Multiple reports have commented on variations in international appropriateness criteria and guidance for ankyloglossia management, noting that some countries recommend treatment, when appropriate, while others do not endorse it.^10,26 Nearly all the survey respondents agreed that there is a need for additional guidance regarding the assessment, diagnosis, and treatment of ankyloglossia. While several societies and organizations have issued statements on ankyloglossia, they tend to provide broad suggestions (e.g., not supportive of universal treatment, treatment recommended in circumstances where breastfeeding is impaired) rather than specific direction for patient management.

Fewer respondents reported using guidance documents to inform the patient selection for treatment, although many acknowledged that there are certain criteria that would exclude patients from gaining access to a frenectomy in their jurisdictions. The findings indicated that physicians (including dentists) are the primary group performing frenectomies in Canada. While this may be the case, studies on the effectiveness of frenectomy have reported that a range of health professionals — family, neonatal, and pediatric doctors; general, pediatric, or specialty surgeons; and lactation or specialist consultants — are involved in conducting the procedure.^29,30 Several survey respondents noted interest in the expansion of the acceptable disciplines able to perform frenectomy.

Beyond guidance, substantial input on factors affecting the optimal diagnosis and treatment of ankyloglossia was provided. Some common barriers expressed related to a perceived lack of guidance, funding, expertise, and education for practitioners, appropriate facilities, access to and cost of care for patients living in rural and remote areas, consensus on how to deliver care, insufficient collaboration among practitioners, and lengthy wait times. In contrast, insight obtained on facilitators may be useful in the development of strategies to address these challenges. Of note, improved awareness of and monitoring of adherence to guidelines, enhanced funding, greater availability of specialized care and facilities for breastfeeding in all geographical settings, expansion of the disciplines involved in referral and treatment, measures to support continuity of care, and better education on ankyloglossia care for health care practitioners were suggested to be supportive of optimal care.

Although no jurisdiction provided concrete data on the temporal trends for the use of frenectomy procedures in their jurisdiction via the survey questionnaire, most of the respondents reported an anecdotal increase. These observations were corroborated by two population-based studies that observed a significant temporal increase in both diagnosing ankyloglossia and conducting frenectomy procedures across Canada.^10,16 Similar usage patterns have been reported in the US, with a more than 10-fold increase in the rate of frenectomy procedures performed from 1997 to 2012.³¹ The increase in the uptake of frenectomy procedures has been suggested to stem from a renewed emphasis on breastfeeding encouraged by health care providers and organizations, including the World Health Organization.^1,2 Input on the drivers behind the increased diagnosis and treatment of ankyloglossia from survey respondents was consistent with this, also suggesting the contribution of increased patient, family, and practitioner awareness about the condition, and the growth in the number of practitioners able to refer to and conduct the procedure.

An increase in the number of public queries and non-clinical media coverage of ankyloglossia has been reported, while formal research and evidence generation research on the topic has slowed.^32,33 Future research initiatives have been suggested to further the understanding of the impact of ankyloglossia on infant feeding, develop better evidence on the effectiveness of frenectomy, determine the impact of variation in practitioner perception of the need for frenectomy on current practice, and on the potential for standardization of diagnostic and treatment practices across professions and settings.^10,31,34

Appendices

References

Government of Canada. Breastfeeding & infant nutrition. 2018; https://www.canada.ca/en/public-health/services/health-promotion/childhood-adolescence/stages-childhood/infancy-birth-two-years/breastfeeding-infant-nutrition.html. Accessed 2018 Aug 30.
World Health Organization. Exclusive breastfeeding for six months best for babies everywhere. 2011; http://www.who.int/mediacentre/news/statements/2011/breastfeeding_20110115/en/. Accessed 2018 Aug 30.
Gionet L. Health at a glance: breastfeeding trends in Canada. Ottawa (ON): Statistics Canada; 2015: https://www.statcan.gc.ca/pub/82-624-x/2013001/article/11879-eng.htm. Accessed 2018 Aug 30.
Statistics Canada. Study: breastfeeding trends in Canada, 2003 and 2011/2012. 2013; https://www150.statcan.gc.ca/n1/daily-quotidien/131125/dq131125c-eng.htm. Accessed 2018 Aug 30.
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Amir LH. Breastfeeding--managing 'supply' difficulties. Aust Fam Physician. 2006;35(9):686-689.
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Chinnadurai S, Francis DO, Epstein RA, Morad A, Kohanim S, McPheeters M. Treatment of ankyloglossia for reasons other than breastfeeding: a systematic review. Pediatrics. 2015;135(6):e1467-1474.
Lisonek M, Liu S, Dzakpasu S, Moore AM, Joseph KS. Changes in the incidence and surgical treatment of ankyloglossia in Canada. Paediatr Child Health. 2017;22(7):382-386.
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Protocol #11: guidelines for the evaluation and management of neonatal ankyloglossia and its complications in the breastfeeding dyad. Rochelle (NY): Academy of Breastfeeding Medicine; 2016: https://abm.memberclicks.net/assets/DOCUMENTS/PROTOCOLS/11-neonatal-ankyloglossia-protocol-english.pdf. Accessed 2018 Aug 31.
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Stanford Medicine. Frenotomy. 2017; https://med.stanford.edu/newborns/professional-education/frenotomy.html Accessed 2018 Aug 30.
Joseph KS, Kinniburgh B, Metcalfe A, Razaz N, Sabr Y, Lisonkova S. Temporal trends in ankyloglossia and frenotomy in British Columbia, Canada, 2004-2013: a population-based study. CMAJ Open. 2016;4(1):E33-40. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4866928/. Accessed 2018 Aug 30.
Hosted in Canada surveys. 2017; https://www.hostedincanadasurveys.ca/. Accessed 2018 Aug 30.
Ghiabi E, Matthews DC. Periodontal practice and referral profile of general dentists in Nova Scotia, Canada. J Can Dent Assoc. 2012;78:c55.
Health promotion guideline: breastfeeding healthy term infants (v3). Vancouver (BC): Perinatal Services BC; 2015: http://www.perinatalservicesbc.ca/Documents/Guidelines-Standards/HealthPromotion/BreastfeedingHealthyTermInfantGuideline.pdf. Accessed 2018 Aug 30.
Hazelbaker A. Assessment tool for lingual frenulum function (ATLFF). 2017; http://www.alisonhazelbaker.com/shop/hatlff-hazelbaker-assessment-tool-for-lingual-frenulum-function. Accessed 2018 Aug 30.
Dobrich C. Frenotomy decision tool for breastfeeding dyads. 2017; https://xa.yimg.com/kq/groups/2bxq7c3tfNVIyJvJ3w--/eUF.YcfufNXgAbegQ9c-/name/Frenotomy+tool+Dobrich+FDTBD_2017.pdf. Accessed 2018 Aug 30.
Jewish General Hospital. Tongue-tie/ankyloglossia and upper lip-tie patient handout. 2018; http://www.jgh.ca/uploads/pfrc/Breastfeeding%20clinic/Ankyloglossia_Frenotomy%20and%20lip-tie-handout%20jul2018.pdf. Accessed 2018 Aug 30.
Kotlow L. Oral diagnosis of abnormal frenum attachments in neonates and infants: evaluation and rreatment of the maxillary and lingual frenum using the erbium: YAG laser. 2004; https://scinapse.io/papers/2335939981. Accessed 2018 Aug 30.
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Lawrence RA, Lawrence RM. Breastfeeding: a guide for the medical profession. 6th ed. Maryland Heights (MO): Mosby, Inc; 2005.
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About this Document

Authors: Kelsey Seal, Teo Quay, Kaitryn Campbell

Acknowledgements: Lesley Dunfield, Gino De Angelis, Paula Murray

Cite As: Ankyloglossia diagnosis and treatment in Canada: an environmental scan. Ottawa: CADTH; 2018. (Environmental scan; no. 79).

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