International Journal of Speech-Language Pathology

ISSN: (Print) (Online) Journal homepage: https://www.tandfonline.com/loi/iasl20

A systematic review: The effects of frenotomy

on breastfeeding and speech in children with
ankyloglossia

Alison Visconti, Emily Hayes, Kristen Ealy & Donna R. Scarborough

To cite this article: Alison Visconti, Emily Hayes, Kristen Ealy & Donna R. Scarborough
(2021): A systematic review: The effects of frenotomy on breastfeeding and speech in
children with ankyloglossia, International Journal of Speech-Language Pathology, DOI:
10.1080/17549507.2020.1849399

To link to this article: https://doi.org/10.1080/17549507.2020.1849399

Published online: 27 Jan 2021.

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International Journal of Speech-Language Pathology, 2021; Early Online: 1–15

A systematic review: The effects of frenotomy on breastfeeding and

speech in children with ankyloglossia

ALISON VISCONTI, EMILY HAYES, KRISTEN EALY & DONNA R. SCARBOROUGH

Department of Speech Pathology and Audiology, Miami University, Oxford, UK

Abstract
Purpose: The primary objective of this systematic review was to determine if frenotomy for ankyloglossia improves breast-
feeding or speech outcomes in infants and children ages birth to 12.
Method: Literature selection focussed on the presence of ankyloglossia, reported as either posterior or submucosal, and
the impact of surgical treatment. The two populations that were included involve infants who were breastfeeding and chil-
dren with speech delays. Six search engines were utilised (PubMed, Medline, Cochrane Database, CINHAL Plus, ERIC
and PsychINFO). The selected articles critically examined study characteristics, measurement tools, outcome measures,
design, and summary of results, and bias.
Result: Five articles met the inclusion criteria related to infants who had undergone a frenotomy and who were examining
changes in breastfeeding outcomes and two articles met the inclusion criteria for changes in speech production following
a frenotomy.
Conclusion: Research supports the use of frenotomy in children with ankyloglossia to reduce nipple pain and improve
maternal self-efficacy during breastfeeding. The classification of ankyloglossia, assessment tools used, age and timing of
frenotomy, in terms of breastfeeding improvements were inconsistent across the studies. Ankyloglossia release for children
with speech delays is currently inconclusive due to lack of objective data and research quality. Overall, the review also
revealed inconsistent definitions of ankyloglossia severity, standardised outcome measures and research protocols.

Keywords: Speech; swallowing; treatment; systematic review

Introduction ankyloglossia occurs when the insertion of the lingual

Ankyloglossia, also known as tongue-tie, is a congeni-
tal abnormality that occurs when the connective tis-
sue, or frenulum, positioned between the floor of the
mouth and the underside of the tongue is abnormally
short, thick, or tight (Messner & Lalakea, 2002;
Emond et al., 2014). Because of this tethering, the
tongue is restricted in mobility and/or range of
motion, which may interfere with breastfeeding or
speech intelligibility (Berry, Griffiths, & Westcott,
2012; Buryk, Bloom, & Shope, 2011; Ghaheri et al.,
2017; Messner & Lalakea, 2002; O’Callahan,
Macary, & Clemente, 2013; Walls et al., 2014). The
location of the frenulum on the undersurface of the
tongue determines one type of classification of anky-
loglossia, commonly described as anterior or poster-
ior. Anterior ankyloglossia encompasses two types
(Type I and Type II) (Coryllos, Genna, Salloum, &
American Academy of Pediatrics, 2004). Type I anky-
loglossia is when the insertion of the lingual frenulum
is located directly on the tip of the tongue. Type II
frenulum is on the inferior surface at the blade of the
tongue, which is located slightly behind the tongue
tip (Coryllos et al., 2004). Posterior ankyloglossia
includes Type III and Type IV. Type III ankyloglossia
occurs when the lingual frenulum is located inferiorly
at the mid-tongue. Type IV ankyloglossia, also known
as a submucosal frenulum, restricts movement at the
base of the tongue. Type IV ankyloglossia are also
described as the tongue appearing similar to a flat
mound and the frenular tissue is not visible (Coryllos
et al., 2004). Both Type III and Type IV ankyloglossia
have been described as having frenular tissue that is
more inelastic than the first two types (Coryllos
et al., 2004).
Ankyloglossia may be classified into levels of sever-
ity in a variety of ways. For instance, first described
by Kotlow (1999), measurements of the lingual
frenulum were obtained in children 18 months to
14 years using a device that provides exact lengths (in
mm) of the lingual frenulum between the origin on

Correspondence: Dr. Donna R. Scarborough, Department of Speech Pathology and Audiology, Miami University, 26 Bachelor Hall, Oxford, OH 45056,
UK. E-mail: scarbod@miamioh.edu

ISSN print/ISSN online ß 2021 The Speech Pathology Association of Australia Limited
Published by Taylor & Francis
DOI: 10.1080/17549507.2020.1849399
2 A. Visconti et al.
the floor of the mouth and insertion point in the
underside of the tongue. This original work suggested
that greater than 16 mm should be considered a
“normal” length. In individuals whose frenulum
length measured between 0 and 16 mm, severity rat-
ings of mild, moderate, severe and complete were
assigned based on the length of the frenulum in incre-
ments of 4 mm. For example, a rating of “complete”
was assigned to individuals whose frenulum length
was between 0 and 3mm (Crippa, Paglia, Ferrante,
Ottonello, & Angiero, 2016; Kotlow, 1999; Yoon
et al., 2017). Some researchers have used the
Hazelbaker Assessment Tool for Lingual Frenulum
Function (HATLFF). Early studies used the original
version of the HATLFF which had a rating scale of
0–24 including 5 appearance items and 7 function
items (HATLFF, 1998, unpublished thesis). The
HATLFF has undergone reliability testing and sug-
gested revisions were made. The revised HATLFF
removed four function items thus the rating scale was
changed to 0–16 (Amir, James, & Donath, 2006).
Researchers then applied their own judgement to rate
severity. Regardless, the original intent of the
HATLFF is to determine the need for frenotomy
rather than rate severity. In addition, the Bristol
Tongue Assessment Tool (BTAT) was developed
using principles from the HATLFF to rate severity
and make the assessment process more cohesive. The
BTAT is a four item, eight-point assessment tool
which measures tongue tip appearance, attachment
of the frenulum to the lower gum ridge, lift of tongue
with mouth wide and protrusion of tongue (Ingram
et al., 2015). The Tongue-tie and Breastfed Babies
(TABBY) assessment tool was then developed to pro-
vide a pictorial representation of the BTAT measure-
ment tool (Ingram, Copeland, Johnson, & Emond,
2019). A score of eight suggests normal tongue func-
tion. A six or seven is considered borderline. A score
at or below a five indicates impairment, and a score
between 0 and 3 indicates more severe ankyloglossia
(Ingram et al., 2015, 2019).
The incidence of anterior ankyloglossia is variable
within the paediatric population. Among neonates
and infants the incidence is reported to be between
1.7% and 4.8% with a male to female ratio of
approximately 3:1 (Lalakea & Messner, 2003;
Messner, Lalakea, Aby, Macmahon, & Bair, 2000).
Another study reported incidence to be between
0.1% and 10.7% for neonates, infants and adoles-
cents (Geddes et al., 2010). Breastfeeding difficulties
have been reported in 12% to 44% of infants with
anterior ankyloglossia (Griffiths, 2004; Ngerncham
et al., 2013; Segal, Stephenson, Dawes, & Feldman,
2007). In one study, 11 out of 15 (70%) of young
children with anterior ankyloglossia had co-occurring
articulation difficulties with the remaining four chil-
dren demonstrating normal speech production
(Messner & Lalakea, 2002). Posterior ankyloglossia is
a clinical phenomenon that has only recently been
described in the literature (Ghaheri, Cole, Fausel,
Chuop, & Mace, 2017; O’Callahan et al., 2013;
Walls et al., 2014), and its efficacy has become a com-
mon parental question to clinicians of late. In general,
prevalence rates of ankyloglossia are difficult to deter-
mine due to limited documentation and a lack of
standardised and reliable measures for classification
(Geddes et al., 2010).
Research reports indicate that the surgical inter-
vention, called a frenotomy, involves cutting the
frenulum found between the inferior surface of the
tongue and the floor of the mouth via a laser or scal-
pel. A frenotomy for ankyloglossia primarily occurs
before 6 months, but ranges up to 6 years of age
(Berry et al., 2012; Buryk et al., 2011; Emond et al.,
2014; Ghaheri et al., 2017; Messner & Lalakea,
2002; O’Callahan et al., 2013; Walls et al., 2014).
Frenectomies and frenuloplasties are alternative sur-
gical interventions for ankyloglossia. A frenectomy is
when the frenulum is completely excised often utilis-
ing a scalpel or laser. A frenectomy can be difficult to
perform on newborns and infants and is more often
performed on older children (Junqueira et al., 2014).
A frenuloplasty is a more complex surgical technique
conducted under general anaesthesia that restruc-
tures the frenulum using plastic surgery techniques in
order to minimise postoperative scar tissue develop-
ment (Baker & Carr, 2015). At this time, little
research exists regarding optimal timing for
intervention.
Reported breastfeeding difficulties associated with
ankyloglossia include nipple pain, poor latch, reduced
milk transfer, infant dehydration and decreased dur-
ation of breastfeeding (Geddes et al., 2010). A num-
ber of clinical measurement tools have been utilised
to qualify these maternal and infant breastfeeding fac-
tors. Examples of such tools include Breastfeeding
Self-Efficacy Scale-Short Form (BSES-SF),
Hazelbaker Assessment Tool for Lingual Frenulum
Function (HATLFF) Infant Breastfeeding
Assessment Tool (IBFAT), LATCH Assessment
Tool, Pain Visual Analogue Scale (VAS) and Short-
Form McGill Pain Questionnaire (SF-MPQ). Brief
descriptions of these tools are found in Table I.
Reports of speech difficulties and intelligibility out-
comes in the literature are mixed. In older children
who are verbal, reported speech difficulties associated
with ankyloglossia include limited tongue mobility
and decreased intelligibility (Messner & Lalakea,
2002; Walls et al., 2014). In contrast, a recent study
has shown that a group of pre-school children who
were diagnosed before 6 months of age with ankylo-
glossia (some treated and others not treated) showed
similar speech production abilities and intelligibility
outcomes between the treated and untreated groups
and to a normal control group (Salt, Claessen,
Johnston, & Smart, 2020). Regardless, in either situ-
ation, breastfeeding challenges or speech delays, con-
cerned parents will seek medical intervention from a
 Effects of frenotomy 3

Table I. Outcome measures/measurement tools descriptions.

Breastfeeding Self-Efficacy Scale-Short A 14-item survey given to mothers to measure breastfeeding efficacy and maternal
Form (BSES-SF) confidence in her ability to breastfeed (Emond et al., 2014; Ghaheri et al., 2017).
Hazelbaker Assessment Tool for Lingual A subjective measure used to grade severity of ankyloglossia consisting of five appearance
Frenulum Function (HATLFF) items (tongue appearance when lifted, frenulum elasticity, length of frenulum, attachment
of lingual frenulum to the tongue) and seven function items including (lateralisation, lift
of tongue, protrusion of the tongue, spread of anterior tongue, cupping of the tongue,
peristalsis, and tongue retraction (Buryk et al., 2011).
Infant Breastfeeding Assessment A subjective measure of infant feeding behaviours completed by the mother including
Tool (IBFAT) questions regarding rooting, necessary stimulation, time needed to latch, feeding
patterns, and satisfaction (Buryk et al., 2011; Emond et al., 2014).
LATCH Scale Used to measure breastfeeding effectiveness, and a score of
8 indicated breastfeeding
difficulties including ability to latch, audible swallowing, type of nipple, comfort, and
hold (Emond et al., 2014).
Pain Visual Analogue Scale (VAS) A 10 cm line used to rate pain with zero indicating no pain and 10 indicating the worst pain
(Emond et al., 2014).
Short-Form McGill Pain Questionnaire Assesses maternal nipple pain through sensory and affective aspects of pain, visual analog
(SF-MPQ) scale, and descriptors for present pain intensity measure (Buryk et al., 2011).

speech pathologist or other healthcare providers.
Much controversy exists regarding the necessity and
effectiveness of a posterior and/or anterior frenotomy
to improve speech and breastfeeding. Therefore, the
purpose of this paper is to complete a systematic
review to explore the outcomes of frenotomy on
breastfeeding and speech production.
Method
Six different combinations of search terms were
developed and entered into six databases including:
PubMed, Medline, Cochrane Database for
Systematic Reviews, CINAHL Plus, ERIC and
PsychINFO. The inclusion criteria consisted of chil-
dren birth to 12 years who presented with a history of
ankyloglossia regardless of the subtype (anterior or
posterior) or classification and whose primary lan-
guage was English. Exclusion criteria involved articles
whose participants were non-English speakers or
were over the age of 12. The rationale for only includ-
ing English language samples was to ensure that sam-
ple populations had consistent speech acquisition
expectations. Although the year of publication was
not constrained, the publication dates for all articles
was between the years of 2002 and 2017 (Table II).
The initial search for evidence identified 1052
articles with the last date searched as 9/18/2018
(Table II). A close examination of these 1052 articles
revealed that 802 were duplicates and subsequently
discarded. Examination of the titles of the remaining
articles resulted in elimination of another 194 articles
because the articles were unrelated to the clinical
question of interest. Evaluation of the abstracts from
the remaining 56 articles resulted in eight articles
selected for full text reviews. Following the full text
reviews, elimination of one article occurred because
the article included non-English speakers and indi-
viduals older than 12 years. Each of the remaining
seven fulfilled all of the requirements to be included
in the review and each underwent a critical appraisal
(Figure 1).
Analysis by two independent teams of researchers
reported on the validity, reliability and applicability
for each of the final seven studies allowed for the assign-
ment of individual evidence level ratings (Table III).
Information was extracted from each included study
on: (1) study characteristics including sample size,
population, methodology, data collection, follow-up,
independent and dependent variables, and signifi-
cant results (2) research design including blinding
and sampling, (3) types of outcome measures uti-
lised (nipple pain, self-efficacy, latch, milk transfer,
tongue mobility, speech sound production) and
associated instrumental validity. Consideration of
participant sampling and research design during the
critical appraisal process at the study level addressed
the risk of bias. Whereas the analysis of attrition,
influence of confounding variables and time between
follow-up periods addressed the risk of outcome
level bias. Human subject’s protocol for this manu-
script was not required per our institutional
review board.
Result
Breastfeeding articles
Seven total articles met the inclusion criteria and
underwent critical appraisal. Of these seven articles
five addressed outcomes related to maternal and
infant breastfeeding factors (Table IV provides
detailed summary). These 5 articles included 3 rand-
omised controlled trials, one prospective study and
one longitudinal study with evidence levels ranging
from 2a to 4b. All five of the studies pertaining to
breastfeeding used a frenotomy as the intervention.
The study sample sizes ranged from 30 to 299 partici-
pants. Infants were enrolled between birth to five day
post-birth and interventions were conducted 2–46
weeks post-frenotomy. Measurement data was col-
lected pre-frenotomy in four out of the five studies,
immediately post-frenotomy in three out of the five
studies with long-term study results noted in three
out of the five studies. All studies used at least two
outcome measures and one utilised five. All studies
use some pain assessment as an outcome measure
whether it was the VAS, SF-MPQ, or included in a
questionnaire. Statistical significance was reported on
at least one outcome measure from each of the five
articles. Only one study reported selection bias
related to maternal motivation and two studies
4 A. Visconti et al.

Table II. Search Strategy including the databases, date of most recent search, terms utilised, and search parameters.
Date of most Limits, filters, and search
Search databases recent search Search terms date parameters
PubMed 9/18/2018 (Ankyloglossia OR Tongue Tie OR Lingual Frenulum) Publication dates or search
Medline AND (Frenulectomy OR Frenotomy OR Frenectomy) dates: any
Cochrane Database for AND (Feeding OR Swallowing OR Breastfeeding) English language
Systematic Reviews (Ankyloglossia OR Tongue Tie OR Lingual Frenulum)
CINAHL AND (Frenulectomy OR Frenotomy OR Frenectomy) Paediatric evidence only:
PsycINFO AND (Speech) children (birth – 12 years)
ERIC (Ankyloglossia OR Tongue Tie OR Lingual Frenulum) Other limits or filters: N/A
AND (Feeding OR Swallowing OR Breastfeeding)
(Ankyloglossia OR Tongue Tie OR Lingual Frenulum)
AND (Speech)
(Frenulectomy OR Frenotomy OR Frenectomy) AND
(Speech)
(Frenulectomy OR Frenotomy OR Frenectomy) AND
(Feeding OR Swallowing OR Breastfeeding)
Idenficaon

Records idenfied through Addional records idenfied

database searching through other sources
(n = 1052) (n = 0)

Records aer duplicates removed

(n = 250)
Screening

Records screened Records excluded

(n = 250) (n = 242)

Full-text arcles assessed Full-text arcles excluded,

for eligibility with reasons
Eligibility

(n = 8) (n = 1; included adult
data)

Studies included in
qualitave synthesis
(n = 7)
Included

Studies included in
quantave synthesis
(meta-analysis)
(n = 0)

Figure 1. Systematic search process for literature identification, review and exclusion (Adapted from PRISMA diagram; Moher, Liberati,
Tetzlaff, & Altman, The PRISMA Group, 2009).

Table III. Evidence level used to critically appraise each article that met the inclusion criteria.
Quality level Definition
1a or 1b Systematic review, meta-analysis, or meta-synthesis of multiple studies
2a or 2b Best study design for domain, randomised controlled trials
3a or 3b Fair study design for domain, controlled clinical trials, prospective cohort studies,
4a or 4b Weak study design for domain, qualitative study, retrospective cohort study; case–control study, longitudinal study,
cross-sectional study
5a or 5b General review, expert opinion, case report, consensus report, or guideline
5 Local Consensus
a ¼ good quality study; b ¼ lesser quality study.
Table IV. Summary of the five articles that were critically examined related to breastfeeding outcomes.
Buryk et al. (2011) Emond et al. (2014) Berry et al. (2012) Ghaheri et al. (2017) O’Callahan et al. (2013)
Study design, evidence level, number of participants, age of participants, summary of methodology
Research design Randomised control trial Randomised control trial Randomised control trial Prospective cohort study Longitudinal study
Evidence level 2a 2a 2b 3a 4b
Number of participants n ¼ 58 n ¼ 107 n ¼ 30 n ¼ 237 n ¼ 299
Population age Infants birth  30 days Infants birth – 2 weeks Infants five  115 days Infants birth 12 weeks Infants two  323 days
Summary of study Immed. post-frenotomy; 2 Pre-frenotomy, five days, and Pre-frenotomy, immed. Pre-frenotomy, 1 week post- Pre-frenotomy, and post-
methodology weeks, 6 months and 12 8 weeks post-frenotomy post-, and 3 months frenotomy and 1 month frenotomy
months post-frenotomy post-frenotomy post-frenotomy (unspecified timeline)

Outcome Measures/Measurement Tools used by each article

LATCH Scale X X
IBFAT X X X
BSES-SF X X
VAS X X
HATLFF X X X
SF-MPQ X
Numeric Scale for Pain X
Web based questionnaire X
Pediatrician assessment X

Results, statistical significance, study bias and outcome level bias

Results of intervention group Significant reduction in
nipple pain and
significant improvements
in IBFAT scores
following frenotomy
p Value Nipple pain:
p < 0.001
IBFAT:
p ¼ 0.029
Study bias Selection bias due to
maternal motivation
Outcome level bias Attrition bias due to loss of
participants during
follow-up
No significant difference in LATCH Significant immediate Significant improvement in Significant reduction in
scores; Significant increase in improvement in BSES-SF and VAS scores latching difficulties and
HATLFF and BSES scores for the breastfeeding following at 1 week and 1 month significant reduction in
intervention group at five days the frenotomy in post-intervention nipple pain at 1 week
post-intervention intervention group post-intervention
LATCH: p < 0.02 BSES-SF: Latch:
p ¼ 0.52 p < 0.001 p < 0.001
HATLFF: p < 0.001 Nipple pain:
p < 0.0001 VAS: p < 0.001
BSES: p < 0.001
p ¼ 0.002 p < 0.001
Did not self-report any bias Did not self-report any bias Did not self-report any bias Did not self-report any bias
Did not self-report any bias Did not self-report any bias Did not self-report any bias Recall bias due to length of
study and dates of
follow-up
Effects of frenotomy
5
6 A. Visconti et al.

reported attrition as being the primary reason for out- Discussion

come level bias.
Speech articles
The remaining two articles that met the inclusion cri-
teria and underwent a critical appraisal were related
to speech delays (Table V provides detailed sum-
mary). Of the two studies, one was a longitudinal
study and one was a retrospective study. However
despite different study designs the rigour of the
experiments classified both of these articles as 4b.
The intervention between these two articles differed.
One utilised frenotomy as the intervention, and the
other used a combination of frenotomy and frenulo-
plasty. One speech related article included 30 partici-
pants, and the other recruited 104 study participants.
One of the articles included children in their third
year of life who underwent a frenotomy within the
first month of life, while the second article included a
wider range of children between the ages of one and
12 years of age. One article collected measurement
data both pre-intervention and post-intervention, and
the other collected data only for post-intervention.
Both studies used at least two outcome measures for
their results. Each utilised a measure for tongue
mobility whether it was completed by a professional
or a parent. Statistical significance was found on at
least one outcome measure. Both studies had study
and outcome level bias related to their study designs
and through parent questionnaires.
Breastfeeding-specific outcome measures
The systematic review examining the body of evi-
dence pertaining to ankyloglossia release and breast-
feeding is moderately strong for improving
breastfeeding factors. The findings support the use of
frenotomy to reduce nipple pain and improve mater-
nal self-efficacy, which may facilitate further breast-
feeding attempts (Berry et al., 2012; Buryk et al.,
2011; Emond et al., 2014; Ghaheri et al., 2017;
O’Callahan et al., 2013). Each study identified
maternal nipple pain as a primary indicator for
breastfeeding difficulties and assessed changes in sub-
jective pain levels pre- and post-intervention. All five
studies regarding breastfeeding utilised maternal
report measurements to determine nipple pain. Of
the five related articles, all mothers reported overall
reduction in nipple pain; however, this finding was
only found to be statistically significant in three
articles (Buryk et al., 2011; Ghaheri et al., 2017;
O’Callahan et al., 2013). The studies, which eval-
uated changes in the mother’s self-efficacy, showed
mixed results. Two studies at the initial follow-up,
reported significant improvements in self-efficacy of
breastfeeding measured by the BSES-SF (Emond
et al., 2014; Ghaheri et al., 2017). However, when
the BSES-SF was readministered at the second fol-
low-up appointment the findings showed no differen-
ces between the control group and the intervention
group (Emond et al., 2014; Ghaheri et al., 2017).
Reduced maternal nipple pain and increased ability

Table V. Summary of the two articles that were critically examined related to speech outcomes.
Messner and Lalakea (2002) Walls et al. (2014)
Study design, evidence level, number of participants, age of participants, summary of methodology
Research design Longitudinal study Retrospective cohort study
Evidence level 4b 4b
Study size n ¼ 30 n ¼ 104
Number of participants Children ages one  12 years Children 36–47 months who had a
frenotomy during first 4 weeks of life
Summary of study methodology Pre-frenotomy/frenuloplasty and 3 months Retrospective analysis during child’s third
post-frenotomy/frenuloplasty year post-frenotomy
Outcome measures/measurement tools used by each article
Tongue mobility (professional) X
Speech evaluation X
Parent questionnaire X X
Tongue mobility (parental) X
Results, statistical significance, study bias and outcome level bias
Results Significant improvements in tongue Significant improvements in parental
protrusion, and tongue elevation post- perception of speech outcomes when
intervention; Significant improvements compared to the No Surgical
in parental perception of tongue Intervention Group
mobility and speech intelligibility post-
intervention
p Value Tongue protrusion: p < 0.01 Speech outcomes: p < 0.0001
Tongue elevation: p < 0.01
Tongue mobility: p < 0.01
Speech intelligibility: p < 0.01
Study bias Response bias due to use of parent Response bias due to use of parent
questionnaire questionnaire; selection bias due
retrospective nature of study
Outcome level bias Bias due to confounding variable Recall bias due to retrospective nature
(tympanostomy tube placement at the of study
time of frenotomy)
 Effects of frenotomy 7

to latch likely correlate with a mother’s self-efficacy confounding variable because of varying developmen-
and willingness to continue to try to breastfeed their tal abilities and breastfeeding expectations. Moving
infant. Consequently, maternal motivation to con- forward, the establishment of an optimal age range
tinue breastfeeding potentially impacts overall breast- for the use of a frenotomy for ankyloglossia would be
feeding outcomes and should be considered when beneficial for research and general interven-
looking at the clinical effectiveness of frenotomy to tion purposes.
improve breastfeeding.
However, this positive recommendation comes Speech
with a caveat. The primary measurement tool, the
The body of evidence for speech is low due to study
HATLFF is an outcome measure intended to identify
design and overall quality of the studies.
the need for frenotomy. The HATLFF has only min-
Furthermore, a paucity of research exists when exam-
imal peer reviewed publication support of its validity
ining ankyloglossia release and speech outcomes with
and some features would be difficult visually inspect
only two studies that met the inclusion and exclusion
at the bedside without instrumentation. Although the
criteria (Messner & Lalakea, 2002; Walls et al.,
content validity of the HATLFF is available, the
2014). Despite the contrast in design, both studies
HATLFF has not undergone a controlled validation
reported statistically significant improvements in
(Ballard, Auer, & Khoury, 2002). Therefore, solely
speech outcomes. One study found statistical signifi-
using this tool as a primary outcome measure con-
cant differences in speech outcomes between the sur-
founds the findings of the systematic review. The
gical and non-surgical group (Walls et al., 2014);
LATCH scale assessment tool, which mixes parame-
however, the reported speech outcomes were solely
ters related both to the infant (i.e. latch and audible
judged by parental perceptions making the responses
swallowing) and to the mother (i.e. type of nipple,
subject to bias. In another study, speech therapists
comfort and hold) was used in two studies (Berry
reported that nine out of 11 children demonstrated
et al., 2012; Emond et al., 2014). Although neither
speech improvements post-intervention (Messner &
study reported statistical significance, because the
Lalakea, 2002). However, the methodology would
duration between pre-intervention and immediate
not be able to be reproduced based on the vague
post-intervention measures were so closely intervaled,
descriptions. Further, the design lacked a standar-
the possibility for improvement measurements in
dised language sampling, blinding by the speech
LATCH scores is unlikely and therefore, may not
therapists, and control regarding simultaneous ear
reflect clinical positive changes.
tube placement. Anecdotal reports by clinicians and/
Three of the studies utilised randomised control
or families frequently indicate improvements in
research designs (Berry et al., 2012; Buryk et al.,
speech following frenotomy; however, research to
2011; Emond et al., 2014). Typical of this type of
support these claims does not exist. In contrast, a
research design in which treatment is initially with-
recent study conducted by Salt et al. (2020) found
held from the control group, the investigators were
that infants who were diagnosed before 6 months of
ethically committed to provide medical intervention
age have no speech intelligibility differences or sound
to families who qualified. As a result, the majority of
production differences as preschoolers when you
those within the control groups underwent a frenot-
compared the treatment versus non-treatment groups
omy at or before the first follow-up. However, the
to a normal control. Further, the authors also found
data from the control group participants that elected
that the untreated group who had been diagnosed
to receive a frenotomy remained with the data from
with ankyloglossia before 6 months of age, no longer
the participants who elected not to receive medical
met the tongue mobility criteria to be classified as a
intervention. Therefore, the reliability of long-term
having a tongue tie as a pre-school child (Salt et al.,
the follow-up data was no longer clean. In addition, a
2020). These findings are consistent with Ruffoli
potential placebo effect could contribute to perceived
et al. (2005), who described a natural recession of the
improvements in breastfeeding, thus possibly skewing
frenulum up to age 6.
overall results (Berry et al., 2012; Buryk et al., 2011).
Selection bias may also exist due to the high motiv-
Overall findings, breastfeeding and
ation of mothers who consented to be in the study
speech delays
and desired to continue breastfeeding their infant.
Although most infants receive a frenotomy prior to The overall purpose of this paper was to complete a
6 months of age, an ideal age range for the procedure systematic review to explore the outcomes on breast-
has yet to be established. As a result, the age inclu- feeding and speech production following ankyloglos-
sion/exclusion criteria differed across the various sia release. One of the primary challenges of
studies, which made comparisons challenging. Each comparing the results, regardless if related to breast-
study implemented different inclusion criteria in feeding or speech delays, is that a clear definition of
regards to participant age acceptability with an overall ankyloglossia is lacking. Recent anatomic studies that
age range from birth to 10 months. The wide age dissected the frenulum from neonates through adults,
range of infants involved across studies is a describe the frenulum as a structure much more
8 A. Visconti et al.
complex than a single cord of connective tissue on the
undersurface of the tongue (Mills, Pransky, Geddes,
& Mirjalili, 2019a; Mills, Keough, Geddes, Pransky,
& Mirjalili, 2019b). The lingual frenulum is described
instead as a “dynamic” structure that is formed by
the merging of the fascia of the floor of the mouth
with the oral mucosa that forms the top layer of the
underside of the tongue with an interweaving of some
muscle fibres. This complex anatomic architecture
allows the tongue to be simultaneously secured while
allowing for a wide range of movement (Mills et al.,
2019a). Further, as a result, the position of the frenu-
lum from the jaw to the ventral surface of the tongue
alters the appearance of the layers of tissue and overall
mobility of the anterior portion of the tongue. Thus
examining this structure in the context of a bimodal –
appears normal versus abnormal, should no longer be
applied during a clinical examination as the anatomic
components of the frenulum are much more complex
than can be identified in this simplistic form. Further,
the authors propose for continuity and accuracy that
the frenulum no longer be described as a single struc-
ture, but the term “midline fold” be utilised which
better reflects the anatomic complexity (Mills
et al., 2019a).
These detailed anatomic studies also call into
question recent clinical discussions regarding
“posterior” ankyloglossia. In both the feeding and
speech arenas, clinicians have questioned how best
manage this type of ankyloglossia. In our review, only
three studies distinguished between anterior and pos-
terior ankyloglossia (Ghaheri et al., 2017; O’Callahan
et al., 2013; Walls et al., 2014). However, these stud-
ies were not robust enough to make any conclusion
regarding breastfeeding or speech improvements fol-
lowing a posterior ankyloglossia release. Further,
based on the new anatomic perspective that we now
have, the general concept of a “posterior tongue tie”
is no longer supported (Mills et al., 2019a, 2019b).
The anatomic findings revealed that individuals with
frenulums that are positioned with a low attachment
point on the floor of the mouth might present with an
apparent fold of tight tissue that can be palpated,
when the individual elevates their tongue–seemingly
restricting mobility. At this time, it is unclear how an
ankyloglossia release will influence tongue mobility
and function in individuals with low attachment
points. Further, the frenulum does not have a direct
connection with the base of the tongue; therefore, the
term “posterior ankyloglossia” is not consistent with
the recent anatomic discovery, and clinician’s should
no longer describe the frenulum in this manner (Mills
et al., 2019b).
Another challenge identified was the lack of utilisa-
tion of objective, universal standard(s) to diagnose
severity of frenulum restriction and subsequent
impact on tongue mobility. This need is true for both
newborn infants and breastfeeding outcomes as well
as for older children who may present with speech
delays. The new anatomic data has also put into ques-
tion the basis for using Coryllos et al. (2004) as a
diagnostic tool and using Kotlow (1999) as a severity
rating. As stand-alone tools, both are now inadequate
to either define or diagnose ankyloglossia (Mills et al.,
2019a). The severity rating by Kotlow also recently
underwent validation in a prospective study of more
than 1000 individuals (Yoon et al., 2017). The results
found that the Kotlow scale had a sensitivity of 16%
and specificity of 78% and is associated with tongue
length (not mobility) and is not influenced by age,
gender, height, weight, or ethnicity (Yoon
et al., 2017).
Clinical bottom line
Which infant or child would benefit from surgical
intervention? This is a difficult question in light of the
variability reported in the research and should be con-
sidered as separate questions if discussing a newborn
with breastfeeding issues versus an older child with
articulation delay. For newborn infants, a consensus
document was just released that details the specific
clinical symptomatology for which infants should be
referred for medical intervention. This consensus
document was created for the specific use to recruit
participants into research studies for infants between
birth and 6 months of age. Two distinct sets of criteria
have been established; please refer to Katz et al.
(2020) for details.
In contrast, a wide range of inclusion criteria have
been utilised to recruit participants with speech
delays and lack of controlling for confounding varia-
bles is evident. At this time, our recommendations for
practicing clinicians are to collaborate with research
institutions to develop robust clinical trials in order to
validate their clinical recommendations if in favour
for the frenotomy procedure. Future research should
be more detailed and systematic in its design. For
example, tongue mobility does not necessarily correl-
ate to speech errors; therefore, other objective meas-
ures should be included. Utilisation of standardised
protocols, such as use of norm referenced articulation
assessments, are encouraged to ensure consistent and
reliable results. Certified speech-language patholo-
gists should conduct standardised and perceptual
measures to ensure inter-rater reliability and greater
consistency across studies. Further, future research
designs could then determine which populations,
ages, comorbidities and severity of ankyloglossia that
would most likely benefit from a frenotomy – if any.
Limitations
This review found observations of both within and
across study limitations. A predominant and recur-
ring limitation across all seven articles was the lack of
objective measures utilised within the studies.
Similarly, great variability was noted in the selected
measurement tools for each study (Berry et al., 2012;

Buryk et al., 2011; Emond et al., 2014; Ghaheri
et al., 2017; Messner & Lalakea, 2002; O’Callahan
et al., 2013; Walls et al., 2014). Another common
limitation was the absence of controls and randomisa-
tion in four of the seven studies, which may have con-
tributed to greater bias at the study level (Ghaheri
et al., 2017; Messner & Lalakea, 2002; O’Callahan
et al., 2013; Walls et al., 2014). Additionally, ethical
concerns (i.e. withholding frenotomy from infants
who may benefit from the procedure) contributed to
the inability to track long-term outcomes (Berry et al.,
2012; Buryk et al., 2011; Emond et al., 2014).
Acknowledgements
The researchers would like to acknowledge Sarah Haslett, Dr.
Susan Brehm, and Dr. Renee Gottliebson, of the Department
of Speech Pathology and Audiology at Miami University as well
as Ann Clonan, Leandre Gerwin, Melanie Jongewaard of
Cincinnati Children’s Hospital Medical Center for assistance in
this project.
Declaration of interest
The authors have no conflicts of interest.
ORCID
Donna R. Scarborough http://orcid.org/0000-0002-
0078-041X
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