International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646

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International Journal of Pediatric Otorhinolaryngology

journal homepage: www.elsevier.com/locate/ijporl

Review article

The effect of tongue-tie division on breastfeeding and speech articulation:

A systematic review
Amanda N. Webb a, Weibo Hao b, Paul Hong c,d,*
a
Faculty of Medicine, Dalhousie University, Halifax, NS, Canada
b
Faculty of Medicine, University of Western Ontario, London, ON, Canada
c
IWK Health Centre, Division of Otolaryngology-Head and Neck Surgery, Dalhousie University, Halifax, NS, Canada
d
School of Human Communication Disorders, Dalhousie University, Halifax, NS, Canada

A R T I C L E I N F O A B S T R A C T

Article history: Objective: To systematically review the outcomes of tongue-tie division procedures in patients with
Received 10 December 2012 ankyloglossia with the goal of (1) deriving clinically oriented insights into the effect of tongue-tie
Received in revised form 28 February 2013 division procedures and (2) identifying needs in knowledge to stimulate further research.
Accepted 7 March 2013
Data sources: Medline, EMBASE, and Cochrane databases were searched without any limitations, for
Available online 26 March 2013
studies published between 1966 and June 2012.
Review methods: Studies were included (level 4 evidence or above) if subjects of any age had
Keywords:
ankyloglossia and underwent tongue-tie release. Outcome measures of interest were any subjective or
Ankyloglossia
Tongue-tie
objective measures of breastfeeding and speech outcomes, or reports of adverse events.
Systematic review Results: In all, 378 abstracts were generated from the literature searches; 20 studies met the criteria for
Frenotomy data extraction and analysis. Of those, 15 studies were observational and 5 were randomized controlled
Frenulectomy trials. Tongue-tie division provided objective improvements in the following: LATCH scores (3 studies);
Frenuloplasty SF-MPQ index (2 studies); IBFAT (1 study); milk production and feeding characteristics (3 studies); and
Breastfeeding infant weight gain (1 study). Subjective improvements were also noted in maternal perception of
Speech articulation breastfeeding (14 studies) and maternal pain scores (4 studies). No definitive improvements in speech
function were reported. The only significant adverse events were recurrent tongue-ties that required
repeat procedures.
Conclusion: Ankyloglossia is a well-tolerated procedure that provides objective and subjective benefits
in breastfeeding; however, there was a limited number of studies available with quality evidence. There
are no significant data to suggest a causative association between ankyloglossia and speech articulation
problems. Aspects of ankyloglossia that would benefit from further research are described, and
recommendations for tongue-tie release candidacy criteria are provided.
ß 2013 Elsevier Ireland Ltd. All rights reserved.

Contents

1. Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 636
2. Methods . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 636
2.1. Protocol and registration . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 636
2.2. Eligibility criteria . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 636
2.3. Information sources . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 636
2.4. Study selection . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 636
2.5. Data extraction and items . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 636
2.6. Risk of bias in individual studies . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 637
2.7. Summary measures . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 637
2.8. Synthesis of results . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 637

Abbreviations: HATLFF, Hazelbaker assessment tool for lingual frenulum function; IBFAT, infant breastfeeding assessment tool; LATCH, latch, audible swallowing, type of
nipple, comfort, and hold; RCT, randomized controlled trial; SF-MPQ, short-form McGill pain questionnaire.
* Corresponding author at: IWK Health Centre, 5850/5980 University Avenue, PO Box 9700, Halifax, NS B3K 6R8, Canada. Tel.: +1 902 470 0841; fax: +1 902 470 8929.
E-mail address: Paul.Hong@iwk.nshealth.ca (P. Hong).

0165-5876/$ – see front matter ß 2013 Elsevier Ireland Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.ijporl.2013.03.008
636 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646

3. Results . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 637
3.1. Study selection . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 637
3.2. Study characteristics (Table 1) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 637
3.3. Objective outcomes (Tables 2 and 3) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 640
3.3.1. Breastfeeding outcomes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 640
3.4. Subjective outcomes (Tables 4–6) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 641
3.5. Speech outcomes (Table 8). . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 643
3.6. Adverse events . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 643
4. Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 644
4.1. Objective outcomes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 644
4.2. Subjective outcomes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 644
4.3. Speech outcomes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 645
4.4. Adverse events . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 645
4.5. Limitations . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 645
4.6. Conclusion and recommendations . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 645
Acknowledgements . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 646
References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 646

1. Introduction score sheets were also devised. The protocol was neither registered
nor published.
Ankyloglossia, or tongue-tie, is a congenital condition charac-
terized by a short, thickened, or abnormally tight lingual frenulum 2.2. Eligibility criteria
[1,2]. This anomaly can cause a varying degree of reduced tongue
mobility and has been associated with functional limitations The research question for this study was formulated as follows:
including: breastfeeding difficulties, atypical swallowing habits, Population: any individuals with ankyloglossia. Intervention:
speech articulation problems, mechanical problems such as tongue-tie release. Comparison: pre- and post-tongue-tie release.
inability to clean the oral cavity, and psychosocial stress [3–5]. Outcomes: subjective and objective measures of breastfeeding or
Prevalence rates of ankyloglossia range from 2.08% to 10.7% speech and adverse events [16].
depending on the patient population and diagnostic criteria used Studies were included if subjects of any age had ankyloglossia
[3,6–8]. Male to female ratios have been reported in the order of and received tongue-tie release procedures to address breastfeed-
3:1 [3,4,8]. The mainstay of management for ankyloglossia is ing and/or speech problems. Tongue-tie release procedures
tongue-tie division, or frenotomy [2,6,9,10]. Historically, freno- included frenotomy or frenulotomy (division of lingual frenulum),
tomies were routinely performed, as ankyloglossia was thought to frenuloplasty (frenotomy with placement of sutures), and fre-
significantly interfere with breastfeeding [11]. It was common nectomy or frenulectomy (excision of lingual frenulum).
practice, in the 18th century, for midwives to divide short Studies were required to meet criteria for level 4 evidence or
frenulums with their fingernails [12]. Yet, with the introduction above, as defined by the Oxford Centre for Evidence Based
of formula, fewer infants were being breastfed, and consequently, Medicine [17], which includes case series, case–control studies,
ankyloglossia posed less of a concern for new mothers [12]. More cohort studies, and randomized control trials. Articles of all
recently, with increasing awareness that breastfeeding provides languages were included. Duplicate studies and case series with
health benefits, more mothers are trying again to breastfeed [13]. less than 5 patients were excluded.
Despite this, there is currently little consensus on the management
practices for ankyloglossia. This was demonstrated in a survey 2.3. Information sources
study of North American paediatricians, otolaryngologists, lacta-
tion consultants, and speech language pathologists [14]. A comprehensive search strategy was used to search the
The purpose of this systematic review was to consider original following databases: MEDLINE, EMBASE, and Cochrane Database of
studies reporting on patients with ankyloglossia who underwent Systematic Reviews (Fig. 1). Titles and abstracts were obtained for
tongue-tie release procedures. This included any studies evaluat- all studies identified by the search strategy. The bibliographies of
ing outcomes related to breastfeeding and/or speech articulation. those studies obtained in full text were hand-searched for any
As well, other outcomes and adverse events, were included with additional relevant studies not identified by the original database
the purpose of deriving clinically oriented insights into the searches.
advantages and disadvantages of tongue-tie division procedures.
This study intends to aid clinicians who are considering tongue-tie 2.4. Study selection
release for patients with ankyloglossia by providing an appraisal of
the literature to date, as well as to identify gaps in knowledge for Two authors (A.W. and W.H.) independently reviewed the list of
the purpose of stimulating future research. titles and abstracts generated by the literature search for any
studies that met the abovementioned criteria. The senior author
2. Methods (P.H.) was involved when there were conflicts. The full-text studies
were further evaluated in regards to the inclusion criteria by 2
2.1. Protocol and registration authors independently, again involving the senior author when
there were conflicts.
The statements of Preferred Reporting Items for Systematic
reviews and Meta-Analyses was followed to report the current 2.5. Data extraction and items
review [15]. A detailed research protocol was designed a priori
with the purpose of defining the study scope, objective, hypothesis, The following data were extracted: study design, level of
and methodology. A data collection form and quality assessment evidence, number of subjects, description of subjects, description
[(Fig._1)TD$IG] A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646
Fig. 1. Details of the literature search strategy.
of intervention, and outcome measures. For quantitative outcome
measures, mean/median values, standard deviations, confidence
intervals, and/or P values were recorded, when possible; data were
synthesized into tables when methodologies were similar enough
to allow it.
The authors assessed the quality of the studies using a seven-point
Quality Assessment Score system (Table 1). One point was awarded
for each of the following characteristics: (1) inclusion/exclusion
criteria clearly stated; (2) assessment method validated; (3)
appropriate comparison group (intra-subject or control group); (4)
observers blinded; (5) adequate sample size/power analysis; (6) well-
defined, consecutive sample; and (7) less than 5% lost to follow-up.
2.6. Risk of bias in individual studies
Risk of selection and publication bias was evaluated at the study
level with consideration of sample collection (consecutive or not),
inclusion and exclusion criteria (clearly stated or not), and
outcome measures (reliable or not). Potential confounding risk
factors, such as continued lactation consultation and speech
therapy, were searched in all studies. Regarding tongue-tie
divisions, all studies were also assessed at the outcome level for
randomization, blinding, and withdrawals.
2.7. Summary measures
Measures were summarized as objective breastfeeding out-
comes, subjective breastfeeding outcomes, speech outcomes, and
reports of adverse events.
2.8. Synthesis of results
Data were synthesized into tables, according to the nature of
the outcome measures. No meta-analysis was performed due to
the heterogeneity of the data and lack of access to raw data.
637
3. Results
3.1. Study selection
A total of 378 abstracts were generated from the database and
bibliography searches. Fig. 2 shows the flow diagram of the search
and exclusion process [15]. A total of 35 studies met the criteria for
full-text review; the 343 rejected abstracts were eliminated either
because they did not discuss ankyloglossia or tongue-tie division,
or for those that did, they did not include any subjects (the vast
majority). After further review, 20 studies met the criteria for data
extraction and analysis [8,18–36]; 15 were excluded for various
reasons, which included case series with less than 5 subjects, no
treatments, abstract only (scientific meeting abstracts), no
reportable outcomes, and articles describing new surgical tech-
niques without outcomes.
3.2. Study characteristics (Table 1)
Of the 20 studies that were included for data extraction and
analysis, 5 were randomized controlled trials [8,20,23,31,32] and 15
were observational [18,19,21,22,24–30,33–35] (14 cases series and
1 case–control trial). Eleven studies provided subjective outcomes, 3
provided objective outcomes and 6 provided both. There were a total
of 1012 subjects in the 20 studies with ages that ranged from 1 day to
23 years. Of the 1012 subjects, 729 underwent frenotomy (57 control
subjects went on to receive frenotomy at a later time) and 167
underwent various types of frenuloplasty procedures. The most
commonly reported indications for tongue-tie division was breast-
feeding problems (non-specific), poor latch, sore or damaged
nipples, inefficient feeding, continuous feeds/infant frustration/
unsatisfied post-feeding, poor weight gain, and speech problems.
Study characteristics are summarized in Table 1.
Quality assessment scores ranged from 2 to 6 out of 7
(Table 1). Twelve studies had no subjects lost to follow-up
638 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646

Table 1
Characteristics of studies included in the systematic review.

Study Country Study design Quality N Patient characteristics Outcomes

score

Amir et al. [30] Australia Case series 3 46 Age: mean of 18 days old at time of assessment, HATLFF
range 3–98 days, median 12.5 days Telephone interview
Sex: 29 boys, 17 girls
Indications: specified breastfeeding problems
and HATLFF
Intervention: after HATLFF, 35/46 infants
underwent outpatient frenotomy
Argiris et al. [18] United Kingdom Case series 4 46 Age: mean of 4 weeks, range 1 day to 12 weeks Devised questionnaire
Sex: 33 boys, 13 girls Pain score
Indications: clinical diagnosis of ankyloglossia;
breastfeeding problems (67% poor latch, 63%
sore nipples, damaged nipples 43%, coming on/
off breast (50%), unsatisfied baby after feeds
(30%), poor weight gain (22%)
Intervention: frenotomy and short observation
on paediatric ward
Ballard et al. [19] United States Case series 4 123 Age: median (25th, 75th percentile) age for HATLFF
those with poor latch 1.2 days (0.7, 2.9); those Observation and maternal
with nipple pain 2.0 days (1.0, 12.0) description of breastfeeding
Sex: boys to girl ratio 1.5:1 Pain score
Indications: breastfeeding problems (70 poor
latch, 53 nipple pain) and HATLFF
Intervention: frenuloplasty (lingual frenulum
division only), anaesthesia for >4 months
Berryet al. [31] United Kingdom RCT 6 60 Age: mean 32 days, median 23 days, range Blinded observation by
5–115 days trained observer and mothers
Sex: boys to girl ratio 2:1 LATCH scoring system
Indications: clinical diagnosis of ankyloglossia; (revised)
breastfeeding problems (78% poor latch, 65% Infant breastfeeding
nipple pain/trauma, 62% inefficient feeding, 32% assessment tool
all three problems) Pain score
Intervention: 27 randomized to receive
outpatient frenotomy; 30 control (3 excluded);
all patients received frenotomy at the end of
study
Buryk et al. [32] United States RCT 6 58 Age: mean 6 days (SD, 6.9; range 1–35) HATLFF
Sex: 38 boys, 20 girls Nipple-pain scale (SF-MPQ)
Indications: breastfeeding problems and HATLFF Infant breastfeeding
Intervention: 30 randomized to receive assessment tool
outpatient frenotomy and 28 sham operation
(option given for frenotomy 2 weeks later; 27
underwent frenotomy)
Dollberg et al. [20] Israel RCT 5 25 Age: range 1–21 days LATCH score
Sex: not specified Pain score
Indications: clinical diagnosis of ankyloglossia;
breastfeeding associated maternal nipple pain
Intervention: group 1 underwent frenotomy,
assessment, sham, assessment; group 2
underwent sham, assessment, frenotomy,
assessment
Dollberg et al. [21] Israel Case–control 3 23 Age: mean years in study group: n = 8, 6.2 (SD, Standardized articulation
1.8); comparison group: n = 7, 6.2 (SD, 1.9); test (assessed by speech
control group: n = 8, 5.8 (SD, 1.9) language pathologists)
Sex: not specified
Indications: clinical diagnosis of ankyloglossia;
breastfeeding problems in the past during
infancy in the study and comparison groups
Intervention: frenotomy
Geddes et al. [35] Australia Case series 3 24 Age: 33  28 days (range 4–131) Submental ultrasound scans
Sex: not specified LATCH score
Indications: clinical diagnosis of ankyloglossia; Pain score
breastfeeding problems Milk transfer
Intervention: frenotomy
Griffiths [22] United Kingdom Case series 4 215 Age: mean 19 days Telephone interview
Sex: boys to girl ratio 2:1
Indications: clinical diagnosis of ankyloglossia;
breastfeeding problems despite professional
support (88% poor latch, 77% painful, sore, or
bleeding nipples, 72% inefficient feeding)
Intervention: outpatient frenotomy
 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646 639

Table 1 (Continued )

Study Country Study design Quality N Patient characteristics Outcomes

score

Heller et al. [23] United States RCT 3 16 Age: mean years in 4-flap frenuloplasty (study) Frenulum measurements
group: n = 11, 5.7  2.14 (range 3.1–9.9); Speech pathology
horizontal-to-vertical (comparison) group: n = 5, assessments
5.56  1.52 (range 3.4–7.1)
Sex: 9 boys, 7 girls
Indications: tight frenulum and speech
articulation problems
Intervention: 4-flap frenuloplasty vs. horizontal-
to-vertical frenuloplasty
Hogan et al. [8] United Kingdom RCT 4 57 Age: mean days in study group: n = 28, 20 Telephone interviews
(median 14); control group: n = 29, 18 (median
15)
Sex: study group boys to girls ratio 1:1; control
group 1.3:1
Indications: clinical diagnosis of ankyloglossia
and breastfeeding problems (82% poor latch,
52% inefficient feeding, 80% pain, 15% mastitis)
or bottle-feeding problems (76% slow feeders,
71% major dribbling, 24% excess wind)
Intervention: study group-outpatient
frenotomy; control group-intensive support
from lactation consultant (48 h later offered
frenotomy)
Khoo et al. [24] United Kingdom Case series 2 62 Age: <90 days Devised questionnaire
Sex: 42 boys, 20 girls
Indications: clinical diagnosis of ankyloglossia
and breastfeeding problems (84% nipple pain,
52% nipple trauma, 84% poor latch, 81% infant
frustration, 44% unsatisfied after feeds, 63%
noisy feeding, 11% prolonged jaundice)
Intervention: outpatient frenotomy
Lalakea and United States Case series 3 6 Age: mean years 17.3  3.2, range 14–23 Devised questionnaire
Messner [25] Sex: not specified Tongue mobility
Indications: clinical diagnosis of ankyloglossia and
subjective speech and/or mechanical problems
Intervention: horizontal-to-vertical frenuloplasty
under local anaesthesia (post-treatment tongue
mobility exercises)
Marmet et al. [29] United States Case series 2 7 Age: mean days 29.4, range 1–70 Subjective maternal
Sex: no specified reports (unclear)
Indications: clinical diagnosis of ankyloglossia
and breastfeeding problems (2 mastitis, 5 poor
latch, 7 poor suck)
Intervention: outpatient frenotomy
Masaitis and United States Case series 3 36 Age: mean days 5.7, median 3, range 1–24 Telephone interviews
Kaempf [26] Sex: 20 boys, 16 girls
Indications: clinical diagnosis of ankyloglossia
(27 poor latch, 27 nipple trauma, 29 tongue
doesn’t cross alveolus, 29 heart-shaped tongue,
24 frenulum attached to tongue tip, 9
breastfeeding failure, 7 poor weight gain, 4
clicking sound, 1 breast abscess)
Intervention: outpatient frenotomy
Messner and United States Case series 3 28 Age: mean years 4.1, range 1–12 (2 lost to Devised questionnaires
Lalakea [36] follow-up) Tongue mobility
Sex: 19 boys, 11 girls Speech evaluation
Indications: clinical diagnosis of ankyloglossia
and anticipated or actual problems with feeding,
speech, or social mechanical issues
Intervention: horizontal-to-vertical frenuloplasty
and post-treatment tongue exercises (other
simultaneous procedures, such as typanostomy
tube placement, was done in some)
Miranda and United Kingdom Case series 3 51 Age: range 12–36 days Devised questionnaire
Milroy [33] Sex: not specified
Indications: clinical diagnosis of ankyloglossia
and breastfeeding problems
Intervention: outpatient frenulotomy
Srinivasan Canada Case series 4 27 Age: mean days 19, median 10, range 2–71 LATCH score
et al. [27] Sex: 18 boys, 9 girls Nipple pain scale (SF-MPQ)
Indications: ‘Frenotomy decision rule for Telephone interview
breastfeeding infants’ and breastfeeding
problems
Intervention: outpatient frenotomy and
lactation counselling
640 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646

Table 1 (Continued )

Study Country Study design Quality N Patient characteristics Outcomes

score

Steehler et al. [28] United States Case series 2 91 Age: respondents age not specified Telephone interview
Sex: respondents sex not specified
Indications: clinical diagnosis of ankyloglossia
and breastfeeding problems
Intervention: outpatient frenotomy (n = 82)
Wallace and United Kingdom Case series 2 11 Age: median 10 days, range 2–31 Telephone interview
Clarke [34] Sex: 9 boys, 2 girls
Indications: clinical diagnosis of ankyloglossia
and breastfeeding problems
Intervention: outpatient frenotomy

[8,18–28]. All studies stated their inclusion criteria and all but 2 SF-MPQ and subsets: Buryk and colleagues reported a significant
studies [21,29] used a well-defined, consecutive sample. improvement in maternal nipple pain following tongue-tie
Comparisons were intra-subject or with a control group in 14 division, compared to controls (sham procedure) (P < 0.001)
studies [8,18–21,23,25,27,28,30–34]. Four studies had blinding [32]. Similarly, in a case series of 27 subjects, tongue-tie division
of observers to either condition, intervention, or both significantly improved maternal nipple pain as measured by both
[20,21,31,32]. Four studies used at least one validated method the SF-MPQ Pain Rating Index (P < 0.0001) and the Present Pain
of assessment [21,27,32,35] and 4 studies included a sample size Intensity scale (P < 0.0001) [27].
calculation [22,24,31,32]. IBFAT: Only 1 study compared pre- and post-division IBFAT
scores [32]. The mean score improved from 9.3  0.69 to
3.3. Objective outcomes (Tables 2 and 3) 11.6  0.81, following division compared to subjects receiving a
sham procedure who did not show any improvements (P < 0.029)
3.3.1. Breastfeeding outcomes [32].
LATCH scores: All three studies [20,27,35] that reported LATCH Milk production and feeding characteristics: Three studies
scores demonstrated improvements post-treatment, although measured milk production and feeding characteristics (Table 3)
only the studies by Geddes et al. [35] and Srinivasan et al. [27] [24,33,35]. There was a significant increase in milk production
showed significant improvements (P < 0.05 and P < 0.0001, (P = 0.035) [35], milk intake (P < 0.01) [35], milk transfer (P < 0.01)
respectively) (Table 2). [35], and time between feeds (P < 0.001) [24]. Correspondingly,
[(Fig._2)TD$IG]
Identification

Records identified through database Additional records identified by

searching hand-searching bibliographies
(n = 378) (n = 0)
Screening

Abstracts screened Records excluded

(n = 378) (n = 343)

Full-text articles assessed Full-text articles excluded

Eligibility

for eligibility (n = 15)

(n = 35) -Case series with n < 5 (4)
-No treatment (3)
-Abstract only (2)
-No outcomes (3)
-Surgical techniques (3)

Studies included in
Included

qualitative synthesis
(n = 20)

Fig. 2. Flow diagram of article selection process.

 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646 641

Table 2 at 2 weeks post-division, neonates were at 56  2 centiles, which was

LATCH score outcomes after tongue-tie release.
a significant increase from 41  3 centiles (P < 0.0001) [33].
Study N Maternal reporting of LATCH scores

Geddes et al. [35] 24 Pre-treatment: 7.9  1.4 3.4. Subjective outcomes (Tables 4–6)
Post-treatment: 9.4  0.8
P < 0.05 Fourteen studies reported subjective outcomes. Of these, 5
Dollberg et al. [20] 25 Pre-treatment: 6.4  2.3 studies reported immediate outcomes, 8 reported short-term
Post-treatment: 6.8  2.0
outcomes (24 h to 6 weeks), and 5 reported long-term outcomes
P = 0.06
Srinivasan et al. [27] 27 Pre-treatment: 6.7  1.2 (12 weeks to 5 years) (Tables 4 and 5).
Post-treatment: 9.2  0.9 Immediate outcomes: The following number (%) of mothers
P < 0.0001 reported immediate improvements in breastfeeding after tongue-
tie division in 3 studies: 32 (70%) [18], 123 (57%) [22], and 4 (40%)
[34]. A study by Berry et al. reported 21 (78%) mothers in the
there was a decrease in length of feeds per minute (P < 0.001) [24], treatment group with improved breastfeeding, whereas only 14
number of feeds per day (P < 0.005) [24], and breastfeeding (47%) mothers in the control group reported improved breastfeed-
sessions per 24 h (P < 0.0001) [33]. Geddes et al. also found that ing (P < 0.02) [31]. Hogan and colleagues reported 24 (85%)
nipple compression was reduced following tongue-tie division mothers in the division group with improved breastfeeding,
based on submental ultrasonography [35]. whereas only 1 mother in the non-division group reported
Weight gain: Miranda and colleagues’ prospective study of 51 improved breastfeeding (P < 0.001) [8].
neonates with breastfeeding difficulties found that tongue-tie Short-term outcomes: Argiris et al. reported 40 (87%) mothers
division was associated with improved weight gain [33]. Specifically, with improved breastfeeding at 6 weeks [18]; Griffith et al.

Table 3
Objective breastfeeding outcome measures.

Study N Objective breastfeeding measures

Buryk et al. [32] 46 SF-MPQ for maternal nipple pain

Study group: 16.77 (SD, 1.88) ! 4.9 (SD, 1.46)
Sham group: 19.25 (SD, 1.9) ! 13.5 (SD, 1.5)
P < 0.001; effect size 0.38
IBFAT for breastfeeding
Study group: 9.3 (SD, 0.69) ! 11.6 (SD, 0.81)
Sham group: 8.48 (SD, 0.73) ! 8.07 (SD, 0.86)
P = 0.029; effect size 0.31
Geddes et al. [35] 24 Total milk production (n = 6)
Pre-treatment: 455  323 g
Post-treatment: 615  289 g
P = 0.035
Milk intake
Pre-treatment: 50.5  29.1 g
Post-treatment: 69.1  31.9 g
P < 0.01
Milk transfer (mL/min)
Pre-treatment: 5.6  3.0 g
Post-treatment: 10.5  5.5 g
P < 0.01
Submental ultrasound
Pre-treatment: compression of nipple (n = 21)
Post-treatment: reduced compression of nipple
Khoo et al. [24] 62 Number of feeds per day
Pre-treatment: 7.4  2.4
Post-treatment: 6.4  1.5
P < 0.005
Length of feed per minute
Pre-treatment: 41.6  27.5
Post-treatment: 24.1  17.4
P < 0.001
Time between feeds per minute
Pre-treatment: 161.1  44.3
Post-treatment: 197.0  45.3
P < 0.001
Miranda and Milroy [33] 51 Weight gain
90% gained in centile
6% remained at same centile
4% dropped 7.5 centiles
Overall gain of 15  1.2 centiles
Weight centile: 41  3 ! 56  2 (P < 0.0001)
Breastfeeding sessions/24 h: 10  0.7 ! 7  0.5 (P < 0.0001)
Bottle-feeding sessions/24 hours: 9  0.6 ! 2  0.7 (P < 0.0001)
Srinivasan et al. [27] 27 SF-MPQ for maternal nipple pain
Pain rating index: 13.7 (SD, 19.0) ! 2.2 (SD, 3.1)
P < 0.0001, 95% CI, 15.544, 7.345
Present pain intensity: 2.2 (SD, 1.3) ! 0.7 (SD, 0.9)
P < 0.0001, 95% CI, 1.952, 1.011
642 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646

Table 4
Subjective maternal breastfeeding outcomes.

Study N Maternal subjective outcomes in breastfeeding

Amir et al. [30] 35 6 (17%) no difference

29 (83%) improved breastfeeding (mean 26 weeks, range 12–46)
18 (51%) better attachment to breast
20 (57%) improved sucking
9 (26%) less pain
6 (17%) improved weight
2 (6%) ‘‘other difference’’
3 (9%) not breastfeeding
Argiris et al. [18] 46 32 (70%) immediate improvement in breastfeeding
40 (87%) improvement in breastfeeding at 6 weeks
36 (78%) improved latch
19 (40%) reduced nipple pain
30 (64%) improved suck
Berry et al. [31] 57 21 of 27 (78%) in the study group had immediate improvement in breastfeeding
14 of 30 (47%) in the control group had immediate improvement in breastfeeding
P < 0.02
Griffiths et al. [22] 215 Immediate assessment
123 (57%) improvement in breastfeeding; 92 (43%) no difference
At 24 h post-treatment
173 (80%) improved breastfeeding at 24 h post-treatment; 40 (19%) unchanged;
2 increased difficulties
Hogan et al. [8] 57 Study group (frenotomy, n = 28)
27 (96%) improved (85% immediately and 15% within 48 h)
1 remained on a nipple shield with continuous feeds
Control group (supportive, n = 29)
1 (3%) improved
Frenotomy offered at 48 h (27/28 improved, 77% immediately and 19% within 48 h)
Study vs. control groups: P < 0.001
Khoo et al. [24] 62 Likert scale from 0 to 10 (0-no difficulty; 10-maximum difficulty)
Pre-treatment: 6.1  2.7
Post-treatment: 1.9  2.6
P < 0.001
Lalakea et al. [25] 6 100% reported subjective gains in at least 3 or 6 categories of mechanical tongue function
Marmet et al. [29] 7 71% reported completely improved breastfeeding (improved latch, suck, nipple soreness,
breast soreness, slow weight gain, milk supply)
29% non-responders had ‘‘severe birth defects’’
Masaitis et al. [26] 36 1 week post-treatment
89% breastfeeding; 11% bottle feeding
75% problem resolved completely; 19% partially; 6% no
94% appropriate infant growth rate; 6% slow growth
100% would choose frenotomy again
Miranda and Milroy [33] 51 Within 2 weeks of treatment
63% improved breastfeeding
89% improved latch
100% improved nipple pain
100% improved nipple cracking and bleeding
Steehler et al. [28] 82 Retrospective report (unspecified time between treatment and interview)
80.4% thought frenotomy was beneficial (86% frenotomy done during first week;
74.3% frenotomy done later; P < 0.003)
82.9% continued to breastfeed long-term (mean 7.09 months)
Wallace et al. [34] 10 40% immediate improvement
30% improvement within 2 weeks
30% no improvement
60% breastfed for at least 4 months

reported 173 (80%) mothers with improved breastfeeding at 24 h
post tongue-tie release [22]. In Hogan and colleagues’ study, 27
(96%) mothers in the treatment group reported improved
breastfeeding within 48 h, whereas only 1 mother in the non-
division group reported improved breastfeeding (P < 0.001) [8].
Lalakea and Messner reported all of their 6 patients having
subjective gains in at least 3 of 6 categories of mechanical tongue
function (e.g., lick ice cream, speech articulation) at 1 month [25].
A similarly positive short-term subjective breastfeeding
improvements were noted in several other studies [26,29,33,34].
Long-term outcomes: Amir et al. reported 29 (83%) mothers who
had improved breastfeeding at follow up of 12–46 weeks [30].
In another study, 54 (92%) mothers reported improved breastfeed-
ing at 3 months, and 33 (56%) reported full resolution [31]. Khoo
et al. reported significantly reduced (P < 0.001) mean feeding
difficulties at 3 months post-treatment [24]. Steehler et al.
reported improvements in 37 (86.0%) mothers whose child
underwent frenotomy during the first week of life at long-term
follow up (3–5 years), while only 29 (74.3%) mothers whose child
underwent frenotomy after the first week of life reported improved
breastfeeding (P < 0.003) [28]. Six (60%) mothers continued to
breastfeed for at least 4 months in another study [34].
Pain scores: Four studies used a 10-point visual analogue scale
(0 no pain; 10 severe pain) to measure maternal feeding pain
before and after tongue-tie release (Table 6). Argiris et al. reported
significantly reduced pains scores (P < 0.01) from 6.63  2.46 to
1.47  1.34 after tongue-tie release [18]. Studies conducted by
Ballard et al. [19] and Dollberg et al. [20] also reported the significant
reductions in pain scores (P < 0.001 and P = 0.001, respectively). In
the study by Berry et al, the mean score in the treatment group
 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646 643

Table 5 Table 7
Long-term breastfeeding outcomes. Subjective maternal comments regarding tongue-tie release.

Study N Time of assessment and breastfeeding Study N Open-ended comments regarding the tongue-
outcomes tie release experience

Amir et al. [30]
Berry et al. [31]
Buryk et al. [32]
Geddes et al. [35]
Griffiths et al. [22] 215
Hogan et al. [8]
Khoo et al. [24]
Masaitis et al. [26]
Srinivasan et al. [27]
35 26 weeks (mean) post-treatment
83% (29) improved feeding
59 3 months post-treatment
92% (54) improved feeding
56% (33 of 59) full resolution
8% (5 of 59) no improvement
65% (38 of 59) still breastfed
4.5 months (mean) post-treatment
51% (30 of 59) still breastfed
58 Overall breastfeeding rates at 2, 6,
and 12 months of age were 66% (36),
44% (23), and 28% (14), respectively;
1, 6, and 14 total patients lost to
follow-up, respectively
16 Mean duration of any breastfeeding
after frenotomy 11.3  5.2 months
(range 5–24 months); 1 mother was
not breastfeeding at 5 months
At 3 months post-treatment
138 (64%) breastfed for at least
3 months
11 (5%) breastfed for 6–12 weeks
68 (32%) breastfed for <6 weeks
40 At 4 months post-treatment
24 (60%) breast-fed for at least
4 months
62 All assessments done at 3 months
(see Table)
36 3 months post-treatment
53% breastfeeding; 47% breastfeeding
100% problem resolved completely
100% appropriate infant growth rate
100% would choose frenotomy again
25 3 months post-treatment
77.8% still breastfeeding
92% no nipple pain
88% thought frenotomy was beneficial
Amir et al. [30] 35 Happy it was done, feeding settled down over
next couple of days, very pleased with
assessment/I understood all the explanation,
couldn’t believe how quickly and easy it was, I
was lucky it was released as early as it was/
would be bottle-fed early if not released
Ballard et al. [19] 123 ‘‘stronger’’, ‘‘smoother’’, ‘‘more natural’’,
‘‘more like a massage’’, ‘‘less chewing or
gumming’’, ‘‘more effective’’, ‘‘getting more
milk out’’
Berry et al. [31] 59 All would choose to have procedure again if
they were in the same situation in the future
Masaitis et al. [26] 36 The frenotomy was done at seven days and it
took two weeks for my nipples to heal
The very next feeding went better and my
nipples healed right away (n = 5)
I needed to pump and bottle-feed for
engorgement, but then the nursing went well
I’m allergic to dairy, so I’m glad breastfeeding
problems resolved so I didn’t have to use say
formula
Baby thinks he should lick everything! (n = 2)
He nursed like he is supposed to and I healed
quickly
I appreciate the telephone follow-up because I
felt so alone
Sometimes she took six tries to latch but she
was an excellent breastfeeder by three
months
positive comments and all participants were reported as being
satisfied [26,30,31].
3.5. Speech outcomes (Table 8)

Four studies reported speech outcomes for subjects with

decreased from 4.1 to 1.6, while the mean score in the control group
decreased from 4.2 to 2.9 [31]. Although more favourable for the
frenotomy group, no significant difference was observed (P = 0.13).
Maternal comments: Four studies included open-ended com-
ments from mothers whose infants underwent tongue-tie division
(Table 7) [19,26,30,31]. Phrases such as ‘‘stronger and smoother’’,
‘‘less chewing or gumming’’, and ‘‘getting more milk out’’ were
commonly stated to describe the quality of latching/breastfeeding
shortly after the procedure [19]. Other studies also demonstrated
Table 6
Maternal subjective breastfeeding pain scores.
Study N Maternal breastfeeding pain scoresa
Argiris et al. [18] 46 Pre-treatment: 6.63 out of 10 (SD 2.46)
Post-treatment: 1.47 out of 10 (SD 1.34)
P < 0.01
Ballard et al. [19] 123 Pre-treatment: 6.9 out of 10 (SD 2.31)
Post-treatment: 1.2 out of 10 (SD 1.52)
P < 0.0001
Berry et al. [31] 28 Study group (n = 14)
Pre-treatment: 4.1 out of 10
Post-treatment: 1.6 out of 10 (SD, 1.9)
Control group (n = 14)
Pre-treatment: 4.2 out of 10
Post-treatment: 2.9 out of 10 (SD, 1.5)
P = 0.13 (95% CI, 0.3 to 2.4)
Dollberg et al. [20] 25 Pre-treatment: 7.1 out of 10 (SD, 1.9)
Post-treatment: 5.3 out of 10 (SD, 2.2)
P = 0.001
a
Pain scored on a visual analogue scale (0-mild pain; 10-extreme pain).
ankyloglossia receiving tongue-tie division [21,23,25,36]. Dollberg
et al. evaluated speech in the following groups: (1) study group
(n = 8) that underwent tongue-tie division during infancy, (2)
comparison group (n = 7) that did not receive treatment during
infancy, and (3) control group (n = 8) comprised of subjects
without ankyloglossia [21]. A standardized articulation test was
administered by 2 speech language pathologists. No significant
differences between the groups with respect to articulation errors
or speech intelligibility were observed, but most articulation errors
occurred in the group 2 [21]. A case series of 6 patients by Lalakea
and Messner reported subjective speech improvements in 2
patients following frenuloplasty and post-treatment tongue
mobility exercises [25]. Another study by the same authors
reported improved speech following tongue-tie division in 9 of 11
subjects based on speech language pathologists’ evaluation pre-
and post-treatment [36] (Table 8).
3.6. Adverse events
Four studies reported specific adverse events. Argiris et al.
reported unquantified blood loss in 24 patients (52%), and 3
patients (6.5%) required a repeat frenotomy procedure [18]. In
another study, 3 mothers (5%) reported a small amount of bleeding
at home following the procedure [31]. Griffiths et al. reported 4
patients (2%) who developed an ‘‘ulcer’’ under the tongue for more
than 48 h; 1 patient (0.5%) was ‘‘sore’’ for more than 24 h, and 1
patient (0.5%) produced brown posset due to swallowed blood
[22]. Steehler et al. reported 8 patients (2.6%) requiring a revision
procedure for incomplete clipping or scarring [28]. Seven studies
644 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646
Table 8
Speech outcomes after tongue-tie division.
Study N Speech outcomes
Dollberg et al. [21] 23 Patients
Study group (underwent frenotomy
during infancy, n = 8)
Comparison group (non-treated
frenotomy during infancy, n = 7)
Control group (no history of
ankyloglossia, n = 8)
Outcome measures
Standardized articulation test
assessed by two speech pathologists
Results
Most articulation errors in the
comparison group, followed by the
study and then control groups, but no
significant difference between the
groups in articulation errors and
speech intelligibility
Heller et al. [23] 16 Patients
Study group (underwent 4-flap z-
freunloplasty)
Comparison group (underwent
horizontal-to-vertical frenuloplasty)
Outcome measures
Tongue measurements
Articulation testing by two speech
pathologists (>10 months post-
treatment)
Results
Greater gain in frenulum length and
tongue protrusion for the study group
Better articulation improvements in
the study group (no statistical
analysis)
Lalakea et al. [25] 6 2 reported subjective improvement in
speech
4 did not report any change
Messner and 15 4 had normal speech pre-treatment (no
Lalakea [36] change post-treatment)
11 had abnormal speech pre-treatment
9 (82%) judged to have improvement
2 had persistent articulation problems
Improved subjective speech
intelligibility reported by parents
(P < 0.01)
(357 patients combined) reported no adverse events
[8,25,27,30,32,33,35].
4. Discussion
There is no simple reliable tool to determine what constitutes
clinically important ankyloglossia. This makes it difficult to
identify a target population for ankyloglossia studies and provide
clear indications for frenotomy. To this end, the relationship
among breastfeeding issues, speech articulation problems, anky-
loglossia, and frenotomy need to be examined more closely. Few
studies have attempted to use more objective or standardized
measures during the initial assessment phase [19,27,30,32] but the
practicality and the usefulness of such measures in the clinical
setting is still questionable.
Pre-treatment assessments are required since not all newborns
with ankyloglossia will have difficulties breastfeeding. Yet, the
severity or degree of ankyloglossia is not easily defined. The
HATLFF may be used to objectively assess the degree of
ankyloglossia but it is still based on underlying subjective clinical
assessments [19,30,32,37,38]. Furthermore, many studies have not
used pre-treatment tools but rather a simple clinical examination
to determine the presence of ankyloglossia, and they showed
significant improvements following tongue-tie division. This may
indicate that a formal pre-treatment assessment, which may be
cumbersome and difficult to validate, may not be strictly
necessary.
4.1. Objective outcomes
The LATCH tool has been validated to identify mothers at risk
for early weaning because of sore nipples [39]. Although this
outcome measure is not specific to ankyloglossia, scores signifi-
cantly improved in two studies following tongue-tie division
[27,35]. This suggests that frenotomy can improve breastfeeding as
evaluated by the LATCH score.
There are many validated pain scores in clinical use, yet none
are specific to breastfeeding related nipple pain. The SF-MPQ is
validated for use in various types of pain [40] and was utilized in 2
studies with positive outcomes being noted after tongue-tie
division in both studies [27,32].
One study used 3 different objective measures but the failure of
such objective scoring was noted in this study [31] and also in
another blinded study of nipple pain [20]. Specifically, the first
study did not explicitly report on their LATCH or IBFAT assessment
scores and noted that the 3-component assessment tool was too
broad to show differences in pre- and post-frenotomy scores [31].
Furthermore, there is some controversy over whether any existing
breastfeeding evaluation tools are truly reliable or valid, including
the IBFAT [41]. Therefore subjective assessments, rather than
objective tools for measuring outcomes related to breastfeeding,
may be the more optimal in some cases [31].
Of the studies using objective measures, many lacked control
groups for inter-subject comparisons. This increases the possibility
that treatment biases could have led to post-treatment benefits. On
the other hand, blinding can be an issue to both parents and
observers in trials with control or comparison groups, since it is
difficult to hide whether a child actually underwent the tongue-tie
release procedure.
4.2. Subjective outcomes
Overall, tongue-tie division was shown to be immediately
effective in improving the maternal breastfeeding experience in
many cases.
Open-ended maternal comments were very positive regarding
the procedure. A formal qualitative (content) analysis across
studies in regards to maternal comments was not performed due to
the lack of raw transcript data (Table 7). However, general themes
of improved breastfeeding and procedural satisfaction emerged.
Tongue-tie division also appeared to provide some long-term
benefits as more than 50% of the mothers were still breastfeeding
at 3 months after division. However, it is unclear whether this was
the result of tongue-tie division or other adjuvant therapies. As
well, some newborns with ankyloglossia and breastfeeding
difficulties may have adapted over time with or without
tongue-tie release [42].
Subjective maternal pain scores on a visual analogue scale is an
important outcome measure due to the lack of validated objective
measures of breastfeeding pain; it is straightforward and has been
proven to be reliable in many studies [43]. Four studies in the
current review used such scale to assess maternal pain and they all
demonstrated significant improvements post-frenotomy [18–
20,31]. However, only 1 study had a control group [31] and
therefore, treatment bias may have contributed to the positive
results.
The optimal timing for tongue-tie division is still unclear. Late
division may produce a frustrated mother and baby, and impair
recovery of normal feeding patterns [44]. Thus, 1 author has
recommended that division for symptomatic babies should be
 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646
done by 2 weeks of age [31]. Another study concluded that infant’s
ability to feed was significantly better if tongue-tie division was
performed during the first week of life [28]. However, this
conclusion was based on a retrospective telephone survey study
with a low response rate (24.9%), which suggests selection bias.
Moreover, early tongue-tie release may prevent adequate time for
the newborn to adjust and also not allow enough time for the
mother and baby to receive maximal conservative therapy.
4.3. Speech outcomes
Currently, there is no strong evidence that ankyloglossia causes
speech problems. Few studies have suggested an association but
they are of low quality and a firm conclusion cannot be drawn.
In a case–control study, with 2 blinded speech pathologists,
who assessed speech intelligibility with a standardized articula-
tion test, there were no significant differences in speech outcomes
between the groups, although treated children had fewer
articulation errors than non-treated children [21]. Interestingly,
this study was carried out in Hebrew which may make the results
less transferable to English speaking and other language speaking
children.
Messner and Lalakea studied 21 children at the age of 2 years or
above who had undergone frenotomy [25]. Although they reported
that 70% of the children had articulation errors believed to be due
to reduced tongue mobility, details of those articulation errors
were lacking, nor did they provide a control group for comparison.
Interestingly, the same group reported in another study, that some
children developed normal speech and compensated for limited
tongue-tip mobility without surgical repair or need for speech
therapy [36].
The study by Heller et al. comparing two different types of
frenuloplasty techniques did not state how the articulation
assessment was performed [23]. Also, the subjects were 3 years
of age or older, which is a demographic where articulation errors
are common regardless of tongue pathology [45]. The follow-up
length for this study was not consistent and it was unclear if
subjects received speech therapy, in addition to surgical interven-
tion.
Another study identified 15 older patients (mean age 28.5 years,
range 14–68) with ankyloglossia and on a non-validated question-
naire, 50% self-identified speech difficulty and 57% noted non-
speech related mechanical limitations [25]. This was the only study
that prospectively evaluated these mechanical issues, but it had a
small sample size with no control group, and there was no formal
speech assessment [25].
In another study, speech improvements were reported in 9 of
15 patients (mean age years 4.1, range 1–12) who underwent
frenuloplasty [36]. However, again, this was a small uncon-
trolled sample, of which, some subjects concurrently received
tympanostomy tubes, suggesting possible underlying hearing
loss. Additionally, multiple speech pathologists performed the
assessments, potentially introducing inter-rater variability. As
well, the type and timing of speech assessments was not
explicitly stated, and it was unclear if subjects received
continued speech therapy [36].
Amongst many speech language pathologists, it is well
recognized that children with ankyloglossia often have normal
speech [46]. In the English language, even the sounds that require
the most amount of tongue movement, such as /l/ and /th/, can be
produced with minimal distortion, with the tongue tip pressed
down instead of up towards the alveolar ridge (/l/), or protruding
out (/th/). Similarly, other sounds that require tongue elevation,
such as /s/ and /z/ can also be produced effectively with the tongue
tip down [46]. Consequently, ankyloglossia should not have a
dramatic impact on speech function in most cases.
645
4.4. Adverse events
Bleeding was frequently observed following tongue-tie release
[18]. However, most patients produced no more than few drops of
blood, and applying local pressure on the floor of mouth was
adequate [20,22,30].
Recurrence of tongue-tie was reported in only 2 [18,28] of 11
studies. Together, they reported a 3.7% recurrence rate in young
infants following outpatient frenotomy. Mostly, these were due to
excessive scarring with re-fusion of the lingual frenulum. Other
rare adverse events in the literature include damage to the tongue
and submandibular ducts [12].
Overall, the current data suggests that there are no major safety
concerns regarding tongue-tie release when performed by trained
healthcare professionals.
4.5. Limitations
Although most studies used consecutive samples, some were
not clearly defined and all studies required the caregiver or the
patient to give consent prior to the tongue-tie release procedure.
To this end, the presence of selection bias cannot be avoided when
considering the outcomes. Moreover, many studies involved
samples from specialized breastfeeding clinics, where motivated
mothers tend to seek additional aid. Again, these subjects may be
more likely to report improvements due to their enthusiasm and
motivation.
The breastfeeding measures were mainly subjective, and
therefore can be biased. For instance, many mothers may be
subject to treatment bias especially in the immediate post-
treatment period. However, there is a lack of ankyloglossia
associated breastfeeding (and speech articulation) outcome
measures that have been validated for use in clinical trials.
Furthermore, subjective outcome measures may be reliable in
many circumstances, and at times even more so than some
objective measures (see above).
Of the 20 studies reviewed, only 5 were RCTs. Therefore, many
studies lacked a comparison or control group which makes the
positive results less compelling. As well, the earlier RCTs were
unblinded and lacked a formal sample size calculation. Further-
more, only one study presented effect size calculation [32].
4.6. Conclusion and recommendations
From this review, we conclude that tongue-tie division
improves many aspects of breastfeeding for most newborns
and their mothers. Not only have tongue-tie release been shown
to facilitate breastfeeding, and enhance milk transfer to the
infant, but they also contribute to protection of maternal nipple
and breast health. Tongue-tie division is also a relatively
straightforward and safe procedure with very low complication
rates. However, the procedure should only be performed by a
trained healthcare professional, in newborns with significant
ankyloglossia and associated breastfeeding problems who
have failed conservative management. It is to be noted that
not all infants with ankyloglossia will have breastfeeding
problems and many will adapt or respond to conservative
therapy.
Moving forward, a quantitative measure of infant nipple latch
(specific to ankyloglossia) is required, and in future studies,
validated measures of the severity of ankyloglossia and breast-
feeding outcomes should be developed and used. To further this
area, more long-term studies are required. Specifically, additional
studies are needed to elucidate the definition and significance of
ankyloglossia with regard to the proper timing of the corrective
procedure.
646 A.N. Webb et al. / International Journal of Pediatric Otorhinolaryngology 77 (2013) 635–646
There are no data in the literature on any significant association
between speech difficulties and tongue-tie in children. Therefore,
we cannot recommend tongue-tie division in early infancy for the
indication of the prevention of future articulation problems.
Funding source
No external funding was secured for this study.
Financial disclosure
The authors have no financial relationships relevant to this
article to disclose.
Conflict of interest
The authors have no conflicts of interest to disclose.
Acknowledgements
The authors would like to acknowledge Darlene Chapman for
her help with the literature search and Pam Parker for helping with
the acquisition of articles.
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