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Review Article
Does frenotomy improve breast-feeding difficulties in infants
with ankyloglossia?
Yasuo Ito
Department of Pediatrics and Pediatric Surgery, International University of Health and Welfare, Atami Hospital, Atami,
Japan
Abstract The aim of this systematic review was to critically examine the existing literature regarding the effectiveness of
tongue-tie division in infants with ankyloglossia, using the new grades of recommendations, assessment, development,
and evaluation (GRADE) rating system. A clinical question was structured according to patient, intervention, compari-
son, and outcome, as follows: in infants with poor breast-feeding and ankyloglossia (patient), does frenotomy (inter-
vention), compared to lactation support alone (comparison), improve feeding (outcome)? An electronic literature search
was systematically conducted from databases including PubMed, Japana Centra Revuo Medicina (Igaku Chuo Zasshi),
CINAHL, and Cochrane Library using the key words “ankyloglossia,” “tongue-tie,” “frenotomy,” and/or “breast-
feeding” in English and equivalent terms in Japanese. The literature search yielded four randomized clinical trials, and
12 observational studies for analysis. The quality of the literature was rated in regard to the two most important outcomes
(sucking/latching, and nipple pain) and five less important outcomes (milk supply/milk production, continuation of
breast-feeding, weight gain, adverse events, and dyad distress) in accordance with the GRADE system. Evidence levels
of the most important outcomes were rated either A (strong evidence) or B (moderate evidence), and less important
outcomes were rated C (weak evidence); every outcome consistently showed a favorable effect of frenotomy on
breast-feeding. The literature review supported an overall moderate quality of evidence for the effectiveness of
frenotomy for the treatment of breast-feeding difficulties in infants with ankyloglossia. No major complications from
frenotomy were reported.
Key words ankyloglossia, breast-feeding, frenotomy, GRADE, systematic review.
Ankyloglossia or tongue-tie is a congenital oral condition char-
acterized by an abnormally short and thick lingual frenulum,
which restricts tongue movement. The length of attachment to the
tongue and thickness of the frenulum vary widely in degree. It is
often classified into mild “partial ankyloglossia”, which is
common, and severe as well as rare “complete ankyloglossia”,
where the tongue is fused to the floor of the mouth.1The diag-
nosis is primarily based on the appearance of the tongue and its
functional disturbances caused by restricted tongue movement.
At present, however, there is no agreement on the clinical criteria
for diagnosis.
The effect of ankyloglossia on breast-feeding has been the
subject of controversy for many years, but ankyloglossia has
become of increased clinical concern in the past two decades
because of the resurgence of breast-feeding.2Many lactation
consultants believe that ankyloglossia can cause breast-feeding
difficulties and that a frenotomy is the treatment of choice.3
Conversely, pediatricians have been the most skeptical about its
effect.3The Japan Pediatric Society also takes the stand that
ankyloglossia does not cause feeding difficulties; therefore,
frenotomy is not necessary in infancy.4
During the last decade, many high-quality studies including
four randomized controlled trials (RCT) have demonstrated a
favorable effect of frenotomy on breast-feeding problems related
to ankyloglossia.5–8 The aim of this systematic review was to
critically examine the existing literature regarding the effective-
ness of tongue-tie division in infants with ankyloglossia, using
the new grading of recommendations assessment, development,
and evaluation (GRADE) rating system.9
Methods
The review was done by rating the level of evidence in accord-
ance with the GRADE handbook.10
Structured clinical question
A clinical question was structured according to patient, interven-
tion, comparison, and outcome as follows: in infants with poor
Correspondence: Yasuo Ito, MD PhD, Pediatrics and Pediatric Surgery,
International University of Health and Welfare, Atami Hospital, 13-1
Higashikaiganncho, Atami 413-0012, Japan. Email: yasuito
@iuhw.ac.jp
This article is based on a study first reported in the Journal of the Japan
Pediatric Society 2014; 118: 462–474, titled “Effectiveness of
frenotomy in breastfeeding difficulties in infants with ankyloglossia:
Systematic review” (in Japanese).
Received 19 February 2014; revised 30 April 2014; accepted 2 June
2014.
bs_bs_banner
Pediatrics International (2014) 56, 497–505 doi: 10.1111/ped.12429
© 2014 Japan Pediatric Society
breast-feeding and ankyloglossia (patient), does frenotomy
(intervention), compared to lactation support alone (comparison),
improve feeding (outcome)?
Patient selection criteria
Literature that focused on neonates and infants <6 months of age
who had breast-feeding problems and ankyloglossia was selected
for analysis. Patients who suffered from other oral anomalies and
disturbances of the central nervous system were excluded from
the review.
Primary outcome measures
The primary outcome measures regarding breast-feeding prob-
lems associated with ankyloglossia and their relative importance
are listed in Table 1. Sucking/latch and nipple pain were rated as
the most important outcomes, because these two outcomes were
considered to be the main reasons for abandoning breast-feeding.
Milk supply/milk production, continuation of breast-feeding,
weight gain, adverse events, and dyad distress were rated as
relatively less important outcomes. The quality of the literature
was assessed in regard to the two most important outcomes and
the five important outcomes according to the GRADE system.
Search strategy
An electronic literature search was systematically performed
using databases including PubMed (January1966–April 2013),
Japana Centra Revuo Medicina (Igaku Chuo Zasshi, January
1983–April 2013), CINAHL, and Cochrane Library using the key
words “ankyloglossia,” “tongue-tie,” frenotomy,” and/or
“breastfeeding” in English and the equivalent terms in Japanese.
The search was limited to articles written in English and Japanese.
Literature selection criteria
The literature selection criteria included RCT and observational
studies (cohort studies, case–control studies, and case series) that
matched the aforementioned patient selection criteria. Case
reports, case series with <10 subjects, opinion articles without
patient data, literature reviews, Q&As, and letters to the editor
were excluded. Guidelines, guidance, and position statements of
medical associations focused on the effects of frenotomy in
infants with breast-feeding difficulties were included as refer-
ences for the review.
Qualitative assessment of literature
The literature was divided into two groups: RCT (high-quality
group) and observational studies (low-quality group). In regard to
the RCT, downgrade factors of evidence level (risk of bias) such
as blinding, intention to treat, incomplete outcome data, selective
reporting, and other bias were assessed. Conversely, for the
observational studies, upgrade factors such as large effect, dose-
dependent gradient, and plausible confounders were assessed as
well as the risk of bias.
Statistical analysis
Meta-analysis was carried out using Review Manager (RevMan
ver. 5.2) of the Cochrane Collaboration.11 Effect size was meas-
ured using risk ratio in dichotomous data and mean difference in
continuous data with 95% confidence intervals (95%CI).
Results
Collected data
A total of 505 titles and abstracts were retrieved with the initial
search terms of ankyloglossia or tongue-tie. Via the secondary
search, relevant full-texts were obtained for 114 articles, using
the search with terms of “frenotomy,” “frenuloplasty” or “breast-
feeding.” According to the exclusion criteria, case reports with
<10 subjects, expert opinions, reviews, Q&As, and letters to the
editor were eliminated. After adding four articles by hand-
searching, four RCT, 12 observational studies,12–23 National Insti-
tute for Health and Care Excellence (NICE) guidance,24 a
guideline of the Academy of Breast feeding Medicine (ABM),25
and a position statement of the Canadian Paediatric Society26 met
the criteria for the review (Fig. 1).
Assessment of literature
The results of qualitative assessment of literature for each
outcome are as follows.
Table 1 Outcomes for evaluation and relative importance
Outcome Relative importance
Sucking/latch Most important
Milk supply/milk production Important
Nipple pain Most important
Continuation of breast-feeding Important
Weight gain Important
Adverse events Important
Dyad distress Important
Publications with the term “ankyloglossia” or
“tongue-tie” in English and Japanese (n=505)
Publications with the term “frenotomy,”“frenuloplasty,”or
“breastfeeding” in English and Japanese (n= 114)
RCT n=4
Observation studies n=12
Guidance n=1
Guideline n=1
Position statement n=1
Excluded:
Case report (<10 subjects),
expert opinion, review, Q&A,
letter to the editor (n=99)
Added:
Observational studies (CINAHL) n=2
Guidance(NICE) n=1
Guideline (hand search) n=1
Fig. 1 Flow diagram of selection of publications. RCT,
randomized controlled trial.
498 Y Ito
© 2014 Japan Pediatric Society
Sucking/latch
There were four RCT, including one non-blinded,5one single-
blinded,7and two double-blinded studies6,8 (Table 2). There was
significant improvement in overall assessment of breast-feeding
assessed by mothers in the frenotomy group, compared to the
placebo group in the two studies.5,8 The mother’s subjective
outcome measures included not only efficiency of the latch but
also factors such as nipple pain and feeding cycle. The results of
a double-blinded study by Berry et al. were less significant
(P<0.02),8reflecting a placebo effect in comparison to those of
a non-blinded study by Hogan et al.(P<0.001).5There was
no significant difference between pre-frenotomy and post-
frenotomy in latch, audible swallowing, type of nipple, comfort,
and hold (LATCH) scores (minimal difficulties, 10 points;
P=0.06).6,27 Significant improvement, however, was seen
in Infant Breast Feeding Assessment Tool (IBFAT) score
(maximum score, 15 points)28 following tongue-tie division (P=
0.029).7The LATCH score includes nipple pain as an assessment
item, but the IBFAT score does not.
In observational studies, sucking/latch immediately improved
in 57–92% after frenotomy,14,19,22 and in approximately 90% after
1–2 weeks.12,21 Assessment via LATCH score in two studies
showed significant improvement after treatment (P<0.0001;6
P<0.0517).
Milk supply/milk production
There was only one observational study on milk intake and milk
production (Table 3).17 Milk transfer (mL/min) was examined in
24 patients before and after frenotomy, and a significant increase
was observed 1 week after frenotomy (P<0.01). The 24 h milk
production (g) was measured in six mothers, and a significant
increase was also observed in these women 1 week after
frenotomy (P=0.035).
Nipple pain
There were three blinded RCT on maternal nipple pain
(Table 4).6–8 Two were assessed using a standard visual analogue
pain scale (maximum score, 10 points);6,8,29 one reported signifi-
cant improvement following tongue-tie release (P=0.001),6but
no significant difference between frenotomy and sham operation
was noted in the other (P=0.13).8A study of nipple pain,
however, using the short-form McGill pain questionnaire (SF-
MPQ; maximum score, 50 points)30 demonstrated marked
improvement in the frenotomy group compared to the sham
group, despite a placebo effect (P<0.001).7
Of five observational studies,13,16,17,21,22 significant improve-
ment in the pain score was reported in three studies (P<0.0001;3
P<0.05;17 P<0.0122); one study also reported improvement in
the SF-MPQ after frenotomy (P<0.0001).16
Continuation of breast-feeding
There were eight reports that discussed continuation of breast-
feeding >3 months after frenotomy, but they contained no con-
trols.5,8,12,14,16,18,19,23 The continuation rate of breast-feeding ranged
from 43%19 to 78%18 at 3 month follow up, which was nearly
twice the UK national average of 29%8at 4 months of age.
Table 2 Sucking/latch (most important outcome): RCT
Characteristics of included studies Risk of bias Summary of findings
First
author
Year
Age No.
participants
Follow up Blinding ITT Incomplete
outcome
data
Selective
reporting
Other
bias
Event ratio or mean ±SD Effects Results
Participant Provider Data
collector
Frenotomy Control Risk ratio or mean
difference (95%CI)
Hogan5
2005
3–70
days
Frenotomy
(n=20)
Placebo
(n=20)
48 h High High High Low Low Low Low 19 of 20 1 of 20
(placebo)
19.00 (2.81–128.69) Overall improvement of
breast-feeding;
significant result:
P<0.001
Dollberg6
2006
1–21
days
Frenotomy
(n=14)
Sham op
(n=11)
Immediate Low Low Low Low Low Low Low 6.8 ±2.0 6.4 ±2.3 (before
operation)
0.40 (−1.20 to 2.00) LATCH score; result not
significant: P=0.06
Buryk7
2011
<30
days
Frenotomy
(n=30)
Sham op
(n=28)
Immediate Low Low High Low Low Low Low 11.6 ±0.81 9.3 ±0.69
(before
operation)
2.30 (1.92–2.68)
(Sham operation
−0.41 [−0.83 to
0.01])
IBFAT score; significant
result: P=0.029
Berry8
2012
≤3
months
Frenotomy
(n=27)
Sham
(n=30)
Immediate Low Low Low Low Low Low Low 21 0f 27 14 of 30
(placebo)
1.67 (1.08–2.57) Overall improvement of
breast-feeding;
significant result:
P<0.02
CI, confidence interval; IBFAT score, Infant Breast Feeding Assessment Tool (maximum score, 15 points);28 ITT, intention to treat; LATCH score, latch, audible swallowing, type of nipple, comfort (breast/nipple), hold (positioning)/help
(minimal difficulty, 10 points).27
Frenotomy in infants with ankyloglossia 499
© 2014 Japan Pediatric Society
Table 3 Milk supply/milk production (important outcome): Observational study
Characteristics of included studies Risk of bias Upgrade factor Summary of findings
First
author
Year
Age No.
participants
Follow
up
Blinding ITT Incomplete
outcome
data
Selective
reporting
Other
bias
Large
effect
Dose-
dependent
gradient
Plausible
confounder
mean ±SD Effects Results
Frenotomy Control Mean difference (95%CI)
Geddes17
2008
Average 3 days 24 >1 week High Low Low Low Low Large N/A N/A Milk transfer (mL/min) (n=24) Milk transfer; significant
result: P<0.01
24 h milk production;
Significant result:
P=0.035
10.5 ±5.5 5.6 ±3.0 (before operation) 4.90 (2.39–7.41)
24 h milk production (g) (n=6))
615 ±289 455 ±323 (before operation) 160.00 (−186.80 to 506.80)
CI, confidence interval; N/A, not applicable; ITT, intention to treat.
Table 4 Nipple pain (most important outcome): RCT
Characteristics of included studies Risk of bias Summary of findings
First
author
Year
Age No.
participants
Follow up Blinding ITT Incomplete
outcome
data
Selective
reporting
Other
bias
mean ±SD Effects Results
Participant Provider Data
collector
Frenotomy Control Mean difference (95%CI)
Dollberg6
2006
1–21
days
Frenotomy
(n=14)
sham
operation
(n=11)
Immediate Low Low Low Low Low Low Low 5.3 ±2.2 7.1 ±1.9 (before
operation)
1.80 (0.28,3.32) Pain score;
significant result:
P=0.001
No data for sham
operation
Buryk7
2011
<30
days
Frenotomy
(n=30)
sham
operation
(n=28)
Immediate Low Low Low Low Low Low Low Frenotomy SF-MPQ (nipple
pain scale);
significant result:
P<0.001
4.9 ±1.46 16.77 ±1.88
(before
operation)
−11.87 (−12.72 to −11.02)
Sham operation
13.5 ±1.5 19.25 ±1.9
(before
operation)
−6.65 (−6.65 to−4.85)
Berry8
2012
≤3
months
Frenotomy
(n=14)
sham
operation
(n=14)
Immediate Low Low Low Low Low Low Low −2.5 ±1.9
(frenotomy)
−1.3 ±1.5
(sham
operation)
−1.20 (−2.47 to 0.07) Pain score; result not
significant:
P=0.13
CI, confidence interval; N/A, not applicable; ITT, intention to treat; Pain score, standard visual analogue pain scale (maximum score 10 points);29 SF-MPQ, short-form McGill pain questionnaire (maximum score 50 points).30
500 Y Ito
© 2014 Japan Pediatric Society
Weight gain
There was only one report that dealt with weight gain (Table 5).21
It noted that the neonates had gained significant weight by 15
centiles 2 weeks after frenotomy (P<0.0001).
Adverse events
A total of 1795 patients from the included articles underwent
frenotomy without any major adverse events.5–8,12–23 Minor bleed-
ing was usually readily controlled by applying gentle pressure to
the site with a sponge. Among the collected articles, one article
reported two cases of hemorrhagic shock following frenotomy in
Nigeria: one was performed by a traditional birth attendant and the
other was performed by an untrained community health worker.31
Dyad distress
There were no reports that specifically discussed distress of the
infant or the mother. Breast-feeding stress was included as part of
the overall assessment by the mother regarding sucking and latch.
Guidelines, guidance, and position statement
In 2004, the ABM developed guidelines for the evaluation and
management of neonatal ankyloglossia and its complications in
the breast-feeding dyad, based on observational studies up to
2002.25 They reported that, although conservative management of
tongue-tie is usually sufficient, frenotomy may be considered
appropriate for partial ankyloglossia and, if necessary, the pro-
cedure should be performed by an experienced physician or
pedodontist.
In 2005, NICE of the UK issued an interventional procedure
overview regarding division of ankyloglossia in infants with
breast-feeding difficulties.24 They referenced data from an unpub-
lished RCT by Hogan et al.5In the guidance, they noted that
current evidence suggests that there are no major safety concerns
about division of ankyloglossia, and the procedure can improve
breast-feeding. They cautioned, however, that to date the evi-
dence was limited.
A position statement of the Canadian Paediatric Society was
published in 2011,26 which referenced a RCT by Hogan et al.,5
but it did not reference a double-blinded RCT by Dollberg et al.
in 2006.6The position statement noted that frenotomy is not
commonly recommended, but if an association between signifi-
cant tongue-tie and major breast-feeding problems is clearly
identified and surgical intervention is deemed necessary,
frenotomy should be performed by a clinician experienced with
the procedure. The American Academy of Pediatric Dentistry
also expresses a similar position in its guideline on pediatric oral
surgery, formulated in 2010.32 It noted that frenuloplasty or
frenectomy may be a successful approach to facilitate breast-
feeding when indicated. Subsequently, two more blinded RCT
were reported,7,8 but, to date, no guidelines based on this new
evidence have been published.
Discussion
The quality of the literature was rated in regard to the two most
important outcomes (sucking/latch and nipple pain) and five
Table 5 Weight gain (important outcome): observational study
Characteristics of included studies Risk of bias Upgrade factor Summary of findings
First
author
Year
Age No.
participants
Follow
up
Blinding ITT Incomplete
outcome
data
Selective
reporting
Other
bias
Large
effect
Dose-dependent
gradient
Plausible
confounders
mean ±SD Effects Results
Frenotomy Control Mean difference
(95%CI)
Miranda21
2010
12–36 days 62 2 weeks High High Low Low Low Very
large
N/A N/A 56 ±2.4 41 ±2.5
(before
operation)
15 (14.05–15.95) Weight gain by
centile; significant
result: P<0.0001
CI, confidence interval; N/A, not applicable; ITT, intention to treat.
Frenotomy in infants with ankyloglossia 501
© 2014 Japan Pediatric Society
relatively less important outcomes (milk supply/milk production,
continuation of breast-feeding, weight gain, adverse events, and
dyad distress) according to the GRADE system.
Sucking or latch was difficult to assess independently from
other outcomes because maternal subjective outcome measures
included nipple pain, feeding time, infant satisfaction, and mater-
nal distress. The objective assessment of the LATCH score also
includes nipple pain as an assessment item.
An RCT by Hogan et al. and an RCT by Berry et al. were both
reported from the same institution, but on different subjects.5,8
The RCT by Berry et al. was double-blinded to supplement the
weakness of the earlier non-blinded study.5,8 Meta-analysis of
these two RCT indicated strong evidence (risk ratio, 2.88;
95%CI: 1.82–4.57) in favor of the frenotomy group compared to
the placebo group (Fig. 2a). Meta-analysis of two observational
studies of LATCH scores also supported the effectiveness
of frenotomy (mean difference, 2.07; 95%CI: 1.64–2.49;
Fig. 2b).16,17
Geddes et al. researched tongue movement during breast-
feeding using submental ultrasonography, and showed that milk
flowed into the infant’s mouth from the mother’s nipple by nega-
tive pressure created by up and down movements of the infant’s
tongue.17 In tongue-tie babies, these actions were disturbed, and
the nipple remained compressed by the baby’s tongue. Release of
the tongue-tie reduced the nipple compression. The rate of milk
transfer increased by almost twofold, and 24 h milk production
also increased in 1 week.
Of three blinded RCT evaluating nipple pain,6–8 one using pain
score and one using SF-MPQ reported significant improvement
of nipple pain immediately after frenotomy, compared with
preoperative pain or pain following a sham operation.6,7 Meta-
analysis of three observational studies also demonstrated a
marked decrease of nipple pain with a mean difference of −5.10
(95%CI: −5.60 to −4.59) in the frenotomy group, compared to the
placebo group (Fig. 2c).13,17,22
The continuation rate of breast-feeding 3 months after
frenotomy was twofold higher than that of the average rate of
4-month-old infants in the UK.12,14,16,18,19 Weight significantly
increased after frenotomy by centile in 2 weeks.21 There were no
major adverse events when the procedure was performed by an
experienced health-care professional. In addition, frenotomy is
an inexpensive procedure; thus, it has minimal impact on health-
care costs.
The results of this systematic review are summarized in the
evidence profile (Table 6). The results of outcomes consistently
showed the effectiveness of frenotomy for breast-feeding
(a)
Study or Subgroup
Berry 2012
8
Hogan 2005
5
Total (95% CI)
Total events
Heterogeneity: Chi² = 9.89, df = 1 (P = 0.002); I² = 90%
Test for overall effect: Z = 4.50 (P < 0.00001)
Events
21
19
40
Total
27
20
47
Events
14
1
15
Total
30
20
50
Weight
93.0%
7.0%
100.0%
M-H, Fixed, 95% CI
1.67 [1.08, 2.57]
19.00 [2.81, 128.69]
2.88 [1.82, 4.57]
Frenotomy Placebo Risk Ratio Risk Ratio
M-H, Fixed, 95% CI
0.02 0.1 110 50
Favors [placebo] Favors [frenotomy]
(b)
Study or Subgroup
Geddes et al. 2008
17
Srinivasan et al. 2006
16
Total (95% CI)
Heterogeneity: Chi² = 5.22, df = 1 (P = 0.02); I² = 81%
Test for overall effect: Z = 9.52 (P < 0.00001)
Mean
9.4
9.2
SD
0.8
0.9
Total
24
27
51
Mean
7.9
6.7
SD
1.4
1.2
Total
24
27
51
Weight
43.5%
56.5%
100.0%
IV, Fixed, 95% CI
1.50 [0.85, 2.15]
2.50 [1.93, 3.07]
2.07 [1.64, 2.49]
After frenotomy Before frenotomy Mean Difference Mean Difference
IV, Fixed, 95% CI
-4 -2 0 2 4
Favors [pre-frenotomy] Favors [post-frenotomy]
(c)
Study or Subgroup
Argiris et al. 201122
Ballard et al. 200213
Geddes et al. 200817
Total (95% CI)
Heterogeneity: Chi² = 11.63, df = 2 (P = 0.003); I² = 83%
Test for overall effect: Z = 19.79 (P < 0.00001)
Mean
1.47
1.2
0.5
SD
1.34
1.52
1.24
Total
46
53
24
123
Mean
6.63
6.9
3.6
SD
2.46
2.31
3
Total
46
53
24
123
Weight
38.9%
46.0%
15.1%
100.0%
IV, Fixed, 95% CI
-5.16 [-5.97, -4.35]
-5.70 [-6.44, -4.96]
-3.10 [-4.40, -1.80]
-5.10 [-5.60, -4.59]
After frenotomy Before frenotomy Mean Difference Mean Difference
IV, Fixed, 95% CI
-10 -5 0 5 10
Favors [post-frenotom
y
] Favors [pre-frenotom
y
]
Fig. 2 Meta-analysis of breast-feeding. (a) Overall improvement evaluated by mother; (b) latch evaluated by latch, audible swallowing, type
of nipple, comfort, and hold (LATCH) score; (c) nipple pain evaluated by pain score. CI, confidence interval.
502 Y Ito
© 2014 Japan Pediatric Society
Table 6 Evidence profile
Outcome Study
design, n
Risk of bias Bias across studies Overall quality
of evidence
Summary of findings
Inconsistency Indirectness Imprecision Publication
bias
Effects (Risk ratio or
mean difference (95%CI))
Results
Sucking/latch RCT, 4 ○○○○○ A Overall improvement
(meta-analysis†): 2.88
(1.82–4.57)
IBFAT: 2.30 (1.92–2.68)
Significant results in 3
studies
Observational
study, 12
×○○○○ B LATCH score
(meta-analysis‡): 2.07
(1.64–2.49)
Significant
improvement in
LATCH score in 2
studies
Milk supply/milk
production
Observational
study, 1
△○○△○ C Milk transfer (mL/min):
4.90 (2.39, 7.41)
Significant in milk
transfer
Nipple pain RCT, 3 ○○○○○ A Pain score: 1.80
(0.28,3.32)
SF-MPQ (Frenotomy:
−11.87 (−12.72 to
−11.02); Sham
operation: −6.65 (−6.65
to −4.85)
Significant results in 2
studies
Observational
study, 5
×○○○○ B Pain score
(meta-analysis§): −5.10
(−5.60 to−4.59)
Significant
improvement in pain
score in 3 studies
Continuation of
breast-feeding
Observational
study, 8
×○○○○ C No statistical analysis Breast-feeding at 3
months after
frenotomy
43.2–77.8%,
Breast-feeding rate
at 4 m/o in UK
average 29%
Weight gain Observational
study, 1
△○○△○ C Weight gain by centile: 15
(14.05–15.95)
Significant result
Adverse events Observational
study, 16
△○○○○ C No statistical analysis No adverse events
†Fig. 2a, ‡Fig. 2b, §Fig. 2c. Bias risk: ○, not serious, △, serious, ×very serious; CI, confidence interval; IBFAT score, Infant Breast Feeding Assessment Tool (maximum score, 15 points);28
ITT, intention to treat; LATCH score, latch, audible swallowing, type of nipple, comfort (breast/nipple), hold (positioning)/help (minimal difficulty, 10 points);27 RCT, randomized controlled
trial; SF-MPQ, short-form McGill pain questionnaire (maximum score 50 points);30 strength of evidence: A, strong; B, moderate; C, weak; D, very weak.
Frenotomy in infants with ankyloglossia 503
© 2014 Japan Pediatric Society
difficulties. Evidence levels of the most important outcomes
(sucking/latch and nipple pain), presented by RCT and meta-
analysis of observational studies, were rated either A (strong
evidence) or B (moderate evidence); the other four important
outcomes were rated C (weak evidence) because they were
purely observational studies, contained a small number of sub-
jects, and/or lacked controls. Further RCT with long-term follow
up are indicated, but this type of study is always affected by
ethics problems. In addition, it is impossible to conceal
frenotomy or sham operation from the mother for a long period of
time.
Among existing guidelines, guidance statements, and position
statements, the NICE guidance is the only one that was formu-
lated via systematic review,24 but it is a brief overview and not
definitive. Despite limited evidence, the NICE guidance recom-
mends frenotomy in infants with breast-feeding difficulties asso-
ciated with ankyloglossia. After the publication of the guidance,
three more RCT were published;6–8 they strengthened the evi-
dence level, which was lacking in the NICE overview.
The present literature review found an overall moderate
quality of evidence regarding the effectiveness of tongue-tie divi-
sion for the treatment of breast-feeding difficulties in infants with
ankyloglossia. There were no major complications from
frenotomy, most likely because it was performed by well-trained
health-care professionals.
To formulate more comprehensive guidelines, a group of pro-
fessionals from all the relevant fields need to collaborate. The
present study should offer the necessary means and information
for a collaborative project.
Conclusions
A systematic review based on the GRADE system was conducted
to examine the clinical question, does frenotomy improve breast-
feeding difficulties in infants with ankyloglossia. In accordance
with the exclusion and inclusion criteria, four RCT and 12 obser-
vational studies were included in this review. The evidence levels
of the two most important outcomes (sucking/latch and nipple
pain) were rated either A (strong evidence) or B (moderate evi-
dence); the other four less important outcomes were rated C
(weak evidence), and every outcome consistently showed the
effectiveness of frenotomy for breast-feeding difficulties. No
serious adverse events were reported. The answer to the clinical
question is that frenotomy does improve breast-feeding difficul-
ties in infants with ankyloglossia. Further collaborative work is
indicated to formulate more comprehensive guidelines.
Acknowledgments
The author is grateful to Hideki Kinugasa, PhD, Professor of
Biostatistics and Clinical Epidemiology, University of Toyama
Graduate School of Medicine and Pharmaceutical Sciences, for
his statistical support, and to Masahiro Yoshida, MD PhD, Chief
of Medical Information Network Distribution Service of Japan
Council for Quality Health Care, for his editorial advice on the
GRADE system. The author received no financial support for this
study and there are no conflicts of interest.
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