Comparison of Frenotomy Techniques for the Treatment of Ankyloglossia in Children.pdf

Systematic Review/Meta-analysis
Comparison of Frenotomy Techniques for
the Treatment of Ankyloglossia in
Children: A Systematic Review
Otolaryngology–
Head and Neck Surgery
1–16
Ó American Academy of
Otolaryngology–Head and Neck
Surgery Foundation 2020
Reprints and permission:
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DOI: 10.1177/0194599820917619
http://otojournal.org
Usman Khan, MSc1
, Jake MacPherson2
,
Michael Bezuhly, MD, MSc1,3
, and Paul Hong, MD, MSc1,2,3
Abstract
Objective. To compare the effectiveness of conventional (CF),
laser (LF), and Z-plasty (ZF) frenotomies for the treatment
of ankyloglossia in the pediatric population.
Data Sources. A comprehensive search of PUBMED,
EMBASE, and COCHRANE databases was performed.
Review Methods. Relevant articles were independently assessed
by 2 reviewers according to the Preferred Reporting Items for
Systematic Reviews and Meta-Analysis (PRISMA) guidelines.
Results. Thirty-five articles assessing CF (27 articles), LF (4
articles), ZF (3 articles), and/or rhomboid plasty frenotomy
(1 article) were included. A high level of outcome heteroge-
neity prevented pooling of data. All 7 randomized controlled
trials (RCTs) were of low quality. Both CF (5 articles with
589 patients) and LF (2 articles with 78 patients) were inde-
pendently shown to reduce maternal nipple pain on a visual
analog or numeric rating scale. There were reports of
improvement with breastfeeding outcomes as assessed on
validated assessment tools for 88% (7/8) of CF articles (588
patients) and 2 LF articles (78 patients). ZF improved
breastfeeding outcomes on subjective maternal reports (1
article with 18 infants) only. One RCT with a high risk of
bias concluded greater speech articulation improvements
with ZF compared to CF. Only minor adverse events were
reported for all frenotomy techniques.
Conclusions. Current literature does not demonstrate a clear
advantage for one frenotomy technique when managing chil-
dren with ankyloglossia. Recommendations for future research
are provided to overcome the methodological shortcomings in
the literature. We conclude that all frenotomy techniques are
safe and effective for treating symptomatic ankyloglossia.
Keywords
ankyloglossia, frenotomy, tongue-tie
Received August 25, 2019; accepted February 23, 2020.
A
nkyloglossia or tongue-tie is a congenital condition
where anatomical variation of the sublingual frenu-
lum can limit normal tongue function.1,2
The most
common problems associated with ankyloglossia are breast-
feeding difficulties, which include maternal nipple pain, poor
latch, poor milk transfer, and poor infant weight gain.3
In
recent years, there has been a drastic increase in the diagnosis
of ankyloglossia and publication of articles investigating
management strategies.4,5
Surgical intervention with frenot-
omy remains the primary treatment for patients who experi-
ence significant symptoms.4
Conventional frenotomy (CF) is a short procedure where
the lingual frenulum is released with scissors or a scalpel. A
laser can also be used and is gaining popularity in the cur-
rent literature.6-12
Z-plasty technique is a modification of
the conventional approach to minimize scar contracture and
is performed with different flap variations.13,14
While sev-
eral reports claim improved outcomes with laser frenotomy
(LF) or Z-plasty frenotomy (ZF), controversy exists regard-
ing the benefit of these techniques compared to CF.13-17
Specifically, studies using LF or ZF suggest enhanced
improvements in functional outcomes such as speech articu-
lation and reduced complications such as decreased blood
loss.8,13,14,18
A recent Cochrane review of 5 randomized
control trials (RCTs) demonstrated the effectiveness of CF
for the treatment of maternal nipple pain during breastfeed-
ing.19
However, the RCTs were reported to be of low qual-
ity with heterogenous outcome assessments and CF was the
only technique included.19
The objective of this systematic review was to address
the controversy regarding the benefits of choosing conven-
tional, laser, or Z-plasty techniques for the treatment of
ankyloglossia in the pediatric population. In particular, func-
tional outcomes were assessed, as was the overall quality of
evidence supporting different surgical techniques.
1
Department of Surgery, Faculty of Medicine, Dalhousie University, Halifax,
Nova Scotia, Canada
2
School of Communication Sciences and Disorders, Dalhousie University,
Halifax, Nova Scotia, Canada
3
Department of Surgery, IWK Health Centre, Halifax, Nova Scotia, Canada
This paper was presented at the Canadian Society of Otolaryngology–Head
and Neck Surgery Annual Meeting; June 3, 2019; Edmonton, Alberta,
Canada.
Corresponding Author:
Usman Khan, MSc, Department of Surgery, Faculty of Medicine, Dalhousie
University, 1459 Oxford Street, Halifax, NS B3H 4R2, Canada.
Email: usman.khan@dal.ca

Methods
Protocol and Research Question
This systematic review was conducted in accordance with
the Preferred Reporting Items for Systematic Reviews and
Meta-Analysis (PRISMA) guidelines. A focused research
question was formulated using the patient/population, inter-
vention, comparison, and outcomes (PICO) criteria (popula-
tion: pediatric patients with ankyloglossia; intervention:
frenotomy; comparison: CF (frenotomy and/or frenulo-
plasty), LF, or ZF; outcomes: subjective and objective
assessments of function [breastfeeding, speech, tongue
movement] and adverse events [bleeding, infection, need for
repeat procedures]).
Study Identification
A computerized search of EMBASE, PUBMED, and
Cochrane Library databases from inception to June 2, 2018,
was performed with the assistance of an experienced librar-
ian at Dalhousie University in Halifax, Nova Scotia (Figure
1). The search strategy included synonyms for ankyloglossia
and frenotomy. The articles were imported to Covidence
(Melbourne, Australia) software for screening and data
extraction. All duplicates were removed. Considering the
lack of high-quality studies and diverse body of literature
on this topic, the eligibility criteria were tailored for a
mixed-methods systematic review. Strict inclusion and
exclusion criteria were outlined for all phases of article
screening (detailed in Suppl. Figure S1 in the online version
of the article). The inclusion/exclusion criteria adhered to
the PICO format to reflect the research question and
included studies that used qualitative or quantitative meth-
ods for assessing outcomes. A reason was chosen for each
article that was excluded. All phases of article screening
were performed by 2 reviewers (U.K. and J.M.) indepen-
dently. A consensus meeting was held to discuss any con-
flicts. If a consensus was not reached, the senior author
(P.H.) was involved to make the final decision.
Data Extraction
A standardized extraction form was generated in the
Covidence software for all articles. The form included the
following items for data extraction: authors, country, partici-
pant number, sex distribution, age, study type, frenotomy
techniques, patient groups, methodology, time to outcome
assessment, outcome results, and statistics. Subjective and
objective outcomes were collected. Quantitative data were
recorded as mean or median values with standard deviation
or quartile ranges when available. Significance was recorded
as P values when available. The data extraction was con-
ducted by 2 reviewers independently and conflicts were
resolved by a consensus meeting of all reviewers.
Quality Assessment
The majority of research available on frenotomies were
observational studies (cohort studies or case series).
Therefore, quality assessment for non-RCTs was performed
using the Methodological Index for Non-Randomized
Studies (MINORS).19
This is a validated assessment tool
designed specifically for non-RCT studies using 12 items,
each of which is scored as 0 (not reported), 1 (reported but
inadequate), or 2 (reported and adequate). The tool allows
separate evaluation of comparative and noncomparative
methods by assigning the first 8 items to noncomparative
studies only (total of 16 points) and an additional 4 items for
comparative studies (total of 24 points). The quality of RCTs
studying CF only was previously evaluated in a Cochrane
review using the Cochrane Collaboration Risk of Bias
Tool (RoB).19
In this study, RCTs investigating any type
of frenotomy were assessed for bias using the modified
RoB 2.0.20
This tool allows an assessment of an overall
risk of bias and eliminates the ‘‘other bias’’ domain. The
overall quality of evidence supporting frenotomy tech-
niques for various outcomes was assessed using the
Grading of Recommendations Assessment, Development,
and Evaluation (GRADE) approach.21
Synthesis of Results
A descriptive approach was undertaken to report the results
of this systematic review. Studies reporting outcomes using
standardized assessment tools were compiled together. The
data were presented in tables for similar outcomes and
arranged based on frenotomy technique. Weighted averages
were calculated for objective outcome measurements.
Univariate comparisons were conducted using a x2
test for
categorical variables and Student t test for continuous vari-
ables. Statistical significance was defined as P .05.
Results
Study Selection
The initial literature search identified a total of 1036 articles
following the removal of duplicates (Figure 1). Abstract
screening led to the inclusion of 99 articles for full-text
review. Sixty-four articles were excluded during the full-
text review for the following reasons: wrong study designs
(47), non-English articles with no translations (6), and
wrong patient population (11). Therefore, a total of 35 arti-
cles met the inclusion criteria for final synthesis (Table
1).3,6,7,11,13,14,22-50
Wrong study designs included studies
with interventions such as labial frenotomy only, assessment
of diagnostic outcomes, reports with only 1 patient, and
mixed indications for surgery that were not specific for dif-
ficulties associated with ankyloglossia. Studies investigating
a patient population that included nonpediatric subjects
were also omitted from the review.
Study Characteristics
CF, LF, and ZF were evaluated in 27 of 35 (77%), 4 of 35
(11%), and 3 of 35 (9%) articles, respectively. One article
evaluating a rhomboid plasty variation was also included.
The total number of patients were 1856 for CF, 108 for LF,
and 243 for ZF or rhomboid-plasty frenotomy. The follow-
ing countries were represented in the review: Australia (2),
Brazil (2), Canada (1), India (1), Iran (1), Ireland (2),
2 Otolaryngology–Head and Neck Surgery

Israel (3), Japan (2), South Korea (1), Spain (1), Thailand
(1), United Kingdom (11), and United States (7).
The overall mean (SD) age of participants was 1.04 (2.3)
years (range, 2 days to 8 years). LF and ZF studies had
older patients with mean ages of 3.1 and 4.0 years, respec-
tively, compared to 0.4 years for CF patients (P .05).
There were more male than female patients for all types of
frenotomy. Indications for frenotomy primarily included
breastfeeding difficulties and speech problems.
Quality of Evidence
The majority of articles assessed breastfeeding, speech, or
tongue movement as postsurgical outcomes (Table 2). A
total of 7 RCTs were included: 1 study compared ZF with
CF, 1 study compared ZF with conventional frenuloplasty
(horizontal to vertical), and 5 compared CF with no treat-
ment or sham control group (see Suppl. Table S1 in the
online version of the article). The remaining articles were
observational studies (case series, case-control, or cohort
studies). Outcomes for all studies were assessed using ques-
tionnaires, interviews, or telephone conversations. The
MINORS criteria were used for non-RCT studies, which
represent the majority of included articles. The research
methodologies were variable, which is reflected in the range
of quality assessment scores (Table 2). The overall quality
remained low as only 4 of 28 articles provided a control
group for comparison. The remainder of the articles were
assessed on the 8 noncomparative parameters. In particular,
the major limitations were the inability to successfully blind
participants and the lack of sample size calculations. The
articles were consistent in stating a clear aim for the study,
inclusion of consecutive patients, and using a protocol for
collecting data.
The overall quality of evidence based on the GRADE
approach was low for CF articles and very low for ZF and
LF articles, respectively. CF RCTs were downgraded to
low quality for imprecision (small study populations) and
risk of bias similar to a previous Cochrane review (CF-only
RCTs).19
However, several observational studies demon-
strated benefit of CF using validated outcome measures
and/or compared with controls in large patient cohorts. ZF
articles were determined to be of very low-quality evidence
due to imprecision (small sample sizes), risk of bias
(unclear randomization, incomplete blinding, lack of alloca-
tion concealment), and limitations in study design (outcome
measures were not validated). LF articles were also very
low quality with observational studies lacking control
groups, although validated outcome measures were used,
and patients were followed prospectively in some cases.
Breastfeeding Outcomes
Objective breastfeeding outcomes were assessed using vali-
dated tools: Breastfeeding Self-Efficacy Scale (BSES),
LATCH scoring tool, and Infant Breastfeeding Assessment
Tool (IBFAT). Of 8 CF studies (588 patients) using vali-
dated instruments for assessment of breastfeeding outcomes,
7 (88%) reported a significant postoperative improvement.
One LF study by Ghaheri et al6
used the BSES scale, report-
ing a mean score increase of 12.5 after frenotomy (P
.001). Another study by the same authors reported a mean
BSES score increase of 10.8 after treatment with revision
LF (P .001).7
One RCT comparing ZF to CF reported
Figure 1. Literature search flow diagram based on the Preferred Reporting Items for Systematic Review and Meta-Analyses (PRISMA).
Khan et al 3

Table 1. Summary of Patient Demographics.
Author Country Frenotomy No. Patient Demographics
Amir et al29
Australia Conventional 35 Age: 3-98 days
Sex: 22 M/13 F
Indications: Breastfeeding difficulties
Other health care professionals: Lactation
consultants were a source of referral
and interviewed patients
postoperatively. Provided additional
counseling if required.
Argiris et al30
United Kingdom Conventional 46 Age: 4 weeks (1 day to 12 weeks)
Sex: 33 M/13 F
consultants were a source of referral,
performed frenotomy in some cases and
interviewed patients postoperatively.
Ballard et al24
United States Conventional 123 Age: Unknown
Sex: 92 M/31 F
Berry et al31
United Kingdom Conventional (30) vs no surgery (30) 60 Age: 32 days
Sex: 40 M/20 F
Billington et al32
United Kingdom Conventional 87 Age: 2-88 days
Buryk et al27
United States Conventional (30) vs sham (28) 58 Age: 6 (1-35 days)
Sex: 38 M/20 F
Other health care professionals: Patients
were referred by lactation consultants.
Martinelli et al33
Brazil Conventional (14) vs no surgery (14) 28 Age: 30-75 days
Sex: 20 M/8 F
Choi et al26
South Korea Z-plasty with genioglossus myotomy 106 Age: 1-10 years
Sex: 73 M/33 F
Dollberg et al34
Israel Conventional (14) vs sham (11) 25 Age: 1-21 days
Dollberg et al35
Israel Conventional 244 Age: 14 days (1-135 days)
Sex: 143 M/101 F
consultants evaluated patients.
Emond et al36
United Kingdom Conventional (53) vs no surgery (52) 105 Age: 11 days
in both treatment and control arms
were provided with professional
lactation consultation for 5 days.
Ferrés-Amat et al23
Spain Frenectomy with rhomboid plasty 101 Age: 8 years (4-14)
Sex: 63 M/38 F
(continued)

Table 1. (continued)
Other health care professionals:
Postoperative rehabilitation and speech
therapy
Fiorotti et al22
Brazil Laser 15 Age: 7 (3-14) years
Sex: 10 M/5 F
Indications: Speech problems
Postoperative speech therapy
Geddes et al37
Australia Conventional 24 Age: 33 (4-131) days
were referred by lactation consultants
after failing therapy.
Ghaheri et al6
United States Laser 58 Age: 4.4 (3.6) weeks
Ghaheri et al7
United States Laser 20 Age: 8.3 weeks (7 days to 37 weeks)
Griffiths38
United Kingdom Conventional 215 Age: 19 days
Sex: 144 M/72 F
were referred after failing professional
support from midwife, lactation
consultant, feeding adviser, or health
visitor.
Hansen et al39
United Kingdom Conventional 44 Age: 49 (3-202) days
Sex: 25 M/19 F
Heller et al13
United States Z-plasty (11) vs frenuloplasty (5) 16 Age: 5.7 (2.14)
Sex: 7 M/4 F
Hogan et al28
United Kingdom Conventional (28) vs no surgery (29) 57 Age: 20 (3-70) days
Sex: 32 M/25 F
Nonsurgical control group received
intensive lactation consultation for 48
hours.
Ito et al40
Japan Conventional 5 Age: 5.4 (3-8) years
Sex: 4 M/1 F
Komori et al11
Japan Laser 15 Age: 5.2 years
Kumar et al41
India Conventional 60 Age: 23.3 (28.7) days
(continued)
Khan et al 5

Other health care professionals: All
patients received lactation consultation
before frenotomy.
Messner and Lalakea25
United States Conventional 30 Age: 4.1 (1-12) years
Sex: 19 M/11 F
Indications: Speech and feeding problems
Other health care professionals: All
patients received preoperative speech
pathology.
Mettias et al42
United Kingdom Conventional 36 Age: 4.1 6 3.2 weeks (before follow-up)
Indications: Breastfeeding, poor tongue
movement
Miranda et al43
Ireland Conventional 51 Age: 12-36 days
Muldoon et al44
Ireland Conventional 89 Age: 11 weeks
consultants, public health nurse, or
midwives referred patients after
therapeutic interventions.
Riskin et al45
Israel Conventional (65) vs no surgery (118) 183 Age: 8 days
Sethi et al46
United Kingdom Conventional 52 Age: 19 (3-120) days
Sex: 35 M/17 F
Sharma and Jayaraj47
United Kingdom Conventional (36) vs no surgery (6) 42 Age: 38 (15-178) days
Sex: 23 M/19 F
Srinivasan et al3
Canada Conventional 27 Age: 19 (2-71) days
Sex:18 M/9 F
Lactation counseling
Wakhanrittee et al48
Thailand Conventional 328 Age: 50 hours
Sex: 194 M/134 F
Wallace and Clarke49
United Kingdom Conventional 10 Age: 10 days (2-31) days
Sex: 8 M/2 F
Walls et al50
United States Conventional (71) vs no surgery (15) 86 Age: 9 days
Yousefi et al14
Iran Z-plasty (25) vs conventional (25) 50 Age: 32 months
Sex: 37 M/13 F
Indications: Breastfeeding problems,
speech difficulties
Abbreviations: F, female; M, male.

Table 2. Summary of Outcomes Assessed.
Author Frenotomy Study Design Study Qualitya
Outcome Assessment
Amir et al29
Conventional CS 6 Method: Postfrenotomy interview
Improved outcomes: 51% better
attachment, 57% improved sucking,
26% less pain, 17% weight
improvement, and 100%
postoperative satisfaction
Argiris et al30
Conventional CS 12 Method: Postfrenotomy survey, pain
score
Improved outcomes: Maternal-
reported improvement and pain
score
Ballard et al24
Conventional CS 8 Method: Latch problems and nipple
pain (0-10 scale)
Improved outcomes: Latch and nipple
pain
Berry et al31
Conventional (30) vs
no surgery (30)
RCT High risk of bias Method: VAS, maternal-reported
improvement
reported improvement
Billington et al32
Conventional CS 7 Method: Postfrenotomy breastfeeding
interview
Improved outcomes: 80% complete
resolution, 15% moderate resolution,
and 5% minimal resolution
Buryk et al27
sham (28)
RCT High risk of bias Methods: IBFAT, SF-MPQ score
Improved outcomes: SF-MPQ, IBFAT
Martinelli et al33
no surgery (14)
PS 15 Methods: Postfrenotomy questionnaire
Improved outcomes: Average number
of sucks and pause length
Choi et al26
Z-plasty with
genioglossus
myotomy
CS 4 Methods: Postfrenotomy interview
Improved outcomes: Speech problems
and scar contracture
Dollberg et al34
sham (11)
RCT High risk of bias Methods: VAS and LATCH
Improved outcomes: VAS
Dollberg et al35
Conventional CS 10 Methods: Postfrenotomy breastfeeding
interview
Improved outcomes: 75% reported
improvement with breastfeeding
Emond et al36
no surgery (53)
RCT High risk of bias Methods: BSES, HATLFF, IBFAT,
LATCH, self-efficacy score, VAS
Improved outcomes: HATLFF and
BSES (5 days only)
Frenectomy with
rhomboid plasty
CS 11 Methods: Tongue mobility
Improved outcomes: Tongue mobility
with rehabilitation
Fiorotti et al22
Laser CS 6 Methods: Postfrenotomy interview
Improved outcomes: Parent-reported
success without complications
Geddes et al37
Conventional CS 9 Methods: Milk intake (g), milk transfer
(mL/min), LATCH, NRS
Improved outcomes: Milk intake, milk
transfer, LATCH, and NRS
(continued)
Khan et al 7

Outcome Assessment
Ghaheri et al6
Laser PS 14 Methods: BSES, I-GERQ-R, VAS
Improved outcomes: BSES, I-GERQ-R,
VAS
Ghaheri et al7
Laser PS 13 Methods: BSES, I-GERQ-R, VAS
Improved outcomes: BSES, I-GERQ-R,
VAS
Griffiths38
Conventional CS 9 Methods: Postfrenotomy phone
interview, intraoperative bleeding,
patient crying
reported improvement
postfrenotomy: 57% immediately,
81% in 24 hours. Tongue extension:
98%. Breastfeeding: after 3 months,
64%.
Hansen et al39
Conventional CS 4 Methods: Phone interview, postsurgical
complications: bleeding, pain,
infection
Improved outcomes: 80% maternal-
reported improvement in
breastfeeding
Heller et al13
Z-plasty (11) vs
frenuloplasty (5)
RCT High risk of bias Methods: Frenulum length (mm),
tongue protrusion (mm), speech
articulation (1 mild-moderate-severe
scale)
Improved outcomes: frenulum length,
tongue protrusion, articulation
Hogan et al27
no surgery (29)
RCT High risk of bias Methods: Postfrenotomy breastfeeding
interview
Improved outcomes: maternal-
reported breastfeeding improvement
Ito et al40
Conventional CS 7 Methods: Speech: substitutions,
omissions, distortions
Improved outcomes: Substitutions,
omissions
Komori et al11
Laser CS 6 Methods: Complications and
postfrenotomy survey
Outcomes: No intraoperative
complications and 1 of 15
postoperative complications
Kumar et al41
Conventional CS 7 Methods: Postfrenotomy survey
Improved outcomes: Latch and
maternal pain
Conventional CS 8 Methods: Postfrenotomy questionnaire
Improved outcomes: Tongue elevation,
tongue protrusion, tongue mobility,
and speech intelligibility
Mettias et al42
Conventional CS 6 Methods: Postfrenotomy questionnaire
and surgical complications
Improved outcomes: Symptom
resolution in 96.8% of patients
(continued)

Outcome Assessment
Miranda and Milroy43
Conventional CS 11 Methods: Breastfeeding sessions,
nipple (pain, cracking, bleedings),
weight centile
Improved outcomes: Nipple bleeding,
nipple pain, nipple cracking, weight
centile, breastfeeding sessions, and
bottle-feeding sessions
Muldoon et al44
Conventional CS 10 Methods: Postfrenotomy
questionnaire, LATCH, VAS, tongue
mobility
Improved outcomes: LATCH score,
tongue extension to lower lip,
tongue extension to lower gum, VAS
Riskin et al45
no surgery (118)
CC 16 Methods: Postfrenotomy questionnaire
Improved outcomes: Self-reported
breastfeeding
Sethi et al46
questionnaire
Improved outcomes: Improvement:
31%, immediate; 15%, 24 hours; 25%,
1 week; 6%, 2 weeks
Sharma and Jayaraj47
no surgery (6)
CC 12 Methods: Postfrenotomy breastfeeding
questionnaire, IBFAT score
Improved outcomes: IBFAT, self-
reported improvement in
breastfeeding
Srinivasan et al3
Conventional CS 13 Methods: LATCH, PRI, PPI
Improved outcomes: LATCH, PRI, PPI
Conventional CS 13 Methods: LATCH, NRS
Improved outcomes: LATCH, NRS
Wallace and Clarke49
survey
Improved outcomes: 70% of mothers
reported breastfeeding improvement.
Walls et al50
no surgery (15)
CC 12 Methods: Speech (Likert scale), motor
activity
Improved outcomes: Speech, motor
activity
Yousefi et al14
Z-plasty (25) vs
conventional (25)
RCT High risk of bias Methods: Hazelbaker, speech
articulation (0-4), maternal
satisfaction with breastfeeding
(latching, mastalgia)
Improved outcomes: Hazelbaker,
articulation, maternal satisfaction
with breastfeeding
Abbreviations: BSES, Breastfeeding Self-Efficacy Scale; CC, case control; CS, case series; HATLFF, Hazelbaker Assessment Tool for Lingual Frenulum Function;
IBFAT, Infant Breastfeeding Assessment Tool; I-GERQ-R, Revised Infant Gastroesophageal Reflux Questionnaire; LATCH, breastfeeding charting tool; NRS,
numeric rating scale; PPI, Present Pain Index; PRI, Pain Rating Index; PS, prospective study; RCT, randomized control trial; SF-MPQ, Short-Form McGill Pain
Questionnaire; VAS, visual analog scale.
a
Quality scores for non-RCT studies are assessed on a scale out of 16 for noncomparative studies and out of 24 for comparative studies based on the
Methodological Index for Non-Randomized Studies criteria. Risk of bias for RCT studies was reported using the modified Cochrane Risk of Bias Assessment
Tool (RoB 2.0).
Khan et al 9

significant improvements in subjective maternal reports
postfrenotomy in both groups with no differences between
frenotomy techniques.14
Maternal nipple pain (MNP) was evaluated with the
visual analog scale (VAS), numeric rating scale (NRS), or
McGill Pain Questionnaire (MPQ) (Tables 2 and 3). Seven
of 9 (78%) CF studies reported a significant postoperative
improvement. Only 2 RCT found no significant improve-
ments for MNP after frenotomy. A patient-weighted mean
improvement in pain scores of –3.6 and –2.7 was observed
following CF and LF, respectively, for non-RCT studies
(Table 4). CF RCTs were not included as no RCTs investi-
gating MNP were available for other frenotomy methods.
Tongue Movement Outcomes
Three studies reported tongue movement as an outcome
(Table 5). Two RCTs reported a significant increase
in tongue protrusion after frenotomy for all treatment
groups.13,14
However, both studies reported a significantly
larger increase in tongue protrusion in the ZF group com-
pared to the CF and conventional frenuloplasty group (hori-
zontal to vertical), respectively. Yousefi et al14
were unclear
about their method of measuring tongue protrusion. Heller
et al13
only had 5 patients in the conventional frenuloplasty
group (horizontal to vertical) and demonstrated a high risk
of bias in their research methods (Table 2).
Speech Outcomes
All studies used different methods for measuring postopera-
tive changes in speech (Table 6). Two ZF studies reported
speech outcomes assessed by speech language pathologists.
Yousefi et al14
reported a significantly higher reduction of
speech errors in the ZF group compared to the CF group.
Heller et al13
reported improvements in speech errors by an
‘‘order of magnitude’’ from baseline; however, no assessment
of statistical significance was provided. For CF, Messner and
Lalakea25
reported a significant improvement in parental-
reported speech problems, and Walls et al50
demonstrated a
significant difference in parental-reported speech outcomes
between treatment and no-treatment groups.
Anesthesia Requirements
Thirteen articles explicitly reported that frenotomy was
performed without anesthesia; 10 articles did not specify
whether anesthesia was used. Out of the articles evaluating
LF, Fiorotti et al22
used topical 10% lidocaine spray fol-
lowed by 1.8 mL of 2% lidocaine solution without a vaso-
constrictor, Ghaheri et al6
applied a topical anesthetic
Table 3. Maternal Nipple Pain Outcomes after Frenotomy Using the Visual Analog or Numeric Rating Scale.
Pain Scores (SD or IQR)
Authors Frenotomy N Prefrenotomy Postfrenotomy Mean Difference
Ghaheri et al6
Laser 58 4.1 (2.9) 1.4 (1.8) –2.7a
Ghaheri et al7
Laser 20 4.3 (2.8) 1.8 (2.1) –2.5a
Geddes et al37
Conventional 24 3.6 (3) 0.5 (1.2) –3.1a
Muldoon et al44
Conventional 89 5.6 (3.3) 2.7 (2.6) –2.9a
Dollberg et al34
Conventional 25 7.1 (1.9) 5.3 (2.2) –1.8a
Emond et al36
Conventional 53 3 (1-4.3)b
0 (0) –2 (–3 to –1)
No treatment 52 3 (2-6)b
0 (0-1) –2 (–3.5 to –0.6)
Ballard et al24
Conventional 123 6.9 (2.3) 1.2 (1.52) –5.7a
Berry et al31
Conventional 14 4.1 1.6 –2.5 (SD 1.9)
No treatment 14 4.2 2.9 –1.3 (SD 1.5)
Conventional 328 5 (3-7) 2 (0-4) –3a
Abbreviations: IQR, interquartile range; SD, standard deviation.
a
Statistically significant difference: P .05.
b
Earliest scores were only available for 5 days post frenotomy with an 8-week endpoint.
Table 4. Weighted Mean Differences of Prefrenectomy and Postfrenectomy Maternal Nipple Pain on a Visual Analog or Numeric Rating
Scale for Non–Randomized Controlled Trial Studies Only.
Frenotomy No. of Studies No. of Patients Weighted Mean Difference
Laser 2 78 –2.7
Conventional 4 564 –3.6

cream (EMLA), and Komori et al11
used a combination of
general (n = 7) and local (n = 8) anesthetic. For the articles
evaluating variations of the frenuloplasty, Ferrés-Amat et
al23
used a local anesthetic (articaine 4% with epinephrine
1:100,000), and both Heller et al13
and Yousefi et al14
per-
formed ZF under general anesthesia. For the articles evalu-
ating CF, only Ito et al,40
Ballard et al,24
and Messner and
Lalakea25
performed tongue-tie release under general
anesthesia. These patients were older children (age .3)
who underwent frenuloplasty.
Adverse Events
No serious adverse events were reported. Minor events are
summarized in Table 7.
Table 6. Summary of Speech Outcomes after Frenotomy.
Authors Frenotomy No. Speech Outcomes
Fiorotti et al22
Laser 15 Speech articulation changes in 10 (66.7%)
Heller et al13
Z-plasty and frenuloplasty 11 (ZF)
5 (F)
ZF group: 91% of patients showed at least 2 orders of improvement in
speech, 64% showed complete resolution of articulation errors, and
9% demonstrated no improvement in speech.
F group: 40% showed 1 order of improvement in speech, and 60% had
no change in articulation.
Ito et al40
Conventional 5 Consonant substitutions: Nineteen substitutions that were observed in
4 patients preoperatively. Decreased to 10 in 3 patients at 1 month, 7
in 3 patients at 3 to 4 months, and 1 in 1 patient at 1 to 2 years
postoperatively.
Omissions: Five were observed in 4 patients preoperatively. Decreased
to 3 in 3 patients at 1 month, 2 in 2 patients at 3 to 4 months, and 1
in 1 patient at 1 to 2 years postoperatively.
Distortions: Thirteen were observed in 5 patients preoperatively.
Decreased to 8 in 4 patients at 3 to 4 months but increased to 11 in
3 patients at 1 to 2 years postoperatively.
Conventional 30 Speech pathology evaluation: Preoperative articulation problems in 15
of 21 children. Articulation improved in 9, no change in 4 who had
normal speech preoperatively, and an ongoing articulation disorder in
2.
Parent perception of speech intelligibility (scale of 1 to 5): improved
from 3.4 to 4.2 (P .01).
Walls et al50
Conventional 71 Parental-reported speech on follow-up: Improvement in CF group (P
.0001).
Parent-reported difference between surgery and no-treatment group: P
= .3781.
Yousefi et al14
Z-plasty and conventional 25 (ZF)
25 (CF)
Postoperative questionnaires: Improvement in articulation and tongue
movement (P .05). Z-plasty had a greater effect on measures of
articulation and tongue movement (P .05).
Abbreviations: CF, conventional frenotomy; F, conventional frenuloplasty (horizontal to vertical); ZF, Z-plasty frenotomy.
Table 5. Summary of Outcomes Assessing Tongue Mobility.
Authors Frenotomy No. Tongue Movement Outcomes
Heller et al13
Z-plasty and frenuloplasty 11 (ZF)
5 (F)
Tongue protrusion: Mean (SD) increase of 36.2 (7.6) mm (P .0001) in
the ZF group and 13.2 (2.6) mm (P = .0003) in the F group
Conventional 30 Tongue protrusion: Mean increase of 11.8 mm postoperatively (P .01)
Tongue elevation: Mean increase of 16.8 mm postoperatively (P .01)
Yousefi et al14
Z-plasty and Conventional 25 (ZF)
25 (CF)
Hazelbaker score: Mean (SD) increase of 2.91 (0.302) mm (P .001) in
the ZF group and 2.10 (0.553) mm (P .001) in the CF group
Tongue protrusion: Mean (SD) increase of 17.56 (4.484) mm (P .001)
in the ZF group and 10.44 (3.787) mm (P .001) in the CF group
Abbreviations: CF, conventional frenotomy; F, conventional frenuloplasty (horizontal to vertical); SD, standard deviation; ZF, Z-plasty frenotomy.
Khan et al 11

Discussion
A systematic review of outcomes for different frenotomy
techniques used for treating ankyloglossia in the pediatric
population was conducted. More RCTs investigating CF
than ZF or LF were identified. In general, the RCTs were
low quality given the high level of heterogeneity in outcome
assessment methods and risk of bias. One RCT investigating
ZF was the only article using CF as a control group.14
All
LF studies did not include a control group.6,7,11,22
The
patient cohorts for ZF were older when compared to con-
ventional frenotomy methods.11,14,26
For this reason, ZF
studies emphasized speech problems as the major functional
indication for frenotomy. However, it is important to note
that the association between speech problems and ankylo-
glossia remains controversial.51
Differences in Frenotomy Procedures
LF was performed in 4 studies that met the inclusion cri-
teria. The largest study with 58 patients used a 1064-nm
InGaAsP semiconductor diode laser.6
Their procedure required
a topical anesthetic and pain control with acetaminophen
postoperatively in some cases.6
Komori et al11
used a CO2
laser with a wavelength of 10.6 mm. Seven of 15 patients
47% required suturing after resection.11
Fiorotti et al22
con-
ducted laser frenotomy with a CO2 laser coupled to Swiftlase
(scanner device) and no sutures were required. Procedures by
Komori et al11
and Fiorotti et al22
were done under general
and/or local anesthesia.
ZF was performed using several different methods. A 4-
flap variation was reported by Heller et al.13
The procedures
described by Choi et al26
combined Z-plasty with a partial
genioglossus myotomy to prevent shortening of the genio-
glossus muscle. Yousefi et al14
did not provide full details
of their operative procedure. Ferres-Amat et al23
performed
a frenotomy with rhomboid plasty and miotomy.
A major practical difference between CF, LF, and ZF is
the use of different types of anesthesia. CF was exclusively
performed without any general anesthesia in infants. CF
was only performed under general anesthesia in older chil-
dren or when frenuloplasty was indicated for speech-
related concerns. The majority of ZF procedures were
conducted under general anesthesia. Although researchers
used a variety of different anesthetic strategies for patients
treated with LF, the most common approach was a topical
anesthetic. The exposure of children to general anesthesia
is a disadvantage of the ZF technique and could limit its
applications.
Breastfeeding Outcomes
The majority of studies reported breastfeeding-related out-
comes postfrenotomy (Table 2). However, only few articles
investigating LF and ZF used validated assessment tools. A
recent LF study by Ghaheri et al6
used the BSES at 1 week
and 1 month postfrenotomy. They also measured maternal
nipple pain with a VAS. Although significant improvements
were reported, there was no control group. A recent Cochrane
review of RCTs comparing CF to sham procedures or controls
was only able to show an improvement with MNP on the
VAS.19
The only study comparing different techniques with
breastfeeding outcomes was an RCT conducted by Yousefi
et al14
comparing ZF with CF. On a 4-point scale (no
change, improved, good improved, and full resolution of
feeding problems), there was significant improvement in
maternal-reported breastfeeding following both ZF and CF.
Interestingly, the difference between the 2 interventions was
not significant.14
Table 7. Adverse Events during or after Frenotomy.
Authors Frenotomy No. Adverse Events
Argiris et al30
Conventional 46 In total, 24 patients (52%) had blood loss during operation; 3
patients had repeat procedures.
Berry et al31
Conventional 57 Three (5%) had bleeding at home after the procedure.
Dollberg et al34
Conventional 244 Seven (3%) reported worsening of breastfeeding difficulties.
Emond et al36
Conventional 53 Four of 99 (4%) patients had repeat procedures.
Frenectomy with rhomboid plasty 101 Postoperative complications in 7 (6%) of the participants (4
tongue bites, 1 hemorrhage, and 2 infections)
Griffiths38
Conventional 215 In total, 128 had an increased cry after division, 121 had
bleeding, and 4 had ulcers under the tongue.
Ghaheri et al6
Laser 58 Eight patients (3%) had a repeat procedure.
Hansen et al39
Conventional 44 Two patients had complications (1 bleeding, 1 in pain) and 2
patients had repeat procedures.
Mettias et al42
Conventional 36 Two patients (5.6%) were distressed, which was considered pain
from the procedure. One patient (2.8%) had mild bleeding on
the day of surgery. Ulceration was also reported in 1 patient
(2.8%).
Yousefi et al14
Z-plasty vs conventional 50 One minor hemorrhage

Overall, CF, LF, and ZF were all sound options for treat-
ing ankyloglossia that causes breastfeeding difficulties.
There was no evidence to suggest that one technique was
superior over others.
Tongue Movement and Speech Outcomes
The functional improvements associated with frenotomy
result from enhancement of tongue mobility. While func-
tional outcomes are the typical measure of treatment benefit,
improvements in tongue mobility may also be used to com-
pare surgical techniques. A validated assessment tool for
scoring tongue structure and function is the Hazelbaker tool,
which can be used to classify the severity of ankyloglossia.1
Three studies evaluated tongue movement or function post-
frenotomy but only 1 study used this tool (Table 5). Two
articles compared ZF vs CF or conventional frenuloplasty
(horizontal to vertical) and reported a significantly higher
increase in tongue protrusion for the ZF group.13,14
Both
articles used only univariate analysis to outline statistical
differences and 1 study included only 5 patients in the
horizontal-to-vertical frenuloplasty control group.14
All ZF
articles mentioned prevention of scar contracture as a bene-
fit of ZF over other techniques.13,14,26
However, our review
suggests that there is no high-quality evidence to conclude
the superiority of ZF over other techniques in improving
tongue mobility.
Speech was also a measured outcome for studies investi-
gating CF and LF. However, the controversy surrounding
the association of speech abnormalities with ankyloglossia,
heterogeneity of outcomes measured, and lack of statistical
comparison prevents any recommendations to be drawn
from these studies.
Surgical Complications and Operating Time
Surgical complications were not explicitly mentioned in all
studies. Therefore, a qualitative review was performed to
determine adverse events (Table 7). Weighted averages
were not computed for these data as the quantification meth-
ods varied in the articles. Overall, there is no compelling
evidence that the choice of frenotomy technique offers any
advantage in preventing surgical complications.
Operation time was not a measured outcome in any
study; however, it was mentioned in the methods section of
2 articles. Buryk et al27
performed CF in an average of 5
minutes. Fiorotti et al22
performed LF, which required 15 to
25 minutes to complete. While there is no clear advantage
of any technique with respect to operative parameters, a
beneficial future study would be to investigate overall cost-
effectiveness.
Nonoperative Management
The most common nonoperative management modalities
used in studies were lactation consultation and speech ther-
apy. There are limited studies in the current literature that
compare frenotomy with nonoperative management tech-
niques. Lactation consultation and other forms of profes-
sional support for breastfeeding difficulties were most
commonly used during preoperative assessment only. A
range of health care professionals were reported to be
involved in supporting patients through breastfeeding diffi-
culties, including infant coordinators, lactation consultants,
midwives, and nurses. In some studies, patients were
referred by lactation consultants to otolaryngologists for
consideration of frenotomy. This would suggest that nono-
perative measures may have been exhausted by the lactation
consultants. Lactation consultation was provided as a con-
trol group in 1 study by Hogan et al,28
who offered patients a
lactation consultation for 48 hours. Patients who did not
improve after 48 hours (27 of 28 patients) were offered a fre-
notomy. From an ethical standpoint, it is important to note that
researchers in studies comparing frenotomy and nonoperative
treatments may be ethically obligated to offer surgical correc-
tion to nonoperative patients after a preestablished time frame.
This provides challenges for randomization and comparing
long-term outcomes between frenotomy and nonoperative
treatments. However, the inclusion of a short-term nonopera-
tive control group as demonstrated by Hogan et al28
or patients
who willingly refused surgery would help outline the differ-
ences between frenotomy and nonoperative treatments.
Study Limitations
While many studies have investigated the utility of different
frenotomy techniques, there are several methodological gaps
preventing a meta-analysis. Only 1 RCT used CF as a com-
parison group. Moreover, only 4 non-RCT studies compared
with a control group. Postsurgical outcomes were frequently
reported using subjective measures. Outcome assessments
were largely heterogeneous, and validated assessment tools
were used in a minority of studies. There were several cases
where patient selection criteria were either not mentioned or
not determined using a validated scoring tool such as the
Hazelbaker tool. This led to considerable selection bias.
Some studies also provided lactation consultation or speech
therapy to patients after frenotomy. In these cases, the bene-
fit of frenotomy could be biased by benefit achieved from
nonoperative treatments. Moreover, statistical calculation of
mean differences with respective confidence intervals was
not consistently available.
Studies in this review were limited to a retrospective
methodology. The current review was restricted to English-
language articles for reasons of accessibility. This review
may be associated with publication bias for those institu-
tions with positive outcomes.
Recommendations
Our review has identified several methodological shortcom-
ings of current research seeking to demonstrate the superiority
of different frenotomy techniques. In addition, our review out-
lines the importance of investigating nonoperative techniques
to manage issues associated with ankyloglossia. Based on the
identified gaps, we have the following recommendations:
1. Control groups: The inclusion of a control group
where patients are treated with CF is critical
Khan et al 13

when comparing outcomes with other frenotomy
techniques. Furthermore, a nonoperative control
group of parents who refuse surgery for ankylo-
glossia would mitigate a potential confirmation
bias among parents who opted for frenotomy. In
particular, a control group that includes compre-
hensive supportive treatment, such as consistent
lactation consultation and speech therapy, would
be extremely valuable to clinicians considering the
paucity of literature assessing nonoperative treat-
ments. These studies would also help determine if
there are any benefits of using supportive treat-
ments in addition to frenotomy for the management
of ankyloglossia in young children.
2. Outcome assessment: The investigation of func-
tional outcomes should use validated tools to limit
heterogeneity and bias. This is a major limitation
in the current literature.
3. Procedural considerations: A controlled comparison
of surgical parameters such as surgery time, com-
plications, scar contracture, and need for repeat
surgeries should be assessed when investigating
different frenotomy techniques. Furthermore, the
use of general anesthesia is a critical factor when
comparing frenotomy techniques as the majority of
children treated for ankyloglossia are infants. In
addition to preventing unnecessary exposure to
general anesthesia in infants, there are financial
burdens associated with using an operating room to
perform frenotomy, when an equally effective man-
agement option is available at the bedside.
4. Patient allocation and randomization: The number
of patients should be equally distributed between
intervention groups when possible. Patients should
also be randomized to intervention groups for
RCTs with careful attention to blinding and alloca-
tion concealment.
5. Treatment effect: For each outcome measurement,
a treatment effect should be reported as a mean dif-
ference with an appropriate confidence interval.
Conclusions
The current evidence outlines the utility of CF, LF, and ZF
for treating symptomatic ankyloglossia in the pediatric pop-
ulation. All frenotomy techniques were found to be safe and
effective procedures for dividing the lingual frenulum.
Articles substantiating the benefits of CF were generally of
a higher quality compared to other frenotomy methods. ZF
was performed on older children with speech-related diffi-
culties, which remain a controversial indication for surgical
correction. CF was more commonly performed without
anesthetic when compared to LF and ZF. The role of sup-
portive treatment by health care professionals is an area of
research that is limited in the current literature. There was
no enhanced benefit of ZF or LF for treating ankyloglossia
in the pediatric population when compared to CF.
Author Contributions
Usman Khan, data collection, management, analysis and interpre-
tation, manuscript writing, manuscript editing, manuscript revision,
final approval for submission, accountable for all aspects of the
work in ensuring that questions related to the accuracy or integrity
of any part of the work are appropriately investigated and resolved;
Jake MacPherson, data collection, management, analysis and
interpretation, manuscript editing, manuscript revision, final
approval for submission, accountable for all aspects of the work in
ensuring that questions related to the accuracy or integrity of any
part of the work are appropriately investigated and resolved;
Michael Bezuhly, study design, data analysis and interpretation,
manuscript editing, manuscript revision, final approval for submis-
sion, accountable for all aspects of the work in ensuring that ques-
tions related to the accuracy or integrity of any part of the work
are appropriately investigated and resolved; Paul Hong, study
design, data analysis and interpretation, manuscript writing, manu-
script editing, manuscript revision, final approval for submission,
accountable for all aspects of the work in ensuring that questions
related to the accuracy or integrity of any part of the work are
appropriately investigated and resolved.
Disclosures
Competing interests: None.
Sponsorships: None.
Funding source: None.
Supplemental Material
Additional supporting information is available in the online version
of the article.
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