International Journal of Pediatric Otorhinolaryngology 138 (2020) 110356

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International Journal of Pediatric Otorhinolaryngology

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

Review Article

Major complications after tongue-tie release: A case report and

systematic review
Paola Solis-Pazmino a, b, Grace S. Kim a, Eddy Lincango-Naranjo b, c, Larry Prokop b,
Oscar J. Ponce b, d, Mai Thy Truong a, *
a
Department of Otolaryngology–Head and Neck Surgery, Stanford University, Stanford, CA, USA
b
Knowledge and Evaluation Research Unit, Mayo Clinic, Rochester, MN, USA
c
Facultad de Ciencias Medicas, Universidad Central El Ecuador, Quito, Ecuador
d
Unidad de Conocimiento y Evidencia, Universidad Peruana Cayetano Heredia, Lima, Peru

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

Keywords: Introduction: The diagnosis of ankyloglossia, or tongue-tie, and the number of frenotomies performed has
Tongue-tie increased over 10-fold from 1997 to 2012 in the United States. The sharpest increase has been in neonates. For
Ankyloglossia parents considering frenotomy for their breastfeeding newborn, there is controversy surrounding the evaluation
Tongue-tie release
of tongue-tie and the benefit of a frenotomy. Complications from tongue-tie procedures are thought to be low,
Frenotomy
Frenuloplasty
though it is not well reported nor studied.
Lingual frenulum Objectives: The aim of this study is to describe a case of a sublingual mucocele after laser frenotomy in a neonate
Major complications with tongue-tie and to investigate major complications reported after tongue-tie release in pediatric patients
Case report through a systematic review of the literature.
Systematic review Case report: We present a 6-week-old female who underwent a laser frenotomy procedure performed by a dentist
who presented with a new cyst under her tongue.
Material and methods: A systematic literature search of articles published from 1965 to April 2020 was conducted
in Ovid MEDLINE(R), Ovid EMBASE, and Scopus. Citations were uploaded into a systematic review software
program (DistillerSR, Ottawa, ON, Canada), followed by full text screening.
Results: 47 major complications were reported in 34 patients, including our patient. Most of the cases were
located in the United States and Europe. The most frequent indications for the procedure were breastfeeding
problems (n = 18) and speech impediment (n = 4). The procedure was performed by dentists (n = 6), lactation
consultants (n = 5), and otolaryngologists (n = 4). The bulk of the major complications after frenotomy included
poor feeding (n = 7), hypovolemic shock (n = 4), apnea (n = 4), acute airway obstruction (n = 4), and Ludwig
angina (n = 2).
Conclusions: Reporting of complications after frenotomy is lacking. Risks to neonates may be different than risks
to older children and adults. Practitioners across different specialties should be monitoring and studying this
more rigorously to better guide patients and families on the risks and benefits of this procedure.

1. Introduction procedures have been reported to improve difficulties with breastfeed­

Ankyloglossia, or tongue-tie, is defined as limited tongue mobility
due to a shortened lingual frenulum. The evaluation of ankyloglossia
and the benefit of a frenotomy to release tongue tie are controversial
topics. The diagnosis of ankyloglossia and the number of frenotomies
performed has increased over 10-fold from 1997 to 2012 in the United
States [1]. The sharpest increase has been in neonates; frenotomy
ing, especially when there is maternal pain with breastfeeding [2]. A
restrictive lingual frenulum has long been thought to affect a child’s
speech and articulation, impede tongue mobility skills for social in­
teractions (such as licking or kissing), and effect the ability of the tongue
to sweep and keep the teeth clean [3]. However, the impact of ankylo­
glossia has recently been extended to include neonatal reflux, colic and
childhood and adult sleep apnea, though the evidence is poor and mostly

* Corresponding author.
E-mail address: mttruong@stanford.edu (M.T. Truong).

https://doi.org/10.1016/j.ijporl.2020.110356
Received 16 May 2020; Received in revised form 28 August 2020; Accepted 30 August 2020
Available online 1 September 2020
0165-5876/© 2020 Elsevier B.V. All rights reserved.
P. Solis-Pazmino et al.
reported on web-based platforms and social media.
The incidence rate of ankyloglossia is reported to range from 0.1 to
10.7% with a 2–3:1 male predominance [4,5]. The diagnosis of
tongue-tie is complicated by definitions of anterior and posterior tongue
tie, which can be difficult to differentiate. “Anterior tongue-tie” is
defined by some as a lingual frenulum that extends to or near the tip of
the tongue. The definition of a “posterior tongue-tie” for some is a
frenulum that inserts in to the posterior portion of the tongue, whereas
others think of it as a submucosal tethering of the tongue, though this
definition may vary from provider to provider as panels have failed to
show consensus on the definition [3]. One observational study showed
that up to 59% of healthy, asymptomatic newborns fit at least one
criteria for a posterior tongue-tie [6]. The true incidence of posterior
tongue-tie, especially when there are associated symptoms, is unknown
[7].
The main surgical procedures treating ankyloglossia are frenotomy
(clipping of the frenulum) and frenuloplasty (operation that lengthens
the frenulum), though the difference in reporting may be gestalt [8].
Frenotomy, which is the procedure more often done in neonates, can be
performed with scissors, cautery, electrocautery, or a laser. For simpli­
fication purposes of this paper, we will refer to tongue-tie procedures as
frenotomies. Surgeons, neonatologists, pediatricians, dentists, mid­
wives, and lactation consultants (typically licensed providers counseling
about breastfeeding techniques and issues) perform frenotomies, and
techniques vary vastly. Complications from ankyloglossia procedures
are thought to be low, though it is not well reported nor studied. Due to
the differing disciplines performing frenotomies and the range of tech­
niques, tracking outcomes and complication rates is difficult.
The benefits after tongue-tie release remain controversial. Some
authors reported that frenotomy provides meaningful improvement in
breastfeeding difficulties and speech problems [9–11]. Moreover, in
2016 the Canadian Agency for Drugs and Technologies in Health
(CADTH) supported the benefit of the frenotomy in short term breast­
feeding outcomes [12]. However, the strength of the evidence sup­
porting these outcomes is low to insufficient.
Most of the studies explicitly reported a low complication rate. Some
of the mild complications mentioned include minor bleeding, pain,
weight loss, and scarring of the surgical site [9]. However, the lack of
consistency in reporting major complication along with methodological
shortcomings of the existing studies limit the certainty of frenotomy
complication rates.
The objectives of this study is to present a case of a neonatal patient
who developed a sublingual mucocele after laser frenotomy and to
conduct a systematic review evaluating the major complications re­
ported in children after a frenotomy is performed.
2. Case report
A 6-week-old female presented to clinic with a history of breast-
feeding difficulties. The patient was diagnosed with tongue-tie by a
local lactation consultant and underwent lingual frenotomy with a laser
by a dentist one week prior to presentation. The parents reported the
infant tolerated the procedure well, had 5 days of pain after the pro­
cedure, and showed improved latch post-procedure. However, they
noted persistent breast-feeding issues and the development of a cyst
under her tongue. The physical examination revealed a 1.2 cm lesion in
the floor of mouth to the right of the midline. The lesion was clinically
consistent with a mucocele and had never ruptured or bled. At two-week
follow up, the infant continued to have breast-feeding concerns and was
supplemented with pumped breast milk from a bottle. The mucocele had
decreased to 5 mm in size, and the parents wished for no surgical
intervention.
3. Material and methods
This report followed the CARE Guideline checklist and the Preferred
2
International Journal of Pediatric Otorhinolaryngology 138 (2020) 110356
Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA)
[13,14].
3.1. Eligibility criteria
For the systematic review, we included case reports and case series
reporting patients younger than 18 years diagnosed with tongue-tie,
who underwent frenotomy to release the lingual frenulum with any
type of major surgical complication. We defined major complications as
requiring medical attention and/or further intervention. This rationale
is supported by two previous systematic reviews where minor bleeding
was defined as a self-limited complication after frenotomy [9,15]. No
restrictions were made based on follow-up period or type of publication.
3.2. Data sources and searches
The systematic review of the literature was performed using the
search terms tongue-tie, ankyloglossia, complication, frenotomy, fre­
nuloplasty, and pediatric from 1965 to April 2020. The databases
included Ovid MEDLINE(R) and Epub Ahead of Print, In-Process &
Other Non-Indexed Citations, and Daily, Ovid EMBASE, Ovid Cochrane
Central Register of Controlled Trials, Ovid Cochrane Database of Sys­
tematic Reviews, and Scopus. The search was restricted to the English
Language (Supplementary 1).
3.3. Study selection
The search strategy results were uploaded into an online software
program (DistillerSR, Evidence Partners, Ottawa, Canada) [16]. The
study selection process was comprised of titles, abstract screening, and
the review of full-text articles. All reports in both steps were reviewed in
an independently and duplicate manner by two authors (P⋅S–P, E.L-N).
Before initiating the processes of study selection, a pilot with five articles
was done so that all reviewers could assess the clarity of the eligibility
criteria and the consistency of their decisions (include or exclude).
Studies that were included by at least one author in the title and abstract
screening proceeded to full-text screening. Disagreements were resolved
in full-text screening, which was reached by consensus. The full-text
screening agreement was substantial (Cohen’s kappa = 0.83).
3.4. Data collection
Data from included studies was extracted independently and in
duplicate in a standardized form by two reviewers (P⋅S–P, E.L-N). A pilot
with three studies was performed for data extraction. The following
information was extracted: 1) general characteristics (first author last
name, publication date, country were the case/s were reported), 2)
characteristics of study participants (age, sex, reason for surgical pro­
cedure); 3) type of surgical procedure (surgical technique, surgical in­
struments, time between frenuloplasty and complication, the person
who performed the procedure); and 4) type of complication and
etiology.
3.5. Risk of bias in individual studies
The risk of bias of case reports and case series was measured with a
tool proposed by Murad et al. [17]. This instrument is composed of eight
questions assessing four domains: (1) selection; (2) ascertainment; (3)
causality; and (4) reporting. Disagreements were resolved by consensus
between two reviewers (P⋅S–P, E.L-N). When two or less questions
applied to the study, it was considered to be at low risk of bias. Studies
were considered at unclear risk when three questions applied. Those
with four or more were considered at high risk of bias (Supplementary
2).
P. Solis-Pazmino et al.
Fig. 1. Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) flow diagram of studies selection process. An evidence-based set of items used
to screen article titles, abstracts and full-texts to assess for eligibility in our systematic review.
4. Results
The search strategy retrieved 456 references and 15 studies were
included (Fig. 1). Eleven were case reports and four were case series. We
identified 34 patients, including our patient, from the 15 studies. There
were 22 males and 12 females. Their age ranged from one day old to 13
years old. The studies originated from the United States (n = 6), Europe
(n = 4), Turkey (n = 1), Nigeria (n = 1), Brazil (n = 1), and New Zealand
(n = 2). The overall risk of bias was judged to be low in 6 studies, unclear
in 4 studies, and high in 5 studies (S1 Appendix).
The most common reasons for undergoing tongue-tie surgery were
infant breastfeeding problems (n = 18), speech impediment (n = 4),
maternal breast/nipple pain (n = 3), and weight lost (n = 3). In 14
patients, the reason was unknown. The providers who performed the
procedure were dentists (n = 6), lactation consultants (n = 5), otolar­
yngologists (n = 4), general practitioners (n = 2), pediatric surgeon (n =
3
International Journal of Pediatric Otorhinolaryngology 138 (2020) 110356
1), pediatrician (n = 1), and oral surgeon (n = 1) while it was unknown
in 14 cases. The techniques used were frenotomy (n = 5), frenuloplasty
(n = 3), frenulectomy (n = 9), a “single tongue release” (n = 1), and
unknown (n = 16). The incision of the lingual frenulum was performed
with a scissor (n = 4), razor blade (n = 1), laser (n = 1), and unknown (n
= 28) (Table 1).
A total of 47 major complications occurred in 34 patients. Table 2
summarizes the complications identified, including patients who pre­
sented with more than one complication. The most common complica­
tions were poor feeding (n = 7) followed by bleeding (n = 3) and
complications related to bleeding such as hypovolemic shock (n = 4)
and infected hematoma (n = 1). Other major complications included
involvement of the airway including acute airway obstruction (n = 4),
Ludwig’s angina (n = 1), and negative pressure pulmonary edema (n =
1). Post-procedure infections were reported as infected hematoma (n =
1), wound infection (n = 1), infected cyst (n = 1) and submandibular
P. Solis-Pazmino et al. International Journal of Pediatric Otorhinolaryngology 138 (2020) 110356

Table 1
Demographic details of reports. NR: not reported.
Country Age of patient (n) Gender Indication for Procedure performed Proceduralist
procedure

Case Reports
Walsh, 1995 [28] Ireland 7 years 1 Male NR Frenotomy NR
Davidson, 2004 USA 4 years 1 Female Speech problems Frenuloplasty NR
[29]
Lin, 2009 [30] USA 13 years 1 Female Speech impediment Frenuloplasty Oral surgeon
Santos, 2012 [31] Brazil 12 years 1 Male Speech impediment Frenectomy NR
Isaiah, 2013 [32] USA 2 years 1 Male Speech impediment Frenulectomy NR
Sirinoglu, 2013 Turkey 3 years 1 Male NR Frenuloplasty NR
[33]
Reid, 2014 [34] New 9 weeks 1 Female Breastfeeding Frenulectomy Lactation consultant
Zealand problems
Maciag, 2016 USA 13 days 1 Male NR Frenulectomy NR
[35]
Stokes, 2017 [36] England 4 years 1 Female NR Frenulectomy NR
Reinholdt, 2019 Denmark 12 days 1 Male Breastfeeding Frenulectomy Otolaryngologist surgeon
[37] problems
Bhalla, 2019 [38] England 4 months 1 Male NR Single tongue tie NR
release
Solis, 2020 USA 2 months 1 Female Breastfeeding Frenotomy Dentist
problems
Case series
Opara, 2012 [25] Nigeria 1 day 2 Male NR Frenotomy Lactation consultant
3 days Male Breastfeeding Frenotomy Lactation consultant
problems
Genther, 2015 USA 2 days 2 Male NR Frenulectomy NR
[39] 7 days Male Breastfeeding Frenulectomy Otolaryngologist surgeon
problems
Tracy, 2017 [40] USA 13 months 2 Female NR Frenulectomy NR
4 years Male NR Frenulectomy NR
Hale, 2019 [20] New 45 days * (6–206 10 Males Breastfeeding NR Dentists (n = 5)
Zealand days) 6 Females Problems (n = 13) General practitioners (n = 2) Lactation
consultants (n = 2)
Breast/nipple pain (n Otolaryngologist surgeons (n = 2)
= 3)
Weight loss (n = 3) Pediatrician (n = 1)
Unknown (n = 2) Pediatric surgeon (n = 1)
Other **(n = 3) Unknown (n = 3)

*Ave age of 16 patients.

**Other reported as unsettledness and jaundice, fussy, windy and slow weight gain.

abscess (n = 3). Finally, damage to local structures and salivary glands
during frenotomy resulted in complications reported as mucoceles (n =
3), massive submandibular edema from obliteration of Wharton’s duct
(n = 1), excess scarring (n = 2), and formation of a floor of mouth cyst (n
= 1). The time between the surgery and the complication ranged from
30 min to 3 weeks.
5. Discussion
Several studies have shown tongue-tie release as a safe and successful
procedure with little to no complications [18,19]. For instance, a prior
systematic review showed 17 studies reporting no significant harm after
a tongue-tie release [9]; however, this may be in part due to the study
design where the primary outcome was to identify breastfeeding effec­
tiveness as opposed to complications. In addition, the review queried
studies completed up to 2014, which would not have included more
recent studies that have identified major complications following fre­
notomies. The annual incidence of complications after frenotomy in
infants less than 1 year old studied prospectively in New Zealand was
13.9 of 100,000 infants [20], and an estimated complication rate of 1%
for moderate to severe complications.
In our study, we identify complications after frenotomy reported
from several different disciplines and performed with different tech­
niques. Though the true incidence of complications after frenotomy may
be low, we wonder if complications are not followed nor reported well
across different specialties. Though it would be very useful information,
it was not clear if all these patients on our review had the diagnosis of
anterior tongue tie, posterior tongue tie, or if they were undergoing a
revision. Parents may report to their primary doctors and pediatricians
with post-procedure complications as opposed to the provider who
performed the frenotomy, limiting ability to track complications. In
addition, pain may be difficult to assess in neonates, which is also
complicated by the fact that there are concerns for giving even over-the-
counter analgesics such as acetaminophen in infancy. There is also a lack
of a standardized training or licensing protocol for providers doing
frenotomies, which can make comparisons between providers difficult.
There is a recent trend towards laser frenotomy for a myriad of
reason, including shorter operative time, concurrent tissue cauterization
and sterilization, hemostasis, less local anesthetic requirement, uniform
depth of incision can be set, and improved visualization due to lack of
interposed instruments [21–23]. However, tissue studies have shown
that mucosal incisions made in porcine tongues with the scalpel showed
less histological tissue injury, thermal damage and inflammation
compared to laser, electrosurgery, and constant-voltage electrocautery
[24]. The authors speculate whether complications after frenotomy may
be higher as there is heightened concern for posterior tongue-tie or for
incomplete release of the frenulum, resulting in more aggressive fre­
notomies being performed in clinic settings. This may result in a higher
risk for bleeding with techniques that do not use heat (such as with
scissors or scalpel) or more pain and risk of injury to local structures
with hotter techniques (such as those done with a laser). Of note, there
was one study from a developing country that reported bleeding and
hypovolemic shock as a complication after frenotomy with a razor blade
[25]. While there are only a few studies specifically looking at

4
P. Solis-Pazmino et al. International Journal of Pediatric Otorhinolaryngology 138 (2020) 110356

Table 2 Table 2 (continued )

Major complications. Major Time between Surgical Proposed cause
Major Time between Surgical Proposed cause complications frenuloplasty instrument of
complications frenuloplasty instrument of and used complication
and used complication complication after
complication after frenotomy
frenotomy
Severe
Case reports hypernatremia
Walsh, Acute airway 30 min NR Impaired (n = 1)
1995 obstruction function of the
NR: not-reported.
[28] genioglossus
muscle *Average.
Davidson, Negative 1h NR NR **Seven patients also had more than one complication, but this data was not
2004 pressure specified in the Hale el al. Study.
[29] pulmonary
edema
frenotomy complication rates in developing countries, limited resources
Lin, 2009 Ludwig’s angina NR NR Abundant oral
[30] flora and training may contribute to higher rates of complications [26].
Santos, Mucocele 3 weeks NR NR In 2019, a study by Caloway et al. investigated parental choice for
2012 proceeding with frenotomy after a multidisciplinary feeding consulta­
[31]
tion and examination, where the mothers learned techniques to address
Isaiah, Infected 2 days Scissors Alpha- and
2013 hematoma gamma
feeding difficulties. Surprisingly, after this intervention 63% did not
[32] hemolytic choose to undergo surgical procedures for tongue-tie release [27]. As
streptococci parents and providers attempt to weigh the risks and benefits prior to a
Sirinoglu, Subacute 2h NR Obliteration of frenotomy, it will be important to understand potential limitations to
2013 massive edema the Wharton
our understanding of the true complication rate of frenotomies.
[33] of the duct
submandibular
region 6. Conclusions
Reid, 2014 Wound 12 h Scissors Staphylococcus
[34] infection aureus
Complications after frenotomy procedures include poor feeding,
Maciag, Ludwig’s angina 2 days NR Methicillin-
2016 resistant
bleeding, infection, risk to the airway and damage to local structures in
[35] Staphylococcus the mouth. As more providers across different disciplines are performing
aureus this procedure with differing techniques, the true complication rate is
Stokes, Mucocele NR NR NR unknown. Ultimately, the lack of consistent reporting of provider details
2017
or technique used limit our ability to determine a higher complication
[36]
Reinholdt, Submandibular 3 days Scissors Staphylococcus risk technique. Therefore, practitioners across specialties will need to
2019 abscess aureus monitor and study frenotomies and complications that arise more
[37] rigorously to better counsel patients and families on the risks and ben­
Bhalla, Acute airway 2 weeks NR Abscess in a
efits of the procedure.
2019 obstruction cyst formed
[38] after
frenotomy Funding
Solis, 2020 Mucocele NR Laser NR
Case series This research did not receive any specific grant from funding
Opara, Hypovolemic 2h NR Postoperative
2012 shock bleeding
agencies in the public, commercial, or not-for-profit sectors.
[25] Hypovolemic 1h Razor blade Postoperative
shock bleeding Declaration of competing interest
Genther, Acute airway 1 day NR Glossoptosis
2015 obstruction
[39] Acute airway 6 days NR Glossoptosis
None.
obstruction
Tracy, Hypovolemic 19 h NR Postoperative Acknowledgments
2017 shock bleeding
[40] Hypovolemic 8 days Scissors Postoperative
The Author Paola Solis is a research student funded by Stanford
shock bleeding
Hale, 2019 Poor feeding (n 3 days* NR NR University..
[20]** = 7) (0–15days
Apnea (n = 4) range) Appendix A. Supplementary data
Pain (n = 3)
Bleeding (n = 3)
Weight loss (n Supplementary data to this article can be found online at https://doi.
= 3) org/10.1016/j.ijporl.2020.110356.
Anemia (n = 2) All Authors have no Conflict of Interest and no disclosures.
Excess scarring
(n = 2)
Unsettledness
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(n = 1)
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