Clinical Review & Education

JAMA Otolaryngology–Head & Neck Surgery | Review

Diagnosis and Treatment of Ankyloglossia in Newborns

and Infants
A Review
Jonathan Walsh, MD; David Tunkel, MD

IMPORTANCE The influence of tongue tie, or ankyloglossia, on breastfeeding is the subject of

growing debate. Restriction of tongue mobility from the frenulum varies greatly among
newborns and infants (hereinafter referred to as infants). Controversies about whether an
infant has ankyloglossia and which infants need treatment are evident with wide variations in
medical practice and a lack of high-quality clinical studies that provide guidance.

OBSERVATIONS Diagnosis and management of ankyloglossia in infants can be a source of

confusion and frustration for clinicians and families. Frenotomy is a low-risk procedure that is
likely to be beneficial with careful patient selection, but the natural history of untreated
ankyloglossia is not well documented. The variability in presentation and treatment
outcomes of ankyloglossia indicate that the complexity of infant feeding and tongue
development is not fully encapsulated in a simplistic ankyloglossia etiologic framework.

CONCLUSIONS AND RELEVANCE Consistent terminology with emphasis on symptomatic

ankyloglossia and a uniform grading system, such as the Hazelbaker Assessment Tool for Author Affiliations: Department of
Otolaryngology–Head and Neck
Lingual Frenulum Function and Coryllos grading, are needed to improve the quality of Surgery, Johns Hopkins School of
research in the future. The ability to make definitive practice guidelines is limited with our Medicine, Baltimore, Maryland.
current understanding of ankyloglossia. Additional research is needed to better understand Corresponding Author: Jonathan
the complexity of infant feeding and the role of ankyloglossia Walsh, MD, Department of
Otolaryngology–Head and Neck
Surgery, Johns Hopkins School of
JAMA Otolaryngol Head Neck Surg. doi:10.1001/jamaoto.2017.0948 Medicine, 601 N Caroline St,
Published online July 13, 2017. Baltimore, MD 21287 (jwalsh31@jhmi
.edu).

T
he influence of ankyloglossia on breastfeeding and articula- Definition
tion is widely discussed and controversial. Diagnosis and sur- Traditionally, ankyloglossia has been described in terms of frenu-
gical treatment appear to have increased during the past few lum attachment at or near the tongue tip. Recently, emphasis has
decades, even without widely accepted diagnostic criteria and treat- shifted from definitions based on anatomy of frenulum attach-
ment indications. Walsh et al1 recently demonstrated large increases ment alone to a more functional focus on the symptoms caused by
in diagnosis and treatment by analyzing a national inpatient database the frenulum. The lingual frenulum, as defined by the International
sample. More newborns and infants (hereinafter referred to as infants) Affiliation of Tongue-Tie Professionals, is “a remnant of tissue in the
are being diagnosed, and many infants with ankyloglossia and pre- midline between the undersurface of the tongue and the floor of the
sumed associated breastfeeding difficulties are being diagnosed and mouth. When it interferes with normal tongue function it is called
treatedinthefirstfewdaysoflife.Similarincreaseswerenotedinalarge ‘symptomatic tongue-tie’ or ‘symptomatic ankyloglossia.’ ”4
sample of infants in British Columbia.2 Increased awareness of the in- No standard definition of ankyloglossia exists. However, ante-
fluence of ankyloglossia on breastfeeding and increased national and rior ankyloglossia, or classic ankyloglossia, is often defined as frenu-
global initiatives for breastfeeding support are likely to be contributors lum attachments at or near the tongue tip. The frenulum attach-
to this increase in diagnosis and treatment. ment at the tongue tip limits tongue mobility and protrusion. Inability
Accurate diagnosis requires a comprehensive understanding of to protrude the tongue beyond the lip vermillion border has been
the anatomical range of normal and abnormal tongue movement, used as an examination finding indicative of ankyloglossia.
lip, nasal airway, and mandible size and neonatal swallow function. Posterior ankyloglossia is symptomatic ankyloglossia with frenu-
The opinions of well-meaning clinicians from a variety of special- lum attachments at the middle to posterior aspect of the undersur-
ties, including otolaryngology, and patient and maternal support ad- face of the tongue. The frenulum can be short, thickened, or even
vocacy groups differ widely about the influence of ankyloglossia and submucosal in some cases but can still restrict tongue mobility. The
the utility of surgery.3 This study will provide a comprehensive re- classic ankyloglossia diagnostic criteria, in which the frenulum at-
view of neonatal ankyloglossia to aid the otolaryngologist in their taches at or near the tip of the tongue, did not incorporate poste-
understanding and management of ankyloglossia. rior ankyloglossia. Published studies on posterior ankyloglossia5-8

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 Clinical Review & Education Review Diagnosis and Treatment of Ankyloglossia in Newborns and Infants

Table 1. Common Grading Systems for Ankyloglossia

Grading System (Source) Anatomical Classification Criteria Posterior Ankyloglossia Classification

Coryllos system (American Academy of Pediatrics Type 1: Attachment of frenulum to the tongue tip, Consists of types 3 and 4 with functional
Section on Breastfeeding,26 2004) usually in front of the alveolar ridge impairment
Type 2: 2- 4 mm behind the tongue tip and on or just
behind the alveolar ridge
Type 3: Attachment to the midtongue and middle of
the floor of mouth
Type 4: Against the base of the tongue
Kotlow system (Kotlow,27 1999) Normal: >16-mm free tongue length Consists of normal and class I with functional
Class I (mild): 12 to 16–mm free tongue length impairment
Class II (moderate): 8 to 11–mm free tongue length
Class III (severe): 3 to 7–mm free tongue length
Class IV (complete): <3-mm free tongue length
Kotlow system revised (Kotlow,28 2011) Class I: 0 to 3–mm attachment from the tongue tip Consists of classes III and IV with functional
Class II: 4 to 6–mm attachment from the tongue tip impairment
Class III: 7 to 9–mm attachment from the tongue tip
Class IV: 10 to 12–mm or submucosal attachment
from the tongue tip
Tongue elevation (Lalakea and Messner,24 2003; Normal: >23 mm NA
Williams and Waldron,29 1985; Notestine,30 1990; Mild: 17-22 mm
and Ruffoli et al,31 2005) Moderate: 4-16 mm
Severe: ≤3 mm
Tongue protrusion (Lalakea and Messner,24 2003; Normal: 20-25 mm NA
Messner and Lalakea,25 2002) Ankyloglossia: <15 mm

Abbreviation: NA, not applicable.

emphasize the concept of symptomatic ankyloglossia. The diagno- failed to incorporate the perhaps greater prevalence of sympto-
sis of posterior ankyloglossia remains controversial. Some matic ankyloglossia. Only in the past decade has posterior ankylo-
investigators9 believe that posterior ankyloglossia can be a normal glossia been more frequently discussed in the literature.6 Common
frenulum attachment and that described feeding difficulties are at- grading systems are shown in Table 1. To highlight the complexity
tributable to other factors. In addition, owing to more recent changes of defining true prevalence, 1 study26 analyzed 200 healthy infants
in the definition of ankyloglossia, posterior ankyloglossia was not in- by using the Coryllos grading system (Table 1), which includes pos-
cluded in studies on the prevalence, presentation, and treatment of terior ankyloglossia criteria. Breastfeeding difficulties were not pre-
ankyloglossia. dicted by frenulum grading in this study.26,32 Other factors that ap-
Some tongue ties are symptomatic, and others are incidental pear to affect diagnosis may be socioeconomic status, region of the
findings without any feeding or speech symptoms. In the context country, first-time motherhood, and infant birth weight.1,2
of other anatomical or functional problems that may affect feed-
ing, which frenulums are truly symptomatic may be difficult to de- Embryology
termine. For example, maxillary lip frenulum tethering is reported Tongue development includes contributions from each of the first
to affect successful breastfeeding, and concurrent lip tie and anky- through fourth branchial arches (Figure 1). From the 4th through the
loglossia can complicate the diagnosis.10 Other factors that affect 10th weeks of gestation, the anterior two-thirds of the tongue is
breastfeeding can be maternal experience, maternal milk produc- formed predominately by 2 lateral lingual proliferations of the first
tion, or breast and nipple anatomy. Extension of expertise in neo- branchial arches, with minor contributions from the second bran-
natal swallowing and breastfeeding across multiple specialties, such chial arch. The third and a small portion of the fourth arch contrib-
as lactation consultants, speech pathologists, nurses, and physi- ute proliferations to form the posterior one-third of the tongue.34
cians, offers a challenge for diagnosis and management. Coordi- The tongue receives complex innervation from the 5th, 7th, 9th,
nated multidisciplinary evaluation is difficult to obtain, especially in 10th, and 12th cranial nerves. The intrinsic muscles of the tongue form
an expedited time frame within the first few weeks after birth. from migration of occipital somites along with the 12th cranial nerve.
Tongue frenulum attachment and feeding difficulty vary greatly in The early tongue is initially only prominences of the floor of the
severity of tethering and of symptoms. Clinicians likely default to mouth and developing mandible. As the lingual prominence en-
treating ankyloglossia despite uncertainty regarding whether the larges with primarily lingual muscular growth, the linguogingival sulci
frenulum is playing a role in the feeding difficulty. deepen to define the mobile tongue.34-36 Although no exact em-
bryologic cause of ankyloglossia is known, possible causes may be
Prevalence a combination of incomplete apoptosis anteriomedially of the lin-
The prevalence of ankyloglossia in infants has been estimated to be gual prominence, overfusion of the lateral lingual prominences, and
0.1% to 12.11%, but this wide range reflects the varying criteria for underdevelopment of anterior tongue length.33 The end result is a
anatomical and symptomatic ankyloglossia.11-24 With regard to the functionally significant frenulum attachment of mucosa and fibro-
prevalence of ankyloglossia, the criteria that were used, such as muscular tissue in the midline.
frenulum length, attachment location, or functional impact, must be
considered. Genetics
Many studies of ankyloglossia11-25 use anatomical attachment A male predominance of ankyloglossia is consistently demon-
criteria alone, identifying only a classic type of ankyloglossia, but likely strated with male to female ratios from 1.1:1 to 3:1.12,15,16,18,19,37,38

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 Diagnosis and Treatment of Ankyloglossia in Newborns and Infants Review Clinical Review & Education

Figure 1. Development of the Tongue Mucosa From the Endoderm of the Pharyngeal Arches

A Four weeks B Five weeks C Six weeks

Foramen
Distal tongue buds cecum
Will form anterior
Foramen cecum two-thirds of tongue Terminal
sulcus

Median
tongue 1
bud
Palatine
2
Copula tonsil

Epiglottis

Will form posterior

Hypopharyngeal one-third of tongue
eminence

D Scanning electron micrograph

Distal tongue bud

Mandibular swelling

Median sulcus

Median tongue bud

Drawings show tongue formation from the first through the fourth arches at gestational weeks 4 to 6. A scanning electron micrograph demonstrates the developing
tongue early in the sixth week of gestation. Adapted with permission from Elsevier publishing.33

However, distinctions between sporadic and familial cases of anky- Simosa,51 and oral-facial digital syndromes.52 Ehlers-Danlos syn-
loglossia have been found.39 Most cases of ankyloglossia are thought drome has been associated with absent frenulum, although this ob-
to be sporadic and have a higher male predilection than the familial servation is not uniformly accepted.53
cases. A genetic etiology is suspected, but different sex ratios and
patterns are seen for specific ankyloglossia types. Environmental or Clinical Presentation
teratogen causes of ankyloglossia have not been reported with any The clinical presentation of symptomatic ankyloglossia includes
consistency.40 O’Callahan et al8 reported that the male predomi- breastfeeding difficulties and examination findings in the infant
nance decreased from 68% for types 1 and 2 ankyloglossia to 59% and the mother. For the child, ankyloglossia can manifest as poor
for type 3, and 46% for type 4 ankyloglossia using the Coryllos clas- latching, frequent loss of latch, prolonged feeding, irritability with
sification. Similar trends were noted by Haham et al.32 feeding, poor weight gain, or inability to breastfeed. On physical
An X-linked cleft palate syndrome has been reported with cleft examination, tongue mobility restriction, heart-shaped tongue
palate and associated ankyloglossia.41-43 This X-linked cleft palate deformity, dimpling, or restriction of tongue protrusion may be
syndrome is caused by the TBX22 gene mutation (OMIM 300307), found. The lingual frenulum can be attached at various locations
a T-box gene involved in early vertebrate development.43 The G pro- along the length of the tongue and the alveolus. In addition to
tein coupled receptor LGR5 (OMIM 606667) has also been demon- attachment location, a thickened or a shortened frenulum might
strated to be a candidate gene in mice.44 impair tongue function. Associated upper-lip frenulum restriction
Familial ankyloglossia has been reported with X-linked and au- is also frequently reported.28 For posterior ankyloglossia, the
tosomal dominant inheritance with incomplete penetrance pat- feeding symptoms are similar; however, the identification of the
terns based on pedigree analysis.39,45,46 A Finnish familial cohort frenulum can often only be achieved with palpation under the
demonstrated autosomal dominant inheritance with no TBX22 tongue or use of a grooved retractor.
mutations.46,47 The mother of an infant with ankyloglossia can experience pain
Several other known syndromes include ankyloglossia associa- with breastfeeding, nipple ulceration, nipple bleeding, poor milk let-
tion, such as Opitz,48 van der Woude,49 Beckwith-Wiedemann,50 down due to inadequate infant suck, mastitis or nipple infections,

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 Clinical Review & Education Review Diagnosis and Treatment of Ankyloglossia in Newborns and Infants

Figure 2. Coryllos Frenulum Classification

A Coryllos type 1 B Coryllos type 2

C Coryllos type 3 D Coryllos type 4

Type 1 indicates attachment of the

frenulum to the tongue tip; type 2,
attachment 2 to 4 mm behind the
tongue tip and on or just behind the
alveolar ridge; type 3, attachment to
the midtongue and middle of the
floor of mouth; and type 4,
attachment against the base of the
tongue.

or incomplete emptying. However, these symptoms are not unique ance score is less than 8 or the function score is less than 11. Perfect
to ankyloglossia. Breastfeeding difficulty can lead to maternal and function scores regardless of appearance do not require fre-
infant frustration, maternal anxiety, feelings of failure or inad- notomy. Function scores of 11 to 14 are acceptable if appearance score
equacy, and early abandonment of breastfeeding. is 10.54,56 By allowing for functional assessment, the HATLFF en-
ables inclusion of posterior ankyloglossia, which may be missed in
Classification attachment-only scales. Routine use of the HATLFF can be difficult
Several classification schemes are published, but none have been because assessment is complex and time-consuming.57,58 The BTAT
used consistently and many are difficult for routine clinical use. The was developed to incorporate much of the benefits of the HATLFF
common grading systems for anatomical criteria use point of tongue but to make it more portable and teachable. The scale has 4 items
attachment, length of frenulum, and tongue protrusion (Table 1). The to grade tongue tip appearance, alveolar attachment location, tongue
Coryllos classification has 4 types of frenulum based on point of lift, and tongue protrusion and was found to correlate well with
attachment26 (Figure 2). Two versions of the Kotlow system have HATLFF findings (range, 0-8, with higher scores indicating greater
been published.27,28 Similar to the Coryllos system, the systems mea- severity of ankyloglossia).55 With emphasis on symptomatic anky-
sure the free tongue length from the tip of the tongue to the frenu- loglossia, anatomical appearance alone is insufficient in evaluation
lum attachment. The frenulum length, as measured from origin to and management of infants, regardless of grading system used.
insertion, can be difficult to measure. Some grading systems use in-
terincisal distance or the tongue protrusion distance as proxy mea- Treatment
surements. Obtaining these measurements in an infant is not often The primary treatment for ankyloglossia in infants is frenotomy. Non-
practical.24,29-31 surgical therapies for symptoms associated with ankyloglossia that
Systems that incorporate function in addition to anatomy are are often implemented by lactation specialist consultation are nipple
the Hazelbaker Assessment Tool for Lingual Frenulum Function shields, changes in positioning, or tongue stretching. Other pro-
(HATLFF)54 and the Bristol Tongue Assessment Tool (BTAT).55 The posed treatments are physical therapy, speech therapy, and alter-
HATLFF is the most complete and complex assessment of form and native and complementary medicine treatments, including cranio-
function of the tongue and lingual frenulum (Table 2), with 10 points sacral therapy, naturopathy, and orofacial myofunctional therapy.
for frenulum appearance and 14 points for tongue function. Fre- Surgical techniques include frenotomy, frenulectomy with or
notomy is suggested for symptomatic ankyloglossia if the appear- without myotomy, and Z-plasty.24,59-62 In most infants, frenotomy

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 Diagnosis and Treatment of Ankyloglossia in Newborns and Infants Review Clinical Review & Education

is sufficient. A common frenotomy technique involves isolation of

Table 2. The Hazelbaker Assessment Tool for Lingual Frenulum Functiona
the frenulum with a grooved retractor and cutting of the frenulum
Item Description Score down to the base of the muscle with scissors. Hemostasis is ob-
Appearance
tained with pressure, oxymetazoline, silver nitrate, or suturing. Other
Appearance of tongue when lifted
techniques include use of carbon dioxide, diode, erbium:YAG, or Nd:
Round or square 2
YAG lasers or electrocautery.28,59,63-65 A randomized clinical trial66
Slight cleft in tip apparent 1
regarding topical anesthesia for office frenotomy concluded that
Heart shaped 0
topical anesthesia options are ineffective and need not be used. In-
Elasticity of frenulum
stead of topical anesthesia, a 24% sucrose solution given orally be-
Very elastic (excellent) 2
fore the procedure accompanied by postprocedure nonnutritive
Moderately elastic 1
Little or no elasticity 0
sucking can help reduce discomfort.67
Length of lingual frenulum when tongue lifted
More rarely, frenulectomy (excision of the frenulum) or frenu-
More than 1 cm or absent frenulum 2 loplasty, which incorporates flap elevation or Z-plasties, have been
1 cm 1 described, but both usually require general anesthesia to perform
<1 cm 0 in infants. No studies have demonstrated these techniques to be
Attachment of lingual frenulum to tongue more effective than frenotomy in infants.
Posterior to the juncture between the body and blade 2 The strength of evidence for the benefits of surgical treatment
of the tongue is limited and even more limited for nonsurgical therapies.68-73 Five
In front of the juncture between the body and the blade 1
of the tongue randomized clinical trials74-78 evaluating frenotomy have been pub-
At the tip with or without notching 0 lished. These trials vary in diagnostic criteria and length of fol-
Attachment of lingual frenulum to inferior alveolar ridge low-up and have large treatment crossover within the study groups.
Attached to floor of mouth 2 In addition, many trials do not control for concomitant developmen-
Attached to the backside of the inferior alveolus 1 tal anomalies, such as retrognathia or lip tie.
Attached to ridge of inferior alveolus 0 Complication rates reported for ankyloglossia procedures are
Function low.3,79 The complications include bleeding, recurrence, lip injury,
Lateralization injury to the Wharton ducts, infection, lingual dysfunction, and air-
Complete 2 way compromise. Bleeding is the most common complication re-
Body of tongue but not tongue tip 1 ported, with a range of 3% to 5%, and most bleeding requires no
None 0 intervention.74,77,80-82 However, in an anonymous survey of ob-
Lift of tongue served complications by clinicians,3 scarring with recurrence of an-
Tip to middle mouth 2 kyloglossia was noted by 14% of responding otolaryngologists but
Only edges to middle mouth 1 only 2% of pediatricians, lactation consultants, and speech patholo-
Extension of tongue 0 gists. Bleeding complications were reported by 8% of otolaryngolo-
Tip over lower lip 2
gists in the same survey.3 More severe complications have been oc-
Tip over lower gum only 1
casionally reported. Lin et al83 reported a case of Ludwig angina
Neither of the above or anterior or middle tongue humps 0
and/or dimples following frenuloplasty. Two cases of severe bleeding with hypovo-
Spread of anterior tongue lemic shock have been reported.84 Two additional reports85,86 have
Complete 2 found airway obstruction after frenotomies in patients with Pierre
Moderate or partial 1 Robin Sequence. Frenotomy in infants with Pierre Robin Sequence
Little or none 0 or other craniofacial anomalies should be considered with caution
Cupping (Box).
Entire edge, firm cup 2
Side edges only or moderate cup 1 Prognosis
Poor or no cup 0 Treatment outcome measures used in studies include maternal re-
Peristalsis ports and structured assessments of breastfeeding, such as the
Complete anterior to posterior 2 Breastfeeding Self-Efficacy Scale, the Infant Breastfeeding Assess-
Partial or originating posterior to tongue tip 1 ment Tool, and the Latch, Audible Swallowing, Type of Nipple, Com-
None or reverse motion (tongue-thrust) 0 fort, Hold assessment.68 The natural history of untreated ankylo-
Snapback
glossia remains unknown owing to the wide range of published ages
None 2
of presentation, ease of and bias toward treatment, and differ-
Periodic 1
ences in symptom severity. In 2015, 2 systematic reviews68,69 ana-
Frequent or with each suck 0
lyzed the published studies regarding the influence of surgical treat-
a
Assessment includes 10 points for frenulum appearance and 14 points for ment of ankyloglossia on breastfeeding and nonbreastfeeding issues.
tongue function. For a function score of 14, regardless of appearance item
These reports noted improvement in breastfeeding and nipple pain
score, surgical treatment not recommended. A functional score of 11 is
acceptable only if the appearance item score is 10. A functional score of less as assessed by maternal report, but the overall strength and qual-
than 11 indicates impaired function. Frenotomy should be considered if ity of evidence in support of frenotomy is low. Several other re-
management fails. Frenotomy is necessary if the appearance item score is less
views had similar findings.70-73 Of the 5 published randomized clini-
than 8. Adapted with permission from Alison Hazelbaker, PhD.54
cal trials,74-78 most of the control patients had crossover to frenotomy

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 Clinical Review & Education Review Diagnosis and Treatment of Ankyloglossia in Newborns and Infants

ankyloglossia.8 The maternal knowledge and prior experience with

Box. Key Clinical Points breastfeeding also may affect the accuracy of measured results. First-
time mothers are more likely to have an infant who is diagnosed with
Diagnosis ankyloglossia and were also less likely to correctly identify whether
Acknowledge the goal and importance of successful breastfeeding frenotomy had been performed in a blinded study.2,77
Evaluate symptoms of the breastfeeding dyad The role of untreated ankyloglossia on speech, malocclusion,
Educate the parents regarding the complex and multifactorial mandibular incisor irregularity, gingival recession, mandibular growth,
nature of feeding difficulty and tongue mobility in older children has been considered. Tongue
Assess frenulum attachment and tongue function mobility can be restricted in ankyloglossia.88 Insufficient data are
Assess for other features (ie, lip tie, retrognathia, craniofacial available to determine association of ankyloglossia with speech ar-
anomalies, and developmental delay) ticulation difficulties.69,70 The concern for malocclusion has been
Decision
reported, but confirmed associations with dentofacial anomalies vary
Offer frenotomy for infants with suspected ankyloglossia and and evidence is limited.15,71,89 Mandibular incisor irregularity has not
breastfeeding difficulty been associated with ankyloglossia Kotlow grade but has been as-
Obtained informed consent including risks of no benefit and sociated with measured frenulum length in 1 study.90 Gingival re-
bleeding cession associated with frenulum attachment is controversial.71

Treatment
Use 24% oral sucrose before frenotomy
International Perspective
Statements from several countries and societies address ankylo-
Apply pressure with gauze with or without oxymetazoline for
immediate hemostasis
glossia and the role of frenotomy. The Canadian Paediatric Society
in 2015,91 American Academy of Pediatrics in 2004,26 American
Use postprocedure nonnutritive sucking or breastfeeding for pain
control and to assist with hemostasis
Academy of Pediatric Dentistry in 2016,92 Australian National Health
and Medical Research Council in 2012,93 the UK National Institute
Ensure adequate breastfeeding dyad support services are
available
for Health and Care Excellence,94 and the Japanese Pediatric
Society95 do not endorse universal frenotomies for infants with an-
Follow-up to assess feeding improvement and complications
kyloglossia based on available literature, but many recommend the
procedure when the ankyloglossia impairs breastfeeding. The Acad-
as part of the study designs. Two trials74,77 evaluated immediate re- emy of Breastfeeding Medicine protocol in 2004 for neonatal an-
sults only, because most control patients had frenotomy shortly af- kyloglossia states that “conservative management of tongue-tie may
ter a sham procedure. Hogan et al75 reported that 28 of 29 control be sufficient, requiring no interventional beyond breastfeeding as-
patients underwent frenotomy within 48 hours. Buryk et al76 re- sistance, parental education, and reassurance.”96(p3) However, the
ported that 27 of 28 control patients had frenotomy within 2 weeks. academy acknowledges that in some patients, tongue-tie release is
Emond et al78 reported that 44 of 52 controls had frenotomy within appropriate but possibly ineffective in solving the clinical problem.96
the 8-week study period. With such heterogeneity, large cross-
over, and limited follow-up within this population, the benefit of fre-
notomy for all infants with ankyloglossia is difficult to assess. In ad-
Conclusions
dition, the need for revision treatment for recurrent symptoms is
difficult to assess, but rates for revision have been reported to range Diagnosis and management of ankyloglossia in the infant can be a
from 2.6%87 and 6.5%.81 source of confusion and disagreement for clinicians and families. Fre-
Some infants will benefit from frenotomy, whereas others who notomy is a low-risk procedure that is likely to be beneficial in the
have multifactorial causes of feeding difficulty may have minimal ben- carefully selected patient, but the natural history of untreated an-
efit from the procedure. Possible factors affecting the results are the kyloglossia is still undetermined. The variability in treatment out-
type of ankyloglossia and presence of associated findings, such as comes and presentation of ankyloglossia indicate that the complex-
lip tie. A recent web-based postintervention maternal report study ity of infant feeding mechanisms and tongue development is not fully
found 100% improvement in latching difficulties for types 1 and 2 encapsulated in simplistic ankyloglossia etiology framework. Con-
(classic) ankyloglossia and 49% improvement for type 4 (poste- sistent terminology with emphasis on symptomatic ankyloglossia
rior) ankyloglossia. For maternal pain, 79% improvement was noted and a uniform grading system, such as the HATLFF and Coryllos
in types 1 and 2 ankyloglossia and 63% improvement in type 4 grades, are needed to improve the quality of research in the future.

ARTICLE INFORMATION Acquisition, analysis, or interpretation of data: Disclosure of Potential Conflicts of Interest and
Accepted for Publication: April 21, 2017. Walsh. none were reported.
Drafting of the manuscript: Walsh.
Published Online: July 13, 2017. Critical revision of the manuscript for important REFERENCES
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Study concept and design: Both authors. have completed and submitted the ICMJE Form for

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