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Comparison of Frenotomy Techniques For The Treatment of Ankyloglossia in Children.2020
Comparison of Frenotomy Techniques For The Treatment of Ankyloglossia in Children.2020
Comparison of Frenotomy Techniques For The Treatment of Ankyloglossia in Children.2020
Otolaryngology–
Head and Neck Surgery
A
nkyloglossia or tongue-tie is a congenital condition Usman Khan, MSc, Department of Surgery, Faculty of Medicine, Dalhousie
where anatomical variation of the sublingual frenu- University, 1459 Oxford Street, Halifax, NS B3H 4R2, Canada.
lum can limit normal tongue function.1,2 The most Email: usman.khan@dal.ca
2 Otolaryngology–Head and Neck Surgery
Figure 1. Literature search flow diagram based on the Preferred Reporting Items for Systematic Review and Meta-Analyses (PRISMA).
Israel (3), Japan (2), South Korea (1), Spain (1), Thailand inclusion of consecutive patients, and using a protocol for
(1), United Kingdom (11), and United States (7). collecting data.
The overall mean (SD) age of participants was 1.04 (2.3) The overall quality of evidence based on the GRADE
years (range, 2 days to 8 years). LF and ZF studies had approach was low for CF articles and very low for ZF and
older patients with mean ages of 3.1 and 4.0 years, respec- LF articles, respectively. CF RCTs were downgraded to
tively, compared to 0.4 years for CF patients (P \ .05). low quality for imprecision (small study populations) and
There were more male than female patients for all types of risk of bias similar to a previous Cochrane review (CF-only
frenotomy. Indications for frenotomy primarily included RCTs).19 However, several observational studies demon-
breastfeeding difficulties and speech problems. strated benefit of CF using validated outcome measures
and/or compared with controls in large patient cohorts. ZF
Quality of Evidence articles were determined to be of very low-quality evidence
The majority of articles assessed breastfeeding, speech, or due to imprecision (small sample sizes), risk of bias
tongue movement as postsurgical outcomes (Table 2). A (unclear randomization, incomplete blinding, lack of alloca-
total of 7 RCTs were included: 1 study compared ZF with tion concealment), and limitations in study design (outcome
CF, 1 study compared ZF with conventional frenuloplasty measures were not validated). LF articles were also very
(horizontal to vertical), and 5 compared CF with no treat- low quality with observational studies lacking control
ment or sham control group (see Suppl. Table S1 in the groups, although validated outcome measures were used,
online version of the article). The remaining articles were and patients were followed prospectively in some cases.
observational studies (case series, case-control, or cohort
studies). Outcomes for all studies were assessed using ques- Breastfeeding Outcomes
tionnaires, interviews, or telephone conversations. The Objective breastfeeding outcomes were assessed using vali-
MINORS criteria were used for non-RCT studies, which dated tools: Breastfeeding Self-Efficacy Scale (BSES),
represent the majority of included articles. The research LATCH scoring tool, and Infant Breastfeeding Assessment
methodologies were variable, which is reflected in the range Tool (IBFAT). Of 8 CF studies (588 patients) using vali-
of quality assessment scores (Table 2). The overall quality dated instruments for assessment of breastfeeding outcomes,
remained low as only 4 of 28 articles provided a control 7 (88%) reported a significant postoperative improvement.
group for comparison. The remainder of the articles were One LF study by Ghaheri et al6 used the BSES scale, report-
assessed on the 8 noncomparative parameters. In particular, ing a mean score increase of 12.5 after frenotomy (P \
the major limitations were the inability to successfully blind .001). Another study by the same authors reported a mean
participants and the lack of sample size calculations. The BSES score increase of 10.8 after treatment with revision
articles were consistent in stating a clear aim for the study, LF (P \ .001).7 One RCT comparing ZF to CF reported
4 Otolaryngology–Head and Neck Surgery
(continued)
Khan et al 5
Table 1. (continued)
Author Country Frenotomy No. Patient Demographics
(continued)
6 Otolaryngology–Head and Neck Surgery
Table 1. (continued)
Author Country Frenotomy No. Patient Demographics
(continued)
8 Otolaryngology–Head and Neck Surgery
Table 2. (continued)
Author Frenotomy Study Design Study Qualitya Outcome Assessment
(continued)
Khan et al 9
Table 2. (continued)
Author Frenotomy Study Design Study Qualitya Outcome Assessment
Table 3. Maternal Nipple Pain Outcomes after Frenotomy Using the Visual Analog or Numeric Rating Scale.
Pain Scores (SD or IQR)
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
significant improvements in subjective maternal reports group (horizontal to vertical) and demonstrated a high risk
postfrenotomy in both groups with no differences between of bias in their research methods (Table 2).
frenotomy techniques.14
Maternal nipple pain (MNP) was evaluated with the Speech Outcomes
visual analog scale (VAS), numeric rating scale (NRS), or All studies used different methods for measuring postopera-
McGill Pain Questionnaire (MPQ) (Tables 2 and 3). Seven tive changes in speech (Table 6). Two ZF studies reported
of 9 (78%) CF studies reported a significant postoperative speech outcomes assessed by speech language pathologists.
improvement. Only 2 RCT found no significant improve- Yousefi et al14 reported a significantly higher reduction of
ments for MNP after frenotomy. A patient-weighted mean speech errors in the ZF group compared to the CF group.
improvement in pain scores of –3.6 and –2.7 was observed Heller et al13 reported improvements in speech errors by an
following CF and LF, respectively, for non-RCT studies ‘‘order of magnitude’’ from baseline; however, no assessment
(Table 4). CF RCTs were not included as no RCTs investi- of statistical significance was provided. For CF, Messner and
gating MNP were available for other frenotomy methods. Lalakea25 reported a significant improvement in parental-
reported speech problems, and Walls et al50 demonstrated a
Tongue Movement Outcomes significant difference in parental-reported speech outcomes
Three studies reported tongue movement as an outcome between treatment and no-treatment groups.
(Table 5). Two RCTs reported a significant increase
in tongue protrusion after frenotomy for all treatment Anesthesia Requirements
groups.13,14 However, both studies reported a significantly Thirteen articles explicitly reported that frenotomy was
larger increase in tongue protrusion in the ZF group com- performed without anesthesia; 10 articles did not specify
pared to the CF and conventional frenuloplasty group (hori- whether anesthesia was used. Out of the articles evaluating
zontal to vertical), respectively. Yousefi et al14 were unclear LF, Fiorotti et al22 used topical 10% lidocaine spray fol-
about their method of measuring tongue protrusion. Heller lowed by 1.8 mL of 2% lidocaine solution without a vaso-
et al13 only had 5 patients in the conventional frenuloplasty constrictor, Ghaheri et al6 applied a topical anesthetic
Khan et al 11
Heller et al13 Z-plasty and frenuloplasty 11 (ZF) Tongue protrusion: Mean (SD) increase of 36.2 (7.6) mm (P \.0001) in
5 (F) the ZF group and 13.2 (2.6) mm (P = .0003) in the F group
Messner and Lalakea25 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) Hazelbaker score: Mean (SD) increase of 2.91 (0.302) mm (P \.001) in
25 (CF) 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.
cream (EMLA), and Komori et al11 used a combination of Lalakea25 performed tongue-tie release under general
general (n = 7) and local (n = 8) anesthetic. For the articles anesthesia. These patients were older children (age .3)
evaluating variations of the frenuloplasty, Ferrés-Amat et who underwent frenuloplasty.
al23 used a local anesthetic (articaine 4% with epinephrine
1:100,000), and both Heller et al13 and Yousefi et al14 per- Adverse Events
formed ZF under general anesthesia. For the articles evalu- No serious adverse events were reported. Minor events are
ating CF, only Ito et al,40 Ballard et al,24 and Messner and summarized in Table 7.
12 Otolaryngology–Head and Neck Surgery
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.
Ferrés-Amat et al23 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- commonly used during preoperative assessment only. A
ing ankyloglossia that causes breastfeeding difficulties. range of health care professionals were reported to be
There was no evidence to suggest that one technique was involved in supporting patients through breastfeeding diffi-
superior over others. culties, including infant coordinators, lactation consultants,
midwives, and nurses. In some studies, patients were
Tongue Movement and Speech Outcomes referred by lactation consultants to otolaryngologists for
The functional improvements associated with frenotomy consideration of frenotomy. This would suggest that nono-
result from enhancement of tongue mobility. While func- perative measures may have been exhausted by the lactation
tional outcomes are the typical measure of treatment benefit, consultants. Lactation consultation was provided as a con-
improvements in tongue mobility may also be used to com- trol group in 1 study by Hogan et al,28 who offered patients a
pare surgical techniques. A validated assessment tool for lactation consultation for 48 hours. Patients who did not
scoring tongue structure and function is the Hazelbaker tool, improve after 48 hours (27 of 28 patients) were offered a fre-
which can be used to classify the severity of ankyloglossia.1 notomy. From an ethical standpoint, it is important to note that
Three studies evaluated tongue movement or function post- researchers in studies comparing frenotomy and nonoperative
frenotomy but only 1 study used this tool (Table 5). Two treatments may be ethically obligated to offer surgical correc-
articles compared ZF vs CF or conventional frenuloplasty tion to nonoperative patients after a preestablished time frame.
(horizontal to vertical) and reported a significantly higher This provides challenges for randomization and comparing
increase in tongue protrusion for the ZF group.13,14 Both long-term outcomes between frenotomy and nonoperative
articles used only univariate analysis to outline statistical treatments. However, the inclusion of a short-term nonopera-
differences and 1 study included only 5 patients in the tive control group as demonstrated by Hogan et al28 or patients
horizontal-to-vertical frenuloplasty control group.14 All ZF who willingly refused surgery would help outline the differ-
articles mentioned prevention of scar contracture as a bene- ences between frenotomy and nonoperative treatments.
fit of ZF over other techniques.13,14,26 However, our review
suggests that there is no high-quality evidence to conclude Study Limitations
the superiority of ZF over other techniques in improving While many studies have investigated the utility of different
tongue mobility. frenotomy techniques, there are several methodological gaps
Speech was also a measured outcome for studies investi- preventing a meta-analysis. Only 1 RCT used CF as a com-
gating CF and LF. However, the controversy surrounding parison group. Moreover, only 4 non-RCT studies compared
the association of speech abnormalities with ankyloglossia, with a control group. Postsurgical outcomes were frequently
heterogeneity of outcomes measured, and lack of statistical reported using subjective measures. Outcome assessments
comparison prevents any recommendations to be drawn were largely heterogeneous, and validated assessment tools
from these studies. were used in a minority of studies. There were several cases
where patient selection criteria were either not mentioned or
Surgical Complications and Operating Time not determined using a validated scoring tool such as the
Surgical complications were not explicitly mentioned in all Hazelbaker tool. This led to considerable selection bias.
studies. Therefore, a qualitative review was performed to Some studies also provided lactation consultation or speech
determine adverse events (Table 7). Weighted averages therapy to patients after frenotomy. In these cases, the bene-
were not computed for these data as the quantification meth- fit of frenotomy could be biased by benefit achieved from
ods varied in the articles. Overall, there is no compelling nonoperative treatments. Moreover, statistical calculation of
evidence that the choice of frenotomy technique offers any mean differences with respective confidence intervals was
advantage in preventing surgical complications. not consistently available.
Operation time was not a measured outcome in any Studies in this review were limited to a retrospective
study; however, it was mentioned in the methods section of methodology. The current review was restricted to English-
2 articles. Buryk et al27 performed CF in an average of 5 language articles for reasons of accessibility. This review
minutes. Fiorotti et al22 performed LF, which required 15 to may be associated with publication bias for those institu-
25 minutes to complete. While there is no clear advantage tions with positive outcomes.
of any technique with respect to operative parameters, a
beneficial future study would be to investigate overall cost- Recommendations
effectiveness. Our review has identified several methodological shortcom-
ings of current research seeking to demonstrate the superiority
Nonoperative Management of different frenotomy techniques. In addition, our review out-
The most common nonoperative management modalities lines the importance of investigating nonoperative techniques
used in studies were lactation consultation and speech ther- to manage issues associated with ankyloglossia. Based on the
apy. There are limited studies in the current literature that identified gaps, we have the following recommendations:
compare frenotomy with nonoperative management tech-
niques. Lactation consultation and other forms of profes- 1. Control groups: The inclusion of a control group
sional support for breastfeeding difficulties were most where patients are treated with CF is critical
14 Otolaryngology–Head and Neck Surgery
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16 Otolaryngology–Head and Neck Surgery
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