10.14639/0392-100X-N2211
Objective. To evaluate outcomes of the surgical and rehabilitative procedures devoted to release the tongue-tie in non-infants when implementing the most commonly used quantitative/ qualitative structured tools for tongue and frenulum assessment. Methods. A scoping review and meta-analysis were conducted following the guidelines of the Preferred Reporting Items for Systematic Review and Meta-Analysis extension for scoping reviews.Results. The systematic search retrieved 603 (Pubmed), 893 (Scopus), and 739 (ISI Web of Science) articles from January 2011 to December 2021. A total of 50 articles were retrieved for full-text review of which 7 were selected and included based on inclusion criteria. The majority of treatment options have been found to significantly improve the anatomical limitation of the tongue with clear benefits on descending functionality.Conclusions. The review highlights an overall improvement in terms of clinical and functional outcomes when using validated tongue assessment tools both before and after frenulum release. This highlights the need for their rigorous implementation in research and clinical practice.
text/xml
1827-675X
2211
en
Marco Alessandrini
http://creativecommons.org/licenses/by-nc-nd/4.0
ACTA Otorhinolaryngologica Italica
Marchesan protocol
Outcomes of tongue-tie release by means of tongue and frenulum assessment tools: a scoping review on non-infants
Text.Serial.Journal
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Reviews
2022-12-29
2022-07-10
2022-12-29
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1
Marco Alessandrini
2022/12/29
10.14639/0392-100X-N2211
492
Department of Clinical Sciences and Translational Medicine, ENT Unit, University of Rome Tor Vergata, Rome, Italy
1827-675X
Marchesan protocol
en
501
Outcomes of tongue-tie release by means of tongue and frenulum assessment tools: a scoping review on non-infants
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ACTA OTORHINOLARYNGOLOGICA ITALICA 2022;42:492-501; doi: 10.14639/0392-100X-N2211




Review


Outcomes of tongue-tie release by means of tongue
and frenulum assessment tools:
a scoping review on non-infants
Impatto della correzione dell’anchiloglossia nei giovani e negli adulti:
revisione mediante valutazioni frenulo-linguali
Martina Arena1*, Alessandro Micarelli1*, Federico Guzzo2, Ilaria Misici1, Diana Jamshir2, Beatrice Micarelli1,
Alberto Castaldo2, Adriano di Benedetto1,3*, Marco Alessandrini4*
1
  Unit of Neuroscience, Rehabilitation and Sensory Organs, UNITER ONLUS, Rome, Italy; 2 Unit of Dentistry and Oral Health,
UNITER ONLUS, Rome, Italy; 3 Occupational Therapy Unit, IRCCS Santa Lucia Foundation, Rome, Italy; 4 Department of Clinical
Sciences and Translational Medicine, ENT Unit, University of Rome Tor Vergata, Rome, Italy
*
  These authors equally share these positions.


SUMMARY
Objective. To evaluate outcomes of the surgical and rehabilitative procedures devoted to
release the tongue-tie in non-infants when implementing the most commonly used quantita-
tive/qualitative structured tools for tongue and frenulum assessment.
Methods. A scoping review and meta-analysis were conducted following the guidelines
of the Preferred Reporting Items for Systematic Review and Meta-Analysis extension for
scoping reviews.
Results. The systematic search retrieved 603 (Pubmed), 893 (Scopus), and 739 (ISI Web of
Science) articles from January 2011 to December 2021. A total of 50 articles were retrieved
for full-text review of which 7 were selected and included based on inclusion criteria. The
majority of treatment options have been found to significantly improve the anatomical limi-
tation of the tongue with clear benefits on descending functionality.
Conclusions. The review highlights an overall improvement in terms of clinical and func-
tional outcomes when using validated tongue assessment tools both before and after frenu-               Received: July 10, 2022
lum release. This highlights the need for their rigorous implementation in research and                 Accepted: September 17, 2022
clinical practice.
                                                                                                        Correspondence
KEY WORDS: tongue-tie release, ankyloglossia, scoping review, Kotlow classification,
                                                                                                        Alessandro Micarelli
Marchesan protocol
                                                                                                        Unit of Neuroscience, Rehabilitation and Sensory
                                                                                                        Organs, UNITER ONLUS, Rome, Italy
RIASSUNTO                                                                                               E-mail: alessandromicarelli@uniteronlus.it
Obiettivo. Considerato il dibattito che ruota attorno alle tecniche correttive dell’anchilo-
glossia in soggetti giovani e adulti, l’obiettivo della presente revisione è quello di chiarire           How to cite this article: Arena M, Micarelli A,
come tali tecniche impattino in soggetti con età superiore ad un anno in termini di risultati             Guzzo F, et al. Outcomes of tongue-tie release
quando valutati mediante i principali strumenti quali-quantitativi di valutazione frenulo-                by means of tongue and frenulum assessment
linguale.                                                                                                 tools: a scoping review on non-infants. Acta
Metodi. La revisione di scopo e la meta-analisi – in accordo alle attuali linee guida – sono              Otorhinolaryngol Ital 2022;42:492-501. https://
state eseguite a seguito di una ricerca sistematica che ha condotto a valutare 603, 893 e 739             doi.org/10.14639/0392-100X-N2211
articoli provenienti rispettivamente da Pubmed, Scopus e ISI Web of Science.
Risultati. 50 di questi articoli sono stati consultati in estenso e 7 selezionati sulla base dei cri-   © Società Italiana di Otorinolaringoiatria
                                                                                                        e Chirurgia Cervico-Facciale
teri di inclusione. La maggior parte dei trattamenti eseguiti ha dimostrato di migliorare le limi-
tazioni funzionali della lingua con considerevole miglioramento delle funzioni ad essa collegate.
                                                                                                                       OPEN ACCESS
Conclusioni. La presente revisione ha mostrato un globale miglioramento in termini clinici
e funzionali esplorati mediante test di valutazione frenulo-linguale sia prima che dopo il              This is an open access article distributed in accordance with
                                                                                                        the CC-BY-NC-ND (Creative Commons Attribution-Non-
trattamento correttivo e rimarca una loro rigorosa implementazione nella pratica clinica                Commercial-NoDerivatives 4.0 International) license. The
e di ricerca.                                                                                           article can be used by giving appropriate credit and mentio-
                                                                                                        ning the license, but only for non-commercial purposes and
PAROLE CHIAVE: frenuloplastica, anchiloglossia, revisione di scopo, classificazione di                  only in the original version. For further information: https://
Kotlow, protocollo Marchesan                                                                            creativecommons.org/licenses/by-nc-nd/4.0/deed.en




492
                                                                                        Impact of tongue-tie release on assessment tools




Introduction                                                     for Systematic Review and Meta-Analysis (PRISMA) ex-
                                                                 tension for scoping reviews (PRISMA-ScR) 19.
Ankyloglossia – or tongue-tie - is a congenital condition
characterised by an abnormally short, thick and/or tight
                                                                 Eligibility criteria
lingual frenulum which can restrict the protrusion and
                                                                 A literature search was performed to identify studies evalu-
elevation of the tongue 1,2, thus resulting in several func-
                                                                 ating outcomes of tongue-tie release procedures and imple-
tional limitations 1,2. The incidence of ankyloglossia in the
                                                                 menting assessment tools in non-infants in the last 10 years.
newborn population varies from 4.8% to 10% and com-
                                                                 The inclusion criteria (Tab. I) of the search strategy design
monly presents as difficulty breastfeeding as an infant 3.
                                                                 are categorised according to the broad Population-Con-
The literature typically differentiates ankyloglossia in neo-
                                                                 cept-Context (PCC) mnemonic recommended for scoping
nates and infants from that occurring during childhood and
                                                                 reviews 20,21. The scoping review focuses on applications
adolescence 1. Beyond its important consequences in the
                                                                 in clinical settings and on physiological research. Thus,
former group (e.g., breastfeeding, breathing complications,
                                                                 studies on patients > 1 year of age were included. Studies
phenomenon of asphyxiation, and food rejection) 4, in the
                                                                 were eligible only if outcomes of morphological/functional
latter typical consequences are represented by speech im-
                                                                 tongue-tie and frenulum length assessment tools, together
pairment 5-7 and functional limitations produce changes in
                                                                 with the results of objective tests investigating disability-
swallowing and chewing, preventing the correct coordina-
                                                                 related aspects of the ankyloglossia (i.e. speech, breath-
tion of muscles. This leads to a number of problems 8,9,
                                                                 ing and feeding), were implemented before and after the
such as oral respiration 10, forward tongue positioning with
                                                                 tongue-tie release procedures, while studies investigating
possible malocclusions 11,12 and consequences on breath-
                                                                 these aspects mainly using self-assessed questionnaires or
ing 13 and postural control 14.
                                                                 sub-items of the main assessment tools were excluded.
Ankyloglossia is usually treated with surgical division of the
                                                                 The search was restricted to observational and interven-
frenulum with closure (frenuloplasty) or without (frenotomy/
                                                                 tional studies involving humans and published in English in
frenulectomy) 15. However, there are no accepted guidelines
on diagnostic and treatment management of ankyloglossia 15.      peer-reviewed journals. Abstracts, conference proceedings
In this scenario, some experiences reported that after surgi-    and reports, retrospective studies, expert opinions, letters to
cal tongue-tie release different degrees of improvements are     the Editor, commentaries, case reports/series and reviews
found in terms of speech, social outcomes 7,16, dysphagia,       were excluded.
sleep-disordered breathing and obstructive sleep apnoea
syndrome (OSAS) 15,17. With respect to the large amount          Information sources, search strategy, and study selection
of evidence depicting improvement in the effectiveness of        A systematic search was performed in PubMed, Scopus, and
frenulectomy on feeding and decreased pain perception            ISI Web of Science electronic databases to identify primary
from the mother 2,3, many debates are still present about the    references from January 2011 to December 2021. The fol-
impact of ankyloglossia and – vice versa – frenulectomy          lowing search string was used: (“ankyloglossia” OR “tongue-
in non-infants on speech, feeding and sleep-related breath-      tie” OR “tongue” OR “lingual frenulum” OR “frenulo-
ing disorders, so that no significant conclusions have been      plasty” OR “frenectomy” OR “frenotomy” OR “tongue-tie
made to date 7,16. This was possibly due to different treat-     division” OR “tongue-tie release” OR “frenulum release”
ment procedures, relations with different functions related      OR “myofunctional therapy” OR “tongue rehabilitation”
to tongue limitations and no consistent assessment methods       OR “speech and language therapy”) AND (“assessment” OR
to evaluate the extent and/or severity of ankyloglossia in       “lingual mobility” OR “mouth opening” OR “articulation”
different studies 18.                                            OR “speech” OR “feeding” OR “swallowing” OR “breath-
Thus, the aim of the present scoping review is to evaluate       ing” OR “phonation” OR “frenulum length”). The database
the impact – related to the main functional limitations of the   search was followed by a review of the citations from eli-
ankyloglossia – of surgical and rehabilitative procedures de-    gible studies. The studies were selected based on their title
voted to release the tongue-tie in non-infants when imple-       and their abstract using the online platform Rayyan 22. The
menting the most commonly used quantitative/qualitative          selected studies were read thoroughly to identify those suit-
structured tools for tongue assessment and frenulum length.      able for inclusion in the scoping review.

                                                                 Data extraction
Scoping review methods                                           Two reviewers (AM and MA) independently extracted the
The scoping review and the meta-analysis were conducted          demographic and experimental data from the selected stud-
following the guidelines of the Preferred Reporting Items        ies. When they disagreed, they reviewed the papers togeth-



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M. Arena et al.




Table I. Inclusion criteria for the scoping review summarised according to the Population-Concept-Context (PCC) mnemonic, recommended for scoping re-
views 20,21.
 Population             Healthy and sick children > 1 years old and adults
                        Any sex
 Concept                Outcome of tongue-tie division procedures, including both surgical and rehabilitative treatments, when evaluated by means of
                        structured tongue and frenulum length assessment tools
 Context                Clinical setting, with focus on oral science, speech therapy, otorhinolaryngology, physiological research
                        Original peer-reviewed research articles (cross-sectional and interventional study design), published in English in the last ten years



er in order to reach joint conclusions. For each study, the
following information were extracted and summarised: the
characteristics of the sample of subjects under evaluation,
outcomes of assessment tools of frenulum length, as well
as of the tongue mobility and anatomy, and possible com-
parators of tongue functionality; type of tongue-tie division
of the investigated study group; main results of the study
with focus on tongue function changes after the tongue-tie
division.

Results
The literature search retrieved 603 (Pubmed), 893 (Sco-
pus), and 739 (ISI Web of Science) articles evaluating the
impact of tongue-tie release on lingual and frenulum as-
sessment tools. A total of 50 articles were retrieved for full-
text review of which 7 were selected and included based on
the inclusion criteria in the last 10 years (Fig. 1). The re-
sults are summarised narratively and presented in Table II,
according to the presence or absence of other complemen-
tary tests administered to evaluate the effect of tongue-tie
release. Collectively, two studies reported outcomes only
using a lingual frenulum protocol validated by Marchesan
and co-workers 14,23, while all other studies implemented
the Kotlow classification 1,13,24-26, and two studies did not
administer further tests to evaluate functionalities related                     Figure 1. Selection process for studies included in the scoping review.
to tongue mobility 1,23, by means of the Quick Tongue Tie
Assessment Tool®. Two studies also reported the results of
the distance between the interincisal margins with the tip                       frenuloplasty associated with myofunctional training 1, re-
of the tongue positioned at the level of the superior retroin-                   spectively, on a specific lingual frenulum protocol validated
cisive papilla (MOTTIP) 13,25 and of the protrusion of the                       by Marchesan et al. in 2010 27 and on the Kotlow classifica-
tongue 13,25, while one also showed outcomes of the maxi-                        tion (Tab. II). Although the work by Marchesan et al. did not
mum opening of the mouth (MAB) 13. One study performed                           perform any statistical analysis on post-surgical tongue-tie
polysomnography 13 and one evaluated speech impair-                              release scores, the authors described improvement in tongue
ment 26. Two studies performed the scalpel frenectomy in                         protrusion and tongue tip shape after 30 days 23. Ferrés-Amat
all subjects 1,23, and 1 in sub-groups of participants 26, while                 et al. found improvement (degree 1 or 2 by Kotlow classifi-
laser-assisted frenuloplasty was performed in all subjects in                    cation) of tongue-tie release in 29% of patients at the first
4 studies 13,14,24,25 or in sub-groups of participants in one 26.                post-surgical evaluation performed at 72 hours and in 96%
Five studies also delivered rehabilitation therapy 1,13,14,24,25.                of patients after post-surgical rehabilitation sessions 1.
Among these investigations, two studies by Marchesan et al.                      Table II also depicts the studies in which frenulum classifi-
and Ferrés-Amat et al. – respectively involving 10 and 101                       cations were associated with other tests that are indirectly
patients – only evaluated the impact of frenuloplasty 23 and                     associated with tongue functionality. Tecco and co-workers



494
                                                                                                                   Impact of tongue-tie release on assessment tools




Table II. Studies included in qualitative analysis after the selection process.
 Aim                       Tongue/             Other tests            Intervention/            Population/                     Results                  Reference
                          frenulum                                      follow-up              pre-release
                        assessment                                                          tongue/frenulum
                            tools                                                             assessment
To describe changes     Lingual frenulum           None           Frenectomy performed   10 subjects (8 males,      30 days after the intervention, Marchesan et al.
in tongue mobility and     protocol by                            by an otolaryngologist   2 females; mean         the best results were for tongue    2012 23
orofacial functions    Marchesan in 2010                                               age = 12.2 ± 12.4 years ) protrusion, while the worst were
                                                                                                               *

after frenectomy                                                    Follow-up: 30 days                               for tongue elevation. Surgery
                                                                                                                 improved the altered shape of the
                                                                                                                    tip of the tongue in 6 subjects,
                                                                                                                  the mouth opening in 6 subjects,
                                                                                                                       the speech in 4 subjects #
To assess changes in Kotlow classification sEMG potentials of          Laser-assisted         Treated group: 13          The Kotlow index passed        Tecco et al.,
the sEMG potentials                        the upper and lower        frenectomy and         subjects with Class       from 3-4 to score 0 (called        2015 24
in the sub-mental,                            fascicles of the    orofacial myofunctional I malocclusion and        ‘‘normal”: > 15 mm) or score 1
orbicularis oris,                         orbicularis oris muscle, therapy in the treated      ankyloglossia (4    (called ‘‘acceptable; 12-15 mm).
and masticatory                             sub-mental areas,              group          females, 9 males; mean        Treated group showed an
muscles after lingual                     masseter, and anterior                           age = 7.0 ± 2.5 years) increase in the sEMG potentials
frenulectomy and                          temporalis during rest Follow-up: T0 (before Control group: 11 subjects        over the follow-up during
OMT in subjects                             position, clenching     treatment), T1 (one with normal occlusion and clenching, swallowing (and
with ankyloglossia,                          of teeth, kissing,       month after the normal lingual frenulum (2 kissing (p < 0.05). During the
characterised by Class                    swallowing, opening of treatment) and T2 (6 females; 9 males; mean           protrusion of the mandible,
I malocclusion.                            mouth and protrusion months after treatment) age = 7 ± 0.8 years)            the masseter and anterior
                                                of mandible         in both treated and                           temporalis significantly decreased
                                                                        control group      Kotlow classification      their sEMG activity, while the
                                                                                          Grade 3 (called “severe sub-mental area increased
                                                                                            tongue-tie”, about            significantly (p < 0.05).
                                                                                            4-8 mm) and Grade During swallowing and protrusion
                                                                                            4 (called “complete of the mandible, the differences
                                                                                            tongue-tie”, about        between the treated and the
                                                                                                  0-4 mm)           control group decreased during
                                                                                                                         the follow-up (p < 0.01)
To investigate if       Lingual frenulum Postural evaluation by Laser frenectomy and 24 patients (8 females,        Interventions induced a            Saccomanno et
there is a correlation     Protocol by   using the Spinometry® orofacial myofunctional         16 males;          significant lingual frenulum           al., 2020 14
between a short        Marchesan IQ 2014     Formetric 4D               therapy         mean age = 15.22 years,     protocol score reduction
lingual frenulum and                                                                     range = 10-26 years)         (p < 0.001) and an
the tonic-postural                                              Follow-up: not reported                            improvement of shoulder
system before                                                                                Mean lingual       opening (N.S.) during maximum
and after a laser                                                                         frenulum protocol       mouth opening and tongue
frenectomy                                                                                 score = 4.7 ± 1.1      to palate with mouth open
                                                                                                                           conditions
To estimate the        Kotlow classification     Pain NRS           Laser frenectomy        125 patients (59             The Kotlow score was       Sfasciotti et al.,
efficacy of lingual                                               (Groups A-D), speech females, 66 males; mean       significantly higher in T3 and     2020 25
frenulectomy                MOTTIP                                therapy (Groups A-B), age = 8.37 ± 2.31 years)    T4 in Group D when compared
with diode-laser              MAB                                   sterile iodoformic    randomised in 5 sub-      to others (p < 0.001). From T1
                           Protrusion                               gauze affixed with        groups (A-E)           to T4 MOTTIP had a trend of
technology through
                      (by Quick Tongue Tie                        suture in Groups A-C.                            growth better in Group D than in
a qualitative          Assessment Tool®)                                                 Kotlow with a length      other groups (p < 0.001). MAB
and quantitative                                                   Follow-up at T0, T1, ≤ 16 mm and lack of          had a lower score in Group C
evaluation                                                         T2, T3, and T4, with tongue functionality         than in other groups from T1
                                                                   7 days of difference                             to T4 (p < 0.001). The value of
                                                                     amongst them.                                 Protrusion was greater in Group
                                                                    No intervention in                             E than in the other groups in T1
                                                                          Group E                                         and T2 (p < 0.001)




                                                                                                                                                          continues u



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M. Arena et al.




Table II. Studies included in qualitative analysis after the selection process (follows).
 Aim                       Tongue/             Other tests            Intervention/                           Population/                          Results                     Reference
                          frenulum                                      follow-up                             pre-release
                        assessment                                                                         tongue/frenulum
                            tools                                                                            assessment
To present the      Kotlow classification                   None                Frenectomy and 101 patients (38 females, In all patients the tongue is          Ferrés-Amat et al.
protocol of action                                                             rhomboidal plasty 63 males), ranking in age      released after the lingual           2016 1
of the Fundació                                                             Myofunctional training: from 4 to 13 years old        frenectomy and plasty.
Hospital de Nens de                                                             begins one week                            Post-surgical check-ups: degree of
                                                                               before the surgical  Kotlow classification ankyloglossia has been improved,
Barcelona (FHNB)
                                                                              intervention so that with Degree 3 (N = 11), considering correction (degrees 1
for the treatment                                                           the patients learn the    Degree 4 (N = 74),    or 2) in 29 (28%) of the patients
of ankyloglossia                                                            exercises without pain Degree 5 (N = 16)               (95% CI: 20%, 38%)
in childhood and                                                               and 24 hours after
adolescence (pre-                                                                   surgery                                Check-up after all the postsurgical
surgical, surgical                                                                                                           orofacial rehabilitation sessions:
technique, post-                                                             Follow-up: 72 hours,                             correction of 97 (96%) of the
surgical)                                                                       15 days, 45 days                            participants (95% CI: 90%, 98%)

                                                                                                                                       Complication in 7 (6%) of the
                                                                                                                                      participants (95% CI: 2%, 13 %)
To evaluate the       Kotlow classification          Polysomnography         Laser frenectomy and 32 female and male                    SG: increased in the scores: Fioravanti et al.,
efficacy of lingual   Ruffoli classification               NRS                   Myofunctional        pediatric patients (4-13            Kotlow (p < 0.001), MAB        2021 13
frenectomy to             (only for pre-                                    exercises to perform at          years old)               (p < 0.01), MOTTIP (p < 0.001)
improve the length of       treatment                                             home (SG)             SG: 16 patients (7              and Protrusion (p < 0.001).
                          assessment)                                          Myofunctional and       females, 9 males); 8           SG: 93.8% mild OSAS and 6.2%
frenulum and severity
                                                                              speech therapy (CG) (50%) had severe OSAS                    moderate OSAS at T1
of OSAS in pediatric           MAB                                                                     and 8 had moderate
patients.                    MOTTIP                                         Follow-up: T0, T1 after        OSAS (50%)                 CG: significant increase in all of
                               Protrusion                                          3 months             CG: 16 patients (7            the scores: Kotlow (p < 0.001),
                             (by means of                                                            females, 9 males); 3 had            MAB (p < 0.01), MOTTIP
                            Quick Tongue Tie                                                        severe OSAS (18.8%) and              (p < 0.01) and Protrusion
                           Assessment Tool®)                                                          13 had moderate OSAS                       (p < 0.01).
                                                                                                              (81.2%)                 CG: 18.75% mild OSAS, 62.5%
                                                                                                                                       moderate OSAS and 18.75%
                                                                                                         Kotlow classification               severe OSAS at T1.
                                                                                                         with class III-IV-V by       No between-groups significant
                                                                                                           Kotlow; Grades              differences in the Kotlow and
                                                                                                          2 and 3 by Ruffoli           Quick Tongue Tie Assessment
                                                                                                                                        Tool® quantitative variables
 To compare                      Kotlow             PRES and REVT to           Simple frenotomy   Out of 37 patients, 19 Before surgery, abnormal tongue-                       Kim et al.,
 surgical outcomes            classification        assess the speech             or the 4-flap    underwent the 4-flap   tie classification was observed,                       2020 26
 of simple frenotomy                                  impairment of             Z-frenuloplasty   Z-frenuloplasty and 18   but after surgery, all the cases
 and the 4-flap                                          children.             performed by the  (age 3.52 ± 0.69 years, except 1 case in frenotomy and
                                                    U-TAP to evaluate            two surgeons     13 males, 6 females),   3 cases in frenuloplasty groups
 Z-frenuloplasty
                                                       the accuracy                                the simple frenotomy      were classified as “normal”.
 according to the                                      of consonant          Follow-up: 3 months (age 4.66 ± 2.05 years,     When compared before and
 articulation test                                    pronunciation              after surgery     10 males, 8 females)      after the surgery, improved
 values and tongue-                                                                                                         tongue-tie classification have
 tie classification                                                                               Kotlow classification: been confirmed. No significant
 in patients with                                                                                      greater than          differences between groups
 ankyloglossia                                                                                       Classification 1         (p > 0.99). Changes in the
 with articulation                                                                                                          articulation test (consonants)
 difficulty                                                                                                                  were statistically significant
                                                                                                                             in both groups (p < 0.001).
                                                                                                                          Most patients improved speech
                                                                                                                               articulation. No significant
                                                                                                                         difference in outcomes between
                                                                                                                                the two surgical groups
                                                                                                                                        (p = 0.28)
NRS: numerical rating scale; MAB: maximum opening of the mouth; MOTTIP: distance between the interincisal margins with the tip of the tongue positioned at the level of the superior
retroincisive papilla; PRES: Preschool Receptive-Expressive Language Scale; REVT: Receptive and Expressive Vocabulary Test; U-TAP: Urimal Test of Articulation and Phonation; SG:
Study Group; CG: Control Group; sEMG: surface electromyography; protrusion: distance between the tip of the protruded tongue and the incisal margin; OMT: orofacial myofunctional
therapy; N.S.: not significant; *Calculated by the authors; no statistical analysis was given in the text. In italic and in bold type are respectively given the follow-up stages and the tongue/
frenulum assessment classification before the release.




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                                                                                        Impact of tongue-tie release on assessment tools




not only found an improvement in Kotlow classification          remnant of the tissue between the ventral surface of the
degrees (from 3-4 to 0-1 after intervention) in 13 patients     tongue and the floor of the mouth 26,28. When it interferes
treated with frenuloplasty and orofacial myofunctional ther-    with normal function, the condition is called “symptomatic
apy, but also found a significant (p < 0.05) increase in sur-   ankyloglossia” 26. The anatomy of the lingual frenulum is
face electromyography (sEMG) potentials over the follow-        poorly described in literature 29,30. Although a recent study
up during clenching and swallowing and a decrease in the        illustrated how it develops from concurrent muscular and
sEMG activity in masseter and anterior temporalis during        mucous components 31, there are no publications providing
protrusion of the mandible 24. Sfasciotti and colleagues ran-   relevant additional details on the physiological morphol-
domly divided 125 patients into 5 groups according to the       ogy. Thus, it is hard to understand how pathological vari-
delivery of frenuloplasty, speech therapy and positioning       ables can determine functional limitations of the tongue in
of a sterile iodoformic gauze affixed with suture and found     children, young patients and adults 32.
that the Kotlow and MOTTIP scores significantly improved        Due to the variety of symptoms related to different func-
during follow-up only in patients undergoing frenuloplasty      tional limitations, a multidisciplinary approach, involving
(p < 0.001) 25. Patients who were delivered the gauze posi-     otolaryngologists, pediatricians, speech pathologists and
tioning and did not receive speech therapy had lower MAB        dental specialists, is advocated. Although its existence has
post-surgical scores compared to the others (p < 0.001)         been acknowledged for centuries 33, the clinical implica-
and patients who did not receive any treatments had sig-        tions of diagnosis and management remain controversial 34.
nificant (p < 0.001) greater values of protrusion within 2      Given the existence of several classification systems and
weeks of follow-up. Fioravanti et al. implemented the same      various diagnostic criteria 35 (none of which are used as a
assessment tools for pre- and post-treatment stages with        clear reference), indications for treatment remain unclear.
polysomnography in 32 patients randomly undergoing              Indications for general surgical treatment include feeding
frenuloplasty and home-delivered myofunctional exercises        difficulty and speech impairment 36 and speech therapy by
(n = 16) or myofunctional and speech therapy (n = 16) 13.       speech pathologists as well as surgical intervention are the
Both groups significantly improved during follow-up along       most commonly reported treatment modalities 26.
all the tongue-related scales, without significant between-     All these aspects are further complicated by the presence of
group differences. The former group had a higher number         all those conventional problems caused by limited move-
of mild OSAS cases post-intervention with respect to the        ment of the tongue which have been frequently pinpointed
latter group in which a higher numbers of moderate and          for both the indication and/or follow-up of interventional
severe OSAS cases were found. Kim et al., in 2020, com-         programmes of tongue tie division, often in absence of
pared the surgical outcomes of simple frenotomy and the         an anatomical landmark of the frenulum and tongue posi-
4-flap Z-frenuloplasty according to the tongue-tie classi-      tion 37-39. In this vision, the strict inclusion criteria applied
fication and speech impairment test values 26. They found       in the present scoping review highlighted that – among all
significant improvement in the within-subjects compari-         the classifications (mostly qualitative) used in the literature
sons of frenulum scale and speech impairment; however,          to evaluate the indication and resolution rate of tongue-tie
no surgical procedure demonstrated an advantage in terms        division 6,16,40,41 – the following quantitative classifications
of improving outcomes.                                          were most widely used for both inclusion and follow-up
Finally, Saccomanno et al. associated the lingual frenulum      strategies: Kotlow classification (normal ≥ 16 mm), Mar-
protocol by Marchesan with postural evaluation using spi-       chesan classification, the MOTTIP (normal ≥ 23 mm),
nometry before and after laser frenectomy and orofacial         MAB (normal MY ≥ 35 mm) and the lingual protrusion.
myofunctional therapy in 24 patients 14. They found that the    In different studies, additional classifications were used to-
intervention induced a significant lingual frenulum proto-      gether 13,25 (Tab. II).
col score reduction (p < 0.001), but not significant improve-
ment of shoulder opening during maximum mouth opening           Changes in Kotlow classification
and tongue to palate with mouth open conditions.                Collectively, studies implementing the Kotlow classifica-
                                                                tion – which stages the ankyloglossia as more severe in
                                                                relation to the distance of the tip of the tongue–lingual in-
Discussion                                                      sertion of the frenulum 42 – demonstrated that frenectomy
Ankyloglossia, also known as tongue-tie, is a congenital        associated with rehabilitation induced a decrease in the se-
oral anomaly characterised by an abnormally short lingual       verity of this classification, passing from stage 3-4 to 0 or
frenulum. The lingual frenulum, according to the Interna-       1 with respect to a matched group of patients not undergo-
tional Affiliation of TongueTie Professionals, is a midline     ing any type of treatment 24. Furthermore, this reduction –



                                                                                                                                  497
M. Arena et al.




which appeared after 21 days of follow-up – was mostly           with respect to all other groups 25 and to be equally benefit-
similar to patients who also underwent rehabilitation in as-     ted by surgical/rehabilitation or only rehabilitation 13.
sociation with surgery, but better than those who received
a sterile iodoformic gauze at the end of the intervention 25.    Consequences on other dysfunctions
Interestingly, after 28 days of follow-up no differences         Given the notions speculating limitations in tongue move-
were found when comparing patients only undergoing sur-          ments are involved in different impairments of speech 5-7,
gery or rehabilitation. This result was also confirmed by        swallowing and chewing, alterations of the correct coor-
another study by Fioravanti and colleagues demonstrating         dination of muscles 8,9, oral respiration 10, forward tongue
that both types of intervention induced a significant reduc-     positioning with possible malocclusions 11,12, OSAS 13 and
tion in the Kotlow classification in two groups of OSAS          postural consequences 14, some authors have evaluated
patients and that no between-group differences were found        some of these descending consequences with regards to the
during follow-up 13. When comparing the effect of simple         tongue-tie division. Underpinnings of these alterations are
frenotomy or the 4-flap Z-frenuloplasty on two groups            thought to be related to adaptation of muscles when tongue
of patients with tongue-tie Kotlow classification > 1 and        function is restricted by a tongue-tie 24. Since the tongue
speech impairment, Kim and colleagues demonstrated that          cannot function as it is supposed to, other muscles have
both techniques significantly improved frenulum length           to help. This leads to a whole cascade of compensations
and, consequently, speech ability in both groups of par-         and adaptations throughout the stomatognathic apparatus,
ticipants with no between-groups effect 26. Finally, Kotlow      involving the reciprocal relationships between intrinsic
classification was found to be significantly improved in a       and extrinsic tongue muscles 24. The former behaves as a
cohort study by Ferrés-Amat et al. in which a large sample       scaffolding by which the intrinsic muscles can be moved
of young participants – in the absence of a control group –      around in the oral cavity, while the latter are continuously
underwent frenectomy with rhomboidal plasty associated           modifying their dimension and contour 24,46. Conversely,
with myofunctional training 1.                                   when a tongue-tie is surgically treated, the patient has no
                                                                 muscle memory of how to use his/her tongue without the
Changes in Marchesan protocol                                    restriction there; since the brain takes time to rewire itself,
The Marchesan protocol – which associates qualitative            rehabilitation can aid in this functional re-education 24. In
anatomical and functional parameters of the tongue with a        this regard, a rehabilitation protocol – together with fren-
quantitative ratio of the measurements of the distance from      ulectomy – was held by Tecco et al. to be pivotal in re-
superior to inferior incisive in with open mouth wide with or    patterning the muscles, ensuring full range of motion of
without the tongue tip touching the incise papilla 27,43 – was   the tongue, and making sure the tissues do not re-attach
found to be improved in a study by the same author involv-       after surgery 47. Over 6-months of follow-up, this is as-
ing 10 participants undergoing frenectomy; it was demon-         sociated with significant changes in the sEMG potentials
strated that the surgical procedure also induced benefits in     of the masticatory and sub-mental muscles in some tasks,
terms of protrusion, shape of the tongue tip, mouth opening      particularly during maximal voluntary clenching, kissing,
and speech 23. Using the same multiparametric diagnostic         and swallowing 24. In line, also an improvement – even if
protocol, Saccomanno and co-workers demonstrated – in            not significant – in term of shoulder opening during mouth
the absence of a control group – that laser frenectomy and       opening and tongue to palate with mouth open when stud-
rehabilitation treatment led to significant reduction in the     ied with spinometry was demonstrated by Saccomanno
lingual frenulum score and changes (even if not significant)     et al. 14, possibly reflecting restoration of the above-men-
in postural parameters 14.                                       tioned neuromuscular cascades induced by the tongue-tie
                                                                 along the tonic-postural system 14,48. According to the lit-
Changes in complementary assessment tools                        erature regarding involvement of tongue-tie in OSAS de-
Finally, three tests were used by the group of Sfasciotti        velopment 17,49, in the study by Fioravanti and co-workers
et al. 25 and Fioravanti et al. 13 with the aim to complete      patients undergoing laser frenectomy and myofunctional
the Kotlow classification with the additional information        exercises had more improvement compared to those un-
included in the Marchesan protocol: i.e. MOTTIP, MAB             dergoing only myofunctional exercises when staging their
and lingual protrusion. These tests, previously used in other    OSAS after 3 months of follow-up, although no between-
studies 44,45, were demonstrated to improve, especially in       group differences in tongue and frenulum assessment were
the first weeks of follow-up, in patients undergoing surgical    found 13. This aspect is relevant since it highlights the im-
tongue-tie without sterile iodoformic gauze positioning at       portance of anatomical and functional restoration when
the end of the intervention with or without speech therapy       tongue-tie is involved in the development of OSAS. In-



498
                                                                                               Impact of tongue-tie release on assessment tools




deed, in these cases the altered lingual posture determines         need of rigorous implementation in research and clinical
a different direction of growth of the jaw bones which can          practice of validated tongue assessment tools both before
lead to orofacial dysmorphia with a reduction in the influx         and after frenulum release. This would have the purpose of
of incoming air with consequent respiratory problems 5,50.          better correlating improvements with benefits on functional
Therefore, it decreases the size of the upper airway support        limitations, standardising results across different research
and progressively increases the risk of upper airway col-           groups and facilitating the transfer of clinical knowledge
lapse during sleep inducing OSAS 5,49, for which the study          between those involved in the diagnostic process of tongue-
by Fioravanti et al. demonstrated that successful treatment         tie and its release.
should involve anatomical release supported by a myofunc-           For future perspectives, the results of the present scoping
tional therapy, beyond the impact on tongue and frenulum            review stress the importance of implementation of oral and
assessment tools 13. Finally, following evidence showing a          extra-oral functional tests which could clinically integrate
benefit of tongue-tie release on speech impairment 51, Kim          anatomical assessment of the tongue and frenulum and the
et al. demonstrated that simple frenotomy and the 4-flap Z-         utility of a multidisciplinary approach that can merge sur-
frenuloplasty equally impacted speech impairment tests 26.          gical and rehabilitative approaches. Following these direc-
In parallel with a similar effect on the post-operative Kot-        tions and the lessons learned by the bias of the selected
low classification improvement, the study reinforced the            study protocols, more clear and suitable assessment tools
idea that limited tongue tip mobility is an underpinning            devoted to thorough assessment of the quantitative and
factor contributing to speech impairment 51 and that its            qualitative consequences of tongue-tie and its release on
simple anatomical release may be useful in reversing the            both oral and extra-oral dysfunctions are needed.
alteration.
                                                                    Conflict of interest statement
Final remarks and future perspectives                               The authors declare no conflict of interest.
When taking into account the “lens” through which the pre-
sent scoping review selected the original studies, it has to be     Funding
noted that some relevant studies were excluded due to the           This research did not receive any specific grant from fund-
lack of a rigorous evaluation of the tongue and the frenulum        ing agencies in the public, commercial, or not-for-profit
before and after the tongue-tie release (see for example 37-39).    sectors.
This aspect assumes relevance i) when in other studies a
certain degree of dyscrasia was demonstrated between                Author contributions
tongue/frenulum assessment tools and functional tests (see          AM, MAr, MA: conceptualization; MAr, AM, ADB, MA:
for example the studies by Saccomanno et al. 14 and Kim
                                                                    methodology; MAr, MA, AM, ADB, FG: formal analysis;
et al. 26) and ii) when – on the other hand – the recognised
                                                                    AM, MAr, MA, DJ: data curation; AM, MAr, BM, IM,
assessment tools have to be considered the reference land-
                                                                    ADB, MA, BM: writing-original draft preparation; MAr,
mark for follow-up, also of disorders related to the tongue-
                                                                    AM, ADB, MA, AC, IM: writing-review and editing; MA,
tie (see for example Tecco et al. 24). Furthermore, the stud-
                                                                    DJ, AM, AC, BM, FG: supervision. All Authors have read
ies included – besides rigorously following the tongue-tie
                                                                    and agreed to the published version of the manuscript.
release with the assessment tools –suffer in some cases
from limitations in terms of small sample size 23 and/or lack
                                                                    Ethical consideration
of a control group 1,14,23, size differences between the study
                                                                    N/A.
and control groups (see for example 13,26) and different tim-
ing of the first follow-up (from 24 hour in Ferres-Amat et
                                                                    Informed consent
al. 1 to 3 months in Kim et al. 26 and Fioravanti et al. 13). All
                                                                    N/A.
these aspects – together with the different treatment options
proposed – make interpretation of the impact of tongue-tie
division on tongue and frenulum assessment equivocal and            References
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