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CaseReportsinDentistry

Volume2014(2014),ArticleID871460,6pages
http://dx.doi.org/10.1155/2014/871460
Case Report

Van der Woude Syndrome with Short Review of the


Literature
Pallavi K. Deshmukh,1 Kiran Deshmukh,2 Anand Mangalgi,3 Subhash
Patil,4 Deepa Hugar,5 and Saraswathi Fakirappa Kodangal1
Department of Oral Medicine and Radiology, H.K.E.S.s S. N.
Institute of Dental Sciences and Research, Gulbarga, Karnataka
585103, India
2
Department of Otorhinolaryngology, M. R. Medical College,
Gulbarga, Karnataka 585103, India
3
Department of Oral and Maxillofacial Surgery, H.K.E.S.s S. N.
Institute of Dental Sciences and Research, Gulbarga, Karnataka
585103, India
4
Department of Community and Preventive Dentistry, H.K.E.S.s S. N.
Institute of Dental Sciences and Research, Gulbarga, Karnataka
585103, India
5
Department of Oral Pathology and Microbiology, H.K.E.S.s S. N.
Institute of Dental Sciences and Research, Gulbarga, Karnataka
585103, India
1

Received 11 February 2014; Revised 29 April 2014; Accepted 26 May


2014; Published 22 June 2014
Academic Editor: Indraneel Bhattacharyya
Copyright 2014 Pallavi K. Deshmukh et al. This is an open access
article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any
medium, provided the original work is properly cited
Ankyloglossia or tongue-tie is a congenital abnormality of the lingual
frenulum. This entity is recognized but poorly defined condition and
has been reported to cause feeding difficulties, dysarthria,
dyspnoea, and social or mechanical problems. The exact
pathophysiology of tongue-tie is unknown. The mucosa covering the
anterior two-thirds of the mobile tongue is derived from the first

pharyngeal arch and deviation of normal development is the most


likely cause of abnormal frenulum length. One of the rare features
associated with VWS is ankyloglossia which is reported by the
authors in the present case. Ankyloglossia is a rare feature
associated with VWS and is not commonly reported in the literature.
Tongue-tie was reported by the authors in the present case [13].

J Indian Soc Periodontol. 2011 Jul-Sep; 15(3): 270272.


doi: 10.4103/0972-124X.85673
PMCID: PMC3200025

Ankyloglossia and its management


Tanay V. Chaubal and Mala Baburaj Dixit
Author information Article notes Copyright and License information
This article has been cited by other articles in PMC.

Abstract

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INTRODUCTION

Etymologically, ankyloglossia originates from the Greek words agkilos (curved) and
glossa (tongue). The same term is used for very different clinical situations: When the
tongue is fused to the floor of the mouth, but also if the lingual frenulum is only short and
thick with slight impairment of tongue mobility. The first use of the term ankyloglossia in the
medical literature dates back to the 1960s, when Wallace[1] defined tongue-tie as a
condition in which the tip of the tongue cannot be protruded beyond the lower incisor teeth
because of a short frenulum linguae, often containing scar tissue.
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CASE REPORT

A 24-year-old male was reported in the department of Periodontics with difficulty in speech
since birth. The ENT and general physical examination was normal. On intraoral
examination, it was found that the individual had ankyloglossia (tongue-tie) and was
classified as Class III by utilizing Kotlow's assessment [Figure 1] and was able to protrude
the tongue up to the lower lip [Figure 2]. There were no malocclusion and recession present
lingual to mandibular incisors. The patient was undertaken for a frenectomy procedure under
local anesthesia with 2% lignocaine hydrochloride and 1:80,000 adrenaline by using a scalpel
method; first a curved hemostat was inserted to the bottom of the lingual frenum at the depth
of the vestibule and clamped into position followed by giving two incisions at the superior
and the inferior aspect of the hemostat. This way, we removed the intervening frenum and got
a diamond shaped wound. Then with the help of the same hemostat, we released the muscle
fibers so as to achieve a good tension free closure of the wound edges [Figures
[Figures33 and and4]4] after which the wound edges were approximated with (4-0) black
braided silk sutures [Figure 5] for the tissues to heal by primary intention thereby minimizing
the scar tissue formation, antibiotic Cap. Amoxicillin (500 mg) thrice a day for 3 days and
non-steroidal anti-inflammatory drug Tab. Ketorolac DT (10 mg) thrice a day for 3 days was
prescribed to prevent post-operative infection and pain. The post-operative period was
uneventful with no delayed hemo-rrhage. Sutures were removed after 1 week [Figure 6]
which showed no scar tissue formation following which the patient was sent for speech
therapy sessions. After a follow-up of 6 months, the tongue showed good healing [Figure 7],
protrusion several mm beyond the lower lip [Figure 8], and normal speech.

Figure 1
Pre-operative view showing ankyloglossia

Figure 2
Pre-operative view showing extension of tongue

Figure 3
Frenectomy incision using scalpel

Figure 4
Completion of frenectomy

Figure 5
Sutures

Figure 6
Post-operative view 1 week

Figure 7
Post-operative view 6 months

Figure 8
Post-operative view 6 months showing adequate extension of tongue
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DISCUSSION

Ankyloglossia is an uncommon congenital oral anomaly that can cause difficulty with breastfeeding, speech articulation.[2] For many years, the subject of ankyloglossia has been
controversial with practitioners of many specialties having widely different views regarding
its significance and management. In many individuals, ankyloglossia is asymptomatic; the
condition may resolve spontaneously or affected individuals may learn to compensate
adequately for their decreased lingual mobility. Some individuals, however, benefit from
surgical intervention frenotomy, frenectomy or frenuloplasty for their tongue-tie. Patients
should be educated about the possible long-term effects of tongue-tie so that they may make
an informed choice regarding possible therapy.[2,3] The prevalence of ankyloglossia reported
in the literature varies from 0.1% to 10.7%. The prevalence is also higher in studies[4]

investigating neonates (1.72% to 10.7%) than in studies[5] investigating children,


adolescents, or adults (0.1% to 2.08%). It can be speculated that some milder forms of
ankyloglossia may resolve with growth, explaining this age-related difference. There is some
evidence that ankyloglossia can be a genetically transmissible pathology. It is unknown
which genetic components regulate the phenotype and penetrance in the patients affected.
More basic research is needed to clarify the exact etiopathogenesis of ankyloglossia.
Ankyloglossia was also found associated in cases with some rare syndromes such as X-linked
cleft palate syndrome,[6] Kindler syndrome,[7] van der Woude syndrome,[8] and Opitz
syndrome.[9] Nevertheless, most ankyloglossias are observed in persons without any other
congenital anomalies or diseases. Speech problems can occur when there is limited mobility
of the tongue due to ankyloglossia. The difficulties in articulation are evident for consonants
and sounds like s, z, t, d, l, j, zh, ch, th, dg[10] and it is especially difficult to roll an r.
Localization of the frenum insertion on the gingiva seemed to be of importance for gingival
sequelae because insertion of the lingual frenulum in the area of the papilla had the highest
association with gingival recession. The term free-tongue is defined as the length of tongue
from the insertion of the lingual frenum into the base of the tongue to the tip of the tongue.
Clinically acceptable, normal range of free tongue is greater than 16 mm. The ankyloglossia
can be classified into 4 classes based on Kotlow's assessment as follows; Class I: Mild
ankyloglossia: 12 to 16 mm, Class II: Moderate ankyloglossia: 8 to 11 mm, Class III: Severe
ankyloglossia: 3 to 7 mm, Class IV: Complete ankyloglossia: Less than 3 mm.2 Class III and
IV tongue-tie category should be given special consideration because they severely restrict
the tongue's movement. A normal range of motion of the tongue is indicated by the following
criteria: The tip of the tongue should be able to protrude outside the mouth; without clefting,
the tip of the tongue should be able to sweep the upper and lower lips easily; without
straining, when the tongue is retruded, it should not blanch the tissues lingual to the anterior
teeth; and the lingual frenum should not create a diastema between the mandibular central
incisors. Ankyloglossia limits the tongue's range of motion. Because of limited mobility of
the tongue in patients with ankyloglossia, the tongue is in a low position and causes forward
and downward pressure favoring the development of mandibular prognathism with maxillary
hypo development. The above mentioned hypothesis that ankyloglossia leads to altered
development of the jaws is mainly based on single observation and speculative interpretations
and there is limited evidence that tongue-tie represents a co-factor in the development of
malocclusions, especially Class III malocclusion. More studies, especially controlled clinical
trials, are needed to establish a clear correlation between malocclusion and ankyloglossia. If
there is no feeding difficulty in the infant, it would be best to have a wait-and-see approach
since the frenulum naturally recedes during the process of an individual's growth between six
months and six years of age. After completion of growth and also during infancy, if the
individuals have a history of speech, feeding, or mechanical/social difficulties surgical
intervention should be carried out. Therefore, surgery should be considered at any age
depending on the patient's history of speech, feeding, or mechanical/social difficulties.
Surgical techniques for the therapy of tongue-ties can be classified into three procedures.
Frenotomy is a simple cutting of the frenulum. Frenectomy is defined as complete excision,

i.e., removal of the whole frenulum. Frenuloplasty involves various methods to release the
tongue-tie and correct the anatomic situation. There is no sufficient evidence in the literature
concerning surgical treatment options for ankyloglossia to favor any one of the three main
techniques.
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CONCLUSION

To conclude, it is important to agree upon one examination method, definition and


classification of tongue-ties to enable comparisons between future observational and
intervention studies. If severe/complete ankyloglossia is present in an adult, there is usually
an obvious limitation of the tongue protrusion, elevation and speech problems which can be
improved following surgical intervention.
PENANGANAN BAYI CELAH BIBIR DAN LANGIT-LANGIT SECARA PROSTODONTIK
(PENGGUNAAN PROSTHETIC FEEDING AIDS) MAKALAH Disusun oleh: Drg. LISDA
DAMAYANTI, Sp. Pros. NIP: 132206506

TEKNIK OPERASI CELAH BIBIR DAN LANGIT-LANGIT YANG DIGUNAKAN DI


SULAWESI SELATAN PADA TAHUN 2010-2013 SKRIPSI Diajukan Kepada
Universitas Hasanuddin Untuk Melengkapi Salah Satu Syarat Mencapai Gelar
Sarjana Kedokteran Gigi SRI HARYUTI J111 10 253 FAKULTAS KEDOKTERAN GIGI
UNIVERSITAS HASANUDDIN MAKASSAR 2013

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