Int. J. Oral Maxillofac. Surg.

2015; 44: 119126

http://dx.doi.org/10.1016/j.ijom.2014.07.007, available online at http://www.sciencedirect.com

Systematic Review
Clinical Pathology

Clinical presentation and

treatment outcomes of
thyroglossal duct cysts: a
systematic review

F. M. Gioacchini1,
M. Alicandri-Ciufelli1, S. Kaleci2,
G. Magliulo3, L. Presutti1, M. Re4
1

Otolaryngology Department, University

Hospital of Modena, Modena, Italy;
2
Department of Diagnostic Medicine, Clinical
and Public Health University Hospital of
Modena, Modena, Italy; 3Department of
Otorhinolaryngology G. Ferreri, La
Sapienza University, Rome, Italy;
4
Otorhinolaryngology Department, Marche
Polytechnic University, Ancona, Italy

F.M. Gioacchini, M. Alicandri-Ciufelli, S. Kaleci, G. Magliulo, L. Presutti, M. Re:

Clinical presentation and treatment outcomes of thyroglossal duct cysts: a systematic
review. Int. J. Oral Maxillofac. Surg. 2015; 44: 119126. # 2014 International
Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights
reserved.

Abstract. The aim of the present review was to analyze the main clinical signs and
symptoms observed in patients with thyroglossal duct cysts (TGDCs). Secondarily
we investigated the outcomes following the different types of treatment of TGDCs
in children and adults. Three selected strings were run on the PubMed database to
retrieve articles on these topics. A double cross-check was performed on citations
and full-text articles were identified using the study inclusion and exclusion criteria.
A meta-analysis was performed of the data obtained. Overall, 356 articles were
identified; 24 (comprising a total of 1371 subjects) satisfied the inclusion and
exclusion criteria. On the basis of the meta-analysis, the presence of a neck cystic
mass was the main clinical presentation of TGDCs, with a mean rate of 75% (95%
confidence interval 7279%). The mean local wound infection rate was 4% (95%
confidence interval 36%), this being the most frequent complication following
treatment. The mean rate of overall recurrence was 11% (95% confidence interval
914%). The Sistrunk procedure appears to be the better choice for the therapy of
TGDCs to avoid recurrences. Further studies on larger cohorts of patients regarding
the minimally invasive treatment options would be helpful to elucidate and endorse
their utilization in selected cases.

In head and neck embryology, the thyroid

gland descends into the neck as the lateral
lingual swellings meet in the midline to
form the tongue. During its migration, the
middle portion of the gland remains attached to the foramen cecum at the base of
the tongue by the thyroglossal duct. This
0901-5027/010119 + 08

connection tracks anteriorly to the hyoid

bone and usually atrophies around the
tenth week of gestation. Incomplete obliteration of the duct gives rise to thyroglossal duct cysts (TGDCs), which are the
most common congenital neck mass, with
a 7% population prevalence.1 They gener-

Key words: thyroglossal duct cysts; Sistrunk

procedure; complications; recurrences.
Accepted for publication 14 July 2014
Available online 15 August 2014

ally present in infants or adolescents, but

since they are frequently asymptomatic,
some individuals are diagnosed with this
defect at age 20 years or older. Both sexes
are equally affected.2
The cyst usually presents as a painless,
slightly mobile, asymptomatic soft mass,

# 2014 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

120

Gioacchini et al.

and most cysts lie in the midline close to

the hyoid bone. However, they can be
located at any site along the pathway of
descent of the thyroid anlage, thus making
the diagnosis of TGDC more difficult.3 A
variant of TGDC may also be found in the
tongue and not present in the neck. These
are often discovered incidentally and have
been termed lingual TGDCs.4
The Sistrunk procedure is the standard
treatment for TGDCs in many institutions
and consists of the resection of the midline
portion of the hyoid bone along with a
wide core of tissue belonging to the midline area between the hyoid and foramen
cecum. Nevertheless, a number of minor
and major complications can be ascribed
to surgical mishaps, and the risk of recurrence has not been eliminated.5 For these
reasons, several authors have proposed
variants of the Sistrunk procedure,68
new medical devices to perform this operation,5 and even alternative types of
treatment.4,911
The objectives of this systematic review
of the literature were to analyze the main
presenting signs and symptoms of TGDCs
and secondarily to focus on the outcomes
and possible complications following the
different techniques adopted for the treatment of this pathology.
Materials and methods

The PRISMA (Preferred Reporting Items

for Systematic Reviews and Meta-Analyses) 2009 guidelines were considered and
applied whenever possible in this systematic review. In May 2013, a literature search
was performed using the following three
search strings in PubMed: (1) (Thyroglossal Cyst[MeSH]) AND Signs and
Symptoms[MeSH]; (2) Thyroglossal
Cyst[MeSH] AND (complications[Subheading] OR Postoperative Complications[MeSH] OR Intraoperative
Complications[MeSH]); (3) Thyroglossal Cyst[MeSH] AND (Recurrence
[MeSH] OR Neoplasm Recurrence,
Local[MeSH]).
The initial search returned a total of 356
results (22 obtained after running the first
string, 241 after running the second string,
and 93 after running the third string).
Abstracts and titles obtained were
screened independently by two of the
authors (FMG and MR) who subsequently
met and discussed disagreements on citation inclusion.
Inclusion criteria for citations were (1)
cohort of patients larger than five elements; (2) English language. The exclusion criterion was clear unrelated
pathologies.

Of the 356 articles, 29 met the initial

inclusion criteria according to both
authors (FMG and MR); these were thus
obtained and reviewed in detail by the
same two authors, who met and discussed
disagreements on article inclusion. Inclusion criteria for full text articles and single
patients identified were (1) confirmed histological diagnosis of TGDC; (2) cohort of
adult population; (3) cohort of juvenile
population. Exclusion criteria were (1)
reports including cases of TGDC thyroid
carcinoma; (2) lack of sufficient information defining exactly the type of treatment
used.
A total of seven studies were excluded
because there were insufficient data on
outcomes (three studies) and treatment
techniques (four studies). A further manual check was performed on the references
included in the articles and two additional
studies were identified that met the inclusion criteria through a review of references
and a concurrent PubMed search. The final
articles included in the present review
were identified (N = 24), and the main
information was extracted and summarized.
By contacting the first authors of five of
the studies included in this review by email (Dr Perkins, Dr Hirshoren, Dr Zhang,
Dr Simon, Dr Kim), we were able to
obtain some missing data which were
not reported within these studies.
Statistical methods

Our primary objective was to evaluate the

possible clinical presentations of TGDC.
The secondary objective was to investigate the different treatment complications
and the recurrence rate of the pathology.
We performed a non-comparative metaanalysis, and the heterogeneity between
studies was assessed by x2-based
Cochrans Q statistic test and I2 metric.
Heterogeneity was considered statistically
significant at P < 0.01 for the Q statistic (to
assess whether observed variance exceeds
expected variance). For the I2 metric
(I2 = 100%  (Q df)/Q), the following
cut-off points were used: I2 = 025%, no
heterogeneity; I2 = 2550%, moderate heterogeneity; I2 = 5075%, large heterogeneity; I2 = 75100%, extreme heterogeneity.
All analyses were performed using Comprehensive Meta-Analysis statistical software, version 2.0 (Biostat, Englewood, NJ,
USA).
Results

After an initial check, full-text retrieval,

and manual cross-checking of references

included in the articles, 24 studies, comprising a total of 1371 subjects, clearly met
the inclusion criteria and were selected for
analysis (Fig. 1). The characteristics of
these selected studies are included in
Table 1.
The majority of the studies were performed with a retrospective cohort design,
although three were prospective. The average length of follow-up was reported in
only 14 studies (range 1.5125 months);
the overall average length of follow-up
was 38.7 months.
Overall, the number of patients in each
study included in this analysis varied from
6 to 231. The age of the cohorts varied
from a mean age of 3 years to a mean age
of 46 years. The vast majority of patients
had cervical TGDCs, while an intralingual
localization was described in 39 cases.
Concerning treatment modalities (Table
1), of the 1371 patients, 1239 (90.4%)
underwent the Sistrunk procedure, either
classical or modified, 73 (5.3%) had a
simple cystectomy, 19 (1.4%) an endoscopic transoral excision, 17 (1.2%) received ethanol sclerotherapy, 17 (1.2%)
OK-432 therapy, and 6 (0.4%) the puncture method.
Clinical presentation and symptoms were
described in 16 articles including a total of
1015 patients (Table 2). We analyzed the
mean rate of different presentations and
symptoms separately: for cervical cystic
mass, the mean rate was 0.75 (95% confidence interval (CI) 0.720.79) and results
were heterogeneous and statistically significant (I2 = 86.7%; P = 0.000); for fistula/
draining sinus, the mean rate was 0.18
(95% CI 0.150.22) and results were heterogeneous and statistically significant
(I2 = 78.1%; P = 0.001); for cervical infection/abscess, the mean rate was 0.34 (95%
CI 0.310.37) and results were heterogeneous
and
statistically
significant
(I2 = 92.0%; P = 0.001); for dysphagia,
the mean rate was 0.09 (95% CI 0.07
0.11) and results were moderately homogeneous and statistically significant (I2 =
69.8%; P = 0.001); for airway obstruction,
the mean rate was 0.06 (95% CI 0.030.09)
and results were heterogeneous and statistically significant (I2 = 0.0%; P = 0.001).
The number of treatment complications
could be obtained for 22 articles including
1230 patients (Table 3). The rate of total
complications occurring in the 22 studies
included is illustrated in Fig. 2. Results
were homogeneous and statistically significant (I2 = 46.5%; P = 0.009). The mean
rate of overall complications was 0.08%
(95% CI 0.060.10%). We analyzed
the single subgroups of complications
separately: for seroma, the mean rate was

Thyroglossal duct cysts

Overall 356 records
identified and screened
through database search

Full text articles assessed for

eligibility (n = 29)

121

Records excluded for following

reasons (n = 327):
- Unrelated pathologies
- Language other than English
- Limited cohort of patients

Full-text articles excluded (n = 7)

for following reason:
- Records without sufficient
clinical information about types
of treatment and outcomes

Additional records
identified through
manual search (n = 2)
Studies included in
qualitative synthesis
(n = 24)

Fig. 1. Flowchart of the review process.

0.03 (95% CI 0.010.04) and results were

homogeneous and not statistically significant (I2 = 24.6%; P = 0.143); for local
wound infection, the mean rate was 0.04
(95% CI 0.030.06) and results were homogeneous and not statistically significant
(I2 = 0.0%; P = 0.738); for haematoma, the
mean rate was 0.01 (95% CI 0.010.03) and
results were homogeneous and not statistically significant (I2 = 0.0%; P = 0.949); for
salivary fistula, the mean rate was 0.01
(95% CI 0.010.02) and results were homogeneous and not statistically significant
(I2 = 0.0%; P = 0.938); for hypothyroidism, the mean rate was 0.01 (95% CI
0.010.02) and results were homogeneous
and not statistically significant (I2 =
0.0%; P = 0.967); for airway stenosis with
stridor, the mean rate was 0.02 (95% CI
0.010.03) and results were homogeneous
and not statistically significant (I2 = 0.0%;
P = 0.553).
The number of complications occurring
after the Sistrunk procedure (classical or
modified) and simple cystectomy was 72/
1171 overall, while it was 2/59 after the
other treatments (Table 4).
The 14 studies reporting a definite follow-up time were assessed to analyze the
total number of recurrences (Table 5). The
rate of overall recurrence described in the
14 studies included is illustrated in Fig. 3.
Results were moderately homogeneous
and statistically significant (I2 = 59.5%,
P = 0.002). The mean rate of recurrence
was 0.11 (95% CI 0.090.14). We also
analyzed the recurrence rates in the different subgroups of patients separately: for
the Sistrunk operation, the mean rate of

recurrence was 0.06 (95% CI 0.040.09)

and results were moderately homogeneous
and statistically significant (I2 = 52.2%;
P = 0.003); for simple cystectomy, the
mean rate of recurrence was 0.53 (95%
CI 0.370.69) and results were moderately
homogeneous and not statistically significant (I2 = 64.1%; P = 0.062); for alternative techniques, the mean rate of
recurrence was 0.01 (95% CI 0.100.
32) and results were homogeneous and
not statistically significant (I2 = 0.0%;
P = 0.624).
Discussion

In the literature, there are few data regarding the incidence of symptoms in patients
with TGDCs. In 16 of 24 articles included
in this review, comprising a total of 1015
patients, we were able to obtain the exact
rates of major presenting symptoms. Classically, the most common presentation of
TGDCs is a painless cystic mass in the
region of the hyoid bone at or near the
midline.26 On the basis of our meta-analysis of the 1015 patients included in this
study, a cervical cystic mass was the main
clinical presentation.
However, although most thyroglossal
duct remnants present as cystic masses,
data in the literature state that up to onequarter of these lesions present as a
draining sinus tract in the midline.26
Conversely, we found a slightly lower
rate of patients who presented with a
fistula or draining sinus at clinical examination. Moreover, unusual presentations such as severe respiratory distress

or sudden infant death syndrome are

described for thyroglossal duct remnant
lesions at the base of the tongue.26 In our
analysis, some cases of airway obstruction were reported, being noticeably
more frequent in patients presenting with
intralingual TGDCs.
Because of their anatomical association
with the oral cavity, TGDCs are prone to
infection. Preoperative infection is the
most common complication of TGDCs,
with rates ranging from 10% to 70%1;
the most common pathogens responsible
include Haemophilus influenzae, Staphylococcus aureus, and Staphylococcus epidermidis.26 In the 1015 subjects examined
in this review, cases of preoperative infection represented an important rate.
Another frequent symptom reported in
our series was dysphagia. Moreover, although not reported in Table 2, some rare
cases of cough, globus, and hoarseness
were described within the 16 articles we
reviewed.
From our research, the Sistrunk procedure (either classical or modified) is the
mainstay of treatment for TGDCs. Although it is known to be a relatively safe
operation, there is, at present, a paucity of
reports in the literature reviewing the complications associated with this surgical
procedure. In 1986, Bennett et al.12 published a report analyzing the outcomes of
64 patients who had undergone operations
for TGDCs at the University of Oklahoma.
Data were inclusive of all techniques
employed (Sistrunk procedure, Schlange
procedure, simple cystectomy) and a 20%
complication rate was reported, these

122

Table 1. Main characteristics of the selected studies.

Simple
cystectomy

Endoscopic
transoral
excision

Ethanol
sclerotherapy

OK-432
therapy

Puncture
method

Authors [Ref.]

Year

Bennett et al. [12]

1986

Retrosp.

64

12.4

57

Josephson et al. [19]

Ewing et al. [20]
Maddalozzo
et al. [13]
Marianowski
et al. [14]
Brousseau
et al. [15]
Turkyilmaz
et al. [17]
Perkins et al. [6]
Shah et al. [8]
Mondin et al. [18]

1998
1999
2001

Retrosp.
Retrosp.
Retrosp.

69
11
35

21.5
35.2
4

64
11
35

5
0
0

0
0
0

0
0
0

0
0
0

0
0
0

2003

Retrosp.

57

57

2003

Retrosp.

62

32

59

2004

Retrosp.

27

6.2

27

2006
2007
2008

Retrosp.
Retrosp.
Retrosp.

231
29
14

4
6
36.2

202
29
14

29
0
0

0
0
0

0
0
0

0
0
0

0
0
0

2008
2009
2009

Retrosp.
Retrosp.
Retrosp.

84
16
155

6.1
3
10.9

71
0
155

13
0
0

0
16
0

0
0
0

0
0
0

0
0
0

2009
2011

Retrosp.
Retrosp.

9
106

N/A
28

0
97

0
9

3
0

0
0

0
0

6
0

Salgarelli
et al. [5]
Ahmed et al. [7]
Zhang et al. [23]

2011

Retrosp.

12

28

12

2011
2011

Retrosp.
Retrosp.

38
7

5.4
24

38
7

0
0

0
0

0
0

0
0

0
0

Kim et al. [10]

Ohta et al. [9]

2011
2012

Prosp.
Prosp.

11
17

39.4
36.4

0
0

0
0

0
0

11
0

0
17

0
0

Chow et al. [11]

2012

Prosp.

46

Simon and
Magit [1]
Hussain et al. [24]

2012

Retrosp.

120

5.4

120

2013

Retrosp.

83

6.1

76

Ubayasiri
et al. [25]

2013

Retrosp.

108

21

108

Total, n (%)

1371 (100%)

1239 (90.4%)

73 (5.3%)

Retrosp., retrospective design; Prosp., prospective design; N/A, not available; mo, months; y, years.

19 (1.4%)

17 (1.2%)

17 (1.2%)

6 (0.4%)

Mean
follow-up,
range
58 mo
(1 mo15 y)
N/A
N/A
1.5 mo
80 mo
(11 mo15 y)
N/A
60 mo
(213 y)
24 mo
N/A
33 mo
(285 mo)
N/A
44 mo
125 mo
(120 y)
12 mo
N/A
(248 mo)
N/A
(618 mo)
43 mo
N/A
(1831 mo)
12 mo
15 mo
(746 mo)
21 mo
(1372 mo)
N/A
14 mo
(642 mo)
N/A

Gioacchini et al.

Sistrunk
operation
(classical or
modified)

Study
design

Lin et al. [3]

Burkart et al. [4]
Hirshoren
et al. [21]
Bai et al. [16]
Ren et al. [22]

No. of
subjects

Mean age
of patients
(years)

123

Thyroglossal duct cysts

Table 2. Main clinical signs and symptoms.
Authors [Ref.]

Cervical
cystic mass

Fistula/draining
sinus

Cervical
infection/abscess

Dysphagia

Airway
obstruction

Bennett et al. [12]

Josephson et al. [19]
Marianowski et al. [14]
Brousseau et al. [15]
Turkyilmaz et al. [17]
Perkins et al. [6]
Shah et al. [8]
Mondin et al. [18]
Burkart et al. [4]
Hirshoren et al. [21]
Bai et al. [16]
Ren et al. [22]
Ahmed et al. [7]
Zhang et al. [23]
Kim et al. [10]
Simon and Magit [1]

45
46
46
30
18
204
22
14
0
155
0
96
26
0
11
119

10
7
0
1
5
1
2
4
0
47
0
12
12
0
0
0

3
7
11
26
4
18
12
0
0
109
0
32
16
0
3
49

2
9
0
9
0
0
2
0
0
7
9
17
0
0
0
3

0
0
0
2
0
0
0
0
7
0
9
0
0
1
0
1

Total, n

832

101

290

58

20

comprising mostly wound infections or

haematomas.
Concerning the articles analyzed in our
review, only Maddalozzo et al.13 focused
specifically on the possible complications
related to the Sistrunk procedure. In their
cohort of 35 paediatric patients treated for
TGDCs, no major complications were
found, while some minor complications
were observed in 29% of patients (six
presenting seroma and four local wound
infections).
Overall, with the exception of two studies (Marianowski et al.14 and Lin et al.3), we
were able to obtain at least the inclusive

number of complications after the Sistrunk

procedure (either classical or modified) and
simple cystectomy for all the articles presented in this review. In general, no major
complications were described, while a total
of 72/1171 minor complications were
found in the postsurgical period (Table 4).
The formation of seroma and local
wound infections represented the two
most frequent complications observed in
this review. Interestingly, Salgarelli et al.,5
who did not report any complications or
recurrences in the 12 patients treated,
applied the Piezosurgery medical device
(Mectron Medical Technology) in their

study. In the authors opinion, this device

works selectively on hard, mineralized
tissue alone allowing maximal cutting
precision during hyoid bone resection with
a secure preservation of soft tissues such
as the thyroid membrane, hypoglossal
nerve, and lingual artery or vein.
Among the articles in our review, Brousseau et al.15 statistically analyzed the relationship between age and the occurrence of
postsurgical complications. Even though
the complication rate was lower in adults
(7%) than in children (24%), the difference
was not statistically significant. In our
review, it was impossible to search for

Table 3. Overall treatment complications.

Authors [Ref.]
Bennett et al. [12]
Turkyilmaz et al. [17]
Maddalozzo et al. [13]
Shah et al. [8]
Ren et al. [22]
Salgarelli et al. [5]
Perkins et al. [6]
Ahmed et al. [7]
Ubayasiri et al. [25]
Brousseau et al. [15]
Mondin et al. [18]
Burkart et al. [4]
Hussain et al. [24]
Hirshoren et al. [21]
Zhang et al. [23]
Ohta et al. [9]
Chow et al. [11]
Bai et al. [16]
Kim et al. [10]
Ewing et al. [20]
Simon and Magit [1]
Josephson et al. [19]
Total, n

Seroma

Local wound
infection

Haematoma

Salivary
fistula

Hypothyroidism

Airway stenosis
with stridor

2
1
6
0
4
0
0
1
1
1
0
0
1
3
0
0
0
0
0
0
0
1

5
0
4
0
5
0
6
2
1
5
0
0
5
8
0
0
0
0
0
0
0
2

1
1
0
0
0
0
0
1
2
0
0
0
0
0
0
0
0
0
0
0
0
0

0
0
0
0
0
0
0
1
0
0
0
0
0
0
0
0
0
0
0
0
0
0

0
0
0
0
0
0
0
0
1
0
0
0
0
1
0
0
0
0
0
0
0
0

0
0
0
0
0
0
0
0
0
0
0
2
0
0
0
0
0
0
0
0
0
0

21

43

124

Gioacchini et al.

Fig. 2. Rate of overall complications after treatment for thyroglossal duct cysts.

correlations between the rate of complications and the age of patients. This was
because of the scarcity of the adult population in many of the studies analyzed in this

review, and secondly, because the presentation of data on complications was often
reported as a single modality for the adult
and child groups.

Surprisingly, from our study, only 2/59

cases of minor complications were found in
patients treated with alternative techniques. These complications were reported
by Burkart et al.4 in their retrospective
analysis of 16 patients treated with mean
endoscopic transoral excision for lingual
TGDCs. Laboured breathing and stridor
were both found during the early postsurgical period and made a second procedure
necessary for laryngomalacia and subglottic granulation, respectively.
On the other hand, no postsurgical complications were reported by Bai et al.16 in
nine patients with lingual TGDCs treated
using their puncture method (six patients)
and transoral excision (three patients). The

Table 4. Treatment complications in relation to different therapeutic options.

Sistrunk operation (classical
or modified)/simple cystectomy
Number of subjects
Seroma
Local wound infection
Haematoma
Salivary fistula
Hypothyroidism
Airway stenosis with stridor
Total complications

Alternative
techniques

1171
21
43
5
1
2
0
72

59
0
0
0
0
0
2
2

Table 5. Recurrences in relation to different treatment techniques.

Authors [Ref.]

Sistrunk operation (classical or

modified)
Total number

Recurrences

Simple cystectomy
Total number

Recurrences

Alternative techniques
Total number

Recurrences

Bennett et al. [12]

Turkyilmaz et al. [17]
Maddalozzo et al. [13]
Perkins et al. [6]
Ahmed et al. [7]
Mondin et al. [18]
Marianowski et al. [14]
Burkart et al. [4]
Hussain et al. [24]
Hirshoren et al. [21]
Ohta et al. [9]
Chow et al. [11]
Bai et al. [16]
Kim et al. [10]

52a
27
35
202
38
14
57
0
76
155
0
0
0
0

1
1
0
16
1
0
9
0
3
4
0
0
0
0

6a
0
0
29
0
0
0
0
7
0
0
0
0
0

2
0
0
18
0
0
0
0
0
0
0
0
0
0

0
0
0
0
0
0
0
16
0
0
17
6
9
11

0
0
0
0
0
0
0
0
0
0
3
1
2
3

Total, n

656

35

42

20

59

Of a total of 64 procedures the follow-up was available only for 58 patients.

Thyroglossal duct cysts

125

Fig. 3. Rate of overall recurrence after treatment for thyroglossal duct cysts.

remaining patients treated with alternative

methods underwent sclerotherapy, which
has gained popularity in the past decade
since Roh et al.27 reported a series of 12
branchial cleft cysts managed with intracystic injection of OK-432. There were no
recurrences in their series, and the use of
OK-432 sclerotherapy has expanded to
plunging ranula, lymphangioma, and
TGDC.28
Sclerotic agents, particularly OK-432,
break down the normal epithelium of the
cystic walls, causing lymphatic fluid reduction and shrinkage through fibrotic
adhesions as a result of inflammatory
reactions; this eventually causes a reduction in the size of TGDCs.29
Ethanol leads to cellular dehydration
and protein denaturation, and it causes
coagulation necrosis, small vessel thrombosis, haemorrhagic infarction, and reactive fibrosis, thus inducing regression.30
Regarding sclerotherapy, it must be noted
that this procedure can be accompanied by
local complications such as inflammation,
haematoma, vocal cord paralysis, and also
systemic effects such as fever, dizziness,
or hypersensitivity.29
In this review, no major or minor complications were observed among all 34
patients who were treated with mean minimally invasive treatment modalities such
as ethanol or OK-432 sclerotherapy and
only Ohta et al.9 reported some cases of
slight fever (37.538.5 8C) for a few days
after injection. However, in our opinion,
these findings are probably related to the
small number of sclerotherapy procedures
summarized in this review.
A major problem in the surgical treatment of TGDCs is their high frequency of
recurrence (610%), which remains a treatment challenge even when the operation is
performed by a skilled surgeon.14 For this
reason, the primary goal of surgery for

TGDCs is the complete removal of the

pathology to prevent possible recurrences,
and although a variety of surgical techniques exist, the Sistrunk procedure remains
the most widely practiced despite the increasing use of minimally invasive techniques. This technique consists of the
excision of thyroglossal tract remnants with
central hyoid excision and with a variable
amount of tongue base excision.6
The factors responsible for recurrences
comprise various entities such as dermal
involvement, young patient age (childhood), rupture of the cyst during the operation, lobulation of the cyst, inflammation
and/or infection, elimination of the coringout procedure in an attempt to follow the
suprahyoid tract, and cases with fistulas.17
According to Brown and Judd31 and
Ward et al.,32 the recurrence rate increases
after operations on younger patients; two
or more preoperative infections are also
associated with an elevated recurrence
rate.1 In our research, the studies by Marianowski et al.14 and Simon and Magit1
documented a statistically significant increased risk with a history of infection.
The rate of overall recurrence in our metaanalysis is very similar to the 10.7% rate
reported by Galluzzi et al.2 in their recent
review, while it appears higher than the 6.6%
rate of recurrence calculated by Mondin
et al.18 However, concerning the recurrence
rates observed when analyzing the different
subgroups of patients separately, our metaanalysis showed better outcomes obtained
with the Sistrunk procedure (classical or
modified), showing a mean rate of recurrence of 0.06 (95% CI 0.040.09).

often the principal manifestation, being

present in many patients affected by this
pathology. Nevertheless, otolaryngologists should be aware that, though rarer,
dysphagia may also be associated with
TGDCs. Furthermore, especially in cases
of intralingual TGDCs, a significant airway obstruction with laboured breathing
and even apnoea could represent the only
symptom reported by the patient.
Our study also highlights some important concepts regarding the treatments for
TGDCs. In fact, on analyzing the different
therapeutic possibilities described in the
literature, the Sistrunk procedure (with all
its possible variants) still appeared to be
the treatment of choice in reducing the rate
of recurrence.
Finally, regarding the alternative treatments for TGDCs described in our review,
further studies on larger cohorts of patients
would be helpful to elucidate and endorse
their utilization in selected cases.
Funding

None.
Competing interests

None declared.
Ethical approval

Not required.
Patient consent

Not required.
Conclusions

In conclusion, regarding the clinical presentation of TGDCs, this review confirmed that a cystic cervical mass is

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Address:
Federico Maria Gioacchini
Otolaryngology Department
University Hospital of Modena
Via del Pozzo 71
41100 Modena
Italy
Tel: +39 3771525135
E-mail: giox83@hotmail.com