Pediatrics International (2019) 61, 1020–1024 doi: 10.1111/ped.

13955

Original Article

Thyroglossal duct cyst: Factors affecting cosmetic outcome and

recurrence

Andrea Batazzi, Stella Leng, Marco Ghionzoli, Roberto Lo Piccolo, Alessandra Martin, Flavio Facchini and
Antonio Messineo
Department of Pediatric Surgery, Children’s Hospital A. Meyer, Florence, Italy

Abstract Background: Thyroglossal duct cyst (TDC) is the most common congenital abnormality in the neck in children.
The purpose of this study was to perform a comprehensive review of all cases of TDC surgically treated at a single
institution and to evaluate the factors that influence the rate of recurrence, and the aesthetic outcome of the surgery
on follow up.
Methods: All cases of TDC surgically treated at the Department of Pediatric Surgery at Meyer Hospital from Jan-
uary 2005 to December 2016 were selected. Charts from 248 patients were reviewed and risk factors for recurrence
evaluated. A questionnaire was submitted to the patients’ parents to determine if postoperative complications were
present and standardized neck pictures were requested, to evaluate the cosmetic result. Microsoft Office Excel 2007
for Windows and Graphpad Prism 6 were used for data management and statistical analysis.
Results: Simple cyst excision and post-inflammatory fibrosis (P < 0.05) were assessed as important risk factors for
the recurrence of TDC. Recurrence rate on Sistrunk procedure was 5%. Variables such as post-inflammatory fibro-
sis before surgery (P < 0.001), the positioning of a drain (P < 0.01) and the development of recurrence (P < 0.001),
negatively influenced the cosmetic result. No thyroglossal duct cyst carcinoma and no long-term postoperative com-
plications were observed.
Conclusions: Recurrence rates were higher in patients who underwent simple cyst excision instead of the Sistrunk
procedure, as already reported in literature. Presence of post-inflammatory fibrosis and positioning of the drain at
surgery were associated with higher rates of recurrence, as well as worse cosmetic outcome.

Key words cosmetic, inflammation, recurrence, scar, thyroglossal duct cyst.

Thyroglossal duct cyst (TDC) is the most common congeni-
tal abnormality in the neck, accounting for approximately
75% of midline neck swellings in children. TDC originates
from persistent epithelial remnants of the thyroglossal duct,
which, during embryogenesis, guides the descent of the thy-
roid gland from the foramen cecum to its final position in
the anterior neck. After the thyroid gland descends, the thy-
roglossal duct remnants usually disappear; their persistence
may give rise to a cyst.1–5
Most cases of TDC are reported through infancy. The usual
presentation is a midline, non-tender, painless palpable mass
that moves at swallowing. Additional symptoms are dysphagia
and cough.6,7
Reported diagnostic investigations include ultrasound (US),
radioisotope scanning and thyroid function tests. Although US
is usually sufficient to confirm the presence of the cyst and to
Correspondence: Antonio Messineo, MD, Department of Pediatric
Surgery, Children’s Hospital A. Meyer, Viale Pieraccini 24,
50100 Firenze, Italy. Email: antonio.messineo@meyer.it
Andrea Batazzi and Stella Leng contributed equally to the study.
Received 29 September 2018; revised 9 April 2019; accepted
24 May 2019.
rule out an ectopic thyroid, it is often unable to differentiate
between TDC and dermoid cyst.1–3,6,8–11
To date, surgery represents the best treatment for TDC. The
primary rationale for surgery in TDC is to avoid complications
associated with chronic infection, such as fibrosis.3,12–17
The simple cyst excision, historically, had a recurrence rate
of 50%. Given this high rate of recurrence, new procedures
were developed. With the introduction of Schlange’s technique
in 1893, in which the central portion of the hyoid bone is
removed as well, the recurrence rate fell to 20%. Successively,
in the 1920s, the Sistrunk procedure was introduced, with a
recurrence rate ≤15%. The Sistrunk procedure consists of
removing the central portion of the hyoid bone and a portion
of duct between the hyoid bone and the foramen cecum.
Minor postoperative complications of Sistrunk’s procedure
include superficial surgical site infection, seroma and abscess
around the sutures. They are associated with very low morbid-
ity and can be easily managed in an outpatient setting. Risk of
major complications following Sistrunk’s procedure is low:
airways, major vessels and nerve injury can be minimized by
identifying key anatomical landmarks and using meticulous
surgical technique. Therefore, more attention to the cosmetic

© 2019 Japan Pediatric Society


outcome of TDC treatment was given: new treatment
approaches such as sclerotherapy using several solutions (e.g.
OK432, ethanol) have been proposed to achieve better cos-
metic results while minimizing surgical risk.18–21
The purpose of the present study was to evaluate factors
that influence the recurrence rate and cosmetic outcome of
surgery, on follow up.
Methods
All cases of TDC surgically treated at the Department of Pedi-
atric Surgery at Meyer Hospital from January 2005 to December
2016 were selected using ICD-9 codes. The data were obtained
from the hospital’s electronic medical records following IRB
approval from Local Ethical Committee. During these 12 years,
248 patients with TDC underwent a total of 270 surgical proce-
dures. The electronic register provided the patient’s gender, date
of birth, date of procedure, and type of procedure.
Data on patient age at the time of the first surgery, recur-
rence rate, number of surgeries per patient, presence of post-
inflammatory fibrosis, and use of drainage, was also revised.
Histopathology of the surgical specimens was also obtained.
Data on the patients who developed recurrence were also
analyzed to study the effect of different variables on the risk
of recurrence: age at surgery, presence of post-inflammatory
fibrosis, type of procedure, and placement of drainage. Post-
inflammatory fibrosis was defined as fibrosis occurring as a
result of repeated infections in the TDC.
Early postoperative complications ≤1 month were beyond
the aim of our study.
The present study also focused on the cosmetic results of the
scar after surgery. After acquiring an appropriate informed con-
sent, a total of 151 families were successfully contacted and
agreed to answer the questionnaire. The questionnaire was
emailed and when email was not possible, a phone message was
sent. The questionnaire consisted of two questions: (i) has your
child had any persistent symptoms or problems related to surgery;
if yes, what; and (ii) after the last operation at our center, did the
child undergo any other treatment or did the child received any
further treatment at other centers; if yes, please specify. We also
asked for photos of the scar from three perspectives (front, right
side, left side), attaching some example pictures.
a b c d
Fig. 1 Representative examples of thyroglossal duct cyst scar cosmetic score (TSCS): (a) grade 1, bad; (b) grade 2, sufficient; (c) grade
3, fair; (d) grade 4, good; (e) grade 5, excellent.
Cosmetic outcome and recurrence in TDC 1021
Seventy-five families (30%) gave a complete response,
sending all the answers to the questionnaire and photos of the
scar. Eleven gave a partial response, answering the questions
but not sending the photos, while 65 did not respond at all.
Scar photos of each patient were evaluated by a team of
five pediatric surgeons from a cosmetic standpoint and each
one gave a score. The score assigned is based on the Hamil-
ton scale. This scale is specific for the photographic assess-
ment of scars, evaluating their thickness, regularity,
vascularity and color. After evaluating each factor individu-
ally, we added an extra step: we calculated the total score.
The total score using the Hamilton scale can range from 0 to
a maximum of 14. For the assessment of TDC, we decided
to create a scale that divides the Hamilton total score into
five grades ranging from 1 to 5: the thyroglossal duct cyst
scar cosmetic scale (TSCS). Hamilton score 12–14 corre-
sponds to grade 1 of TCSC; 9–11, to grade 2; 6–8, to grade
3; 3–5, to grade 4; and 0–2, to grade 5. Grade 1 refers to a
bad aesthetic result of the scar; grade 2, sufficient result;
grade 3, fair result; grade 4, good result; and grade 5, an
excellent result (Fig. 1). The average score for each patient
was calculated and its relationship with the following vari-
ables was investigated: gender, age at surgery, years since
surgery, presence of post-inflammatory fibrosis, use of drain,
further interventions in addition to the first one, and develop-
ment of recurrence.22
Statistical analysis
Microsoft Office Excel 2007 for Windows and Graphpad
Prism 6 were used for data management, statistical analysis
and to create figures. Significance was calculated using the
Fisher’s exact test and Student t-test. P < 0.05 was considered
statistically significant.
Results
Demographic data
Two hundred and seventy surgical procedures were carried out
in 248 TDC patients from January 2005 to December 2016. A
total of 126 patients (51%) were male and 122 (49%), female.
e
© 2019 Japan Pediatric Society
1022 A Batazzi et al.

Neither sex was significantly associated with TDC. Most *

patients developed TDC at ≤5 years of age (62%), in line with
a
the literature.1–5 Age at surgery ranged from 2 months to 16
150
years, with an average of 5 years. A total of 188 of 248 chil- No fibrosis
dren (76%) underwent surgery at ≤5 years of age, while 60
(24%) were treated at >5 years of age.7 Fibrosis
No long-term postoperative complications and no further 100
treatment in other centers were reported in the questionnaire
given to the patients’ family.
In every operation, the excised material was sent for
50
histopathology: all specimens were negative for carcinoma.
Fibrosis
No fibrosis
Recurrence
0

Eighteen patients (7%) had TDC recurrence after surgery.
Four of these patients (2%) developed a second recurrence
and required a second operation. Time between first surgery
and development of recurrence was variable (range, 2–55
months; mean, 14 months). Of the 18 patients who devel-
oped a first recurrence, 14 (77%) developed it at ≤5 years
of age, while four (23%) were aged >5 years. The recur-
rence rate in younger patients was 7.4%, while older
patients reported a recurrence rate of 6.6%. Age at surgery
was not found to significantly influence the risk of recur-
rence (P > 0.05).
Regarding the effect of post-inflammatory fibrosis on the
risk of recurrence, in the present series of 248 patients, 112
had signs of post-inflammatory fibrosis: although in 98 of
them (87.5%) the first surgery was successful, 14 of them
(12.5%) developed a recurrence. Of the 136 children who did
not have post-inflammatory fibrosis: 132 of them (97%) did
not develop any recurrence; and four (3%) developed one.
Post-inflammatory fibrosis was found to significantly increase
the risk of recurrence (P < 0.05; Fig. 2a).
As for the influence of drain placement after surgery, in the 52
patients in whom a drain was used: 48 (92%) did not develop any
recurrence, while four (8%) developed one. In the patients in
whom a drain was not used: 182 (93%) did not develop any recur-
rence; and 14 (7%) developed one. Drain placement was not found
to influence the risk of recurrence (P > 0.05).
In 234 patients (95%) the Sistrunk procedure was carried
out, with 13 developing a recurrence (5%). In 14 patients (5%),
a simple cyst excision without removal of the body of the hyoid
bone was performed, with five developing a recurrence (35%;
Fig. 2a). Simple cystectomy was performed in these patients
because there was no apparent relationship between the thy-
roglossal duct and the hyoid bone. Simple cyst excision was
associated with a higher rate of recurrence (P < 0.05; Fig. 2b).
Cosmetic outcome
The TCSC score of the 75 patients who sent photos was stud-
ied with regard to several variables. The mean TCSC score
was 3.1, with the lowest score at 1, and the highest at 5.
The patients examined consisted of 35 girls and 40 boys.
The average score for the female patients was 3. In the male
No. patients
*
b
250 Sistrunk
200
150
100
50
Sistrunk
Cyst excision
Cyst excision
0
Recurrence No recurrence
Fig. 2 Recurrence (18 cases) in thyroglossal duct cyst patients
according to (a) presence of fibrosis (, yes; , no) and (b) type of
procedure performed (, Sistrunk procedure; , cyst excision). *P <
0.05.
group, one child (2.5%) was not evaluated because of inade-
quate picture quality; the average score was 3.2. No signifi-
cant difference between the sexes was seen from a cosmetic
standpoint.
The relationship between cosmetic outcome of the scar and
age at surgery (considering the last surgery in the case of recur-
rence) was evaluated. Patients were divided into three groups:
preschoolers, schoolers and adolescents. Preschoolers (aged <6
years) had an average score of 3.1; schoolers (aged 6–12 years),
an average score of 3.1; and adolescents (aged >12 years), an
average score of 3.3. Age at surgery was not found to signifi-
cantly influence the cosmetic outcome (P > 0.05).
The relationship between cosmetic outcome and number of
years since operation (considering the last surgery in the case
of recurrence) was established. Patients were divided into two
groups according to the number of years since surgery. Group
1 consisted of 17 patients who underwent surgery in the
previous 3 years (from beginning of 2013 to 2016) with an
average score of 3.6; group 2 consisted of 58 patients who

© 2019 Japan Pediatric Society


Fig. 3 Thyroglossal duct cyst scar cosmetic score (TSCS) vs (a) years since surgery, (b) presence of post-inflammatory fibrosis, (c)
placement of drain and (d) recurrence. *P < 0.05; **P < 0.01.
underwent surgery ≧3 years ago (before 2013) with an aver-
age score of 3. This relationship was statistically significant
(P < 0.05; Fig. 3a).
Presence of post-inflammatory fibrosis, drain and develop-
ment of recurrence was linked to worse cosmetic results. Four-
teen patients (19%) with post-inflammatory fibrosis received
an average cosmetic score of 2.3. Conversely, 61 patients
(81%) without post-inflammatory fibrosis had an average
score of 3.3 (P < 0.01; Fig. 3b). In 19 cases (25%), a drain
was placed. These patients received an average score of 2.4.
In the 56 patients (75%) in whom a drain was not positioned,
the average score was 3.4 (P < 0.01; Fig. 3c). Eight patients
(10%) who developed recurrence received an average score of
1.9, while 67 patients (90%) who did not develop recurrence
received an average score of 3.3 (P < 0.01; Fig. 3d). The dif-
ference was statistically significant for all these variables.
Discussion
Thyroglossal duct cyst is the most common congenital abnor-
mality in the neck. Diagnosis is simple and carried out
through clinical evaluation and US. Sistrunk’s procedure rep-
resents the treatment of choice, with complications being a
very rare occurrence. In the present study, no permanent com-
plications were reported.
No cases of carcinoma were found in the present series.
This rare carcinoma is usually diagnosed in adult patients,
Cosmetic outcome and recurrence in TDC 1023
mainly in the fourth decade of life, with isolated cases occur-
ring during childhood. Therefore, all cases of TDC at the
present center were excised and histologically analyzed, to
diagnose this carcinoma, in the rare chance of its occurrence
at an early age.11
Although new surgical and medical techniques are emerging,
Sistrunk procedure offers the best risk : benefit ratio, with a
recurrence rate of 5%, as confirmed in the present study. Recur-
rence risk was not influenced by gender, age at surgery, or drain
placement, while the simple cyst excision technique and pres-
ence of post-inflammatory fibrosis were identified as the main
risk factors for TDC recurrence. These findings confirm previ-
ous retrospective studies. Knowledge of these risk factors can
allow better surgical management of TDC patients: precautions
can be taken to lower the recurrence rate: such as performing
Sistrunk’s procedure instead of simple cyst excision, whenever
possible, and to avoid post-inflammatory fibrosis by treating
TDC soon after diagnosis. At the present center, TDC patients
who were not treated promptly upon diagnosis had worsened
cosmetic outcome, given the associated higher risk of post-in-
flammatory fibrosis. To lower this occurrence, in the past 3
years (2013–2016) we chose to intervene at diagnosis, and the
results confirm the positive effect of our approach both on recur-
rences and on cosmetic outcome.23,24
As more attention has been focused on cosmetic outcome,
factors contributing to scar healing have been studied in detail.
These can be divided into local and systemic factors. Local
© 2019 Japan Pediatric Society
1024 A Batazzi et al.
factors include oxygenation and infection, while systemic fac-
tors are many and include age, sex and use of anti-inflamma-
tory medications.25
The present study has identified several risk factors affect-
ing the cosmetic result of the scar: presence of post-inflamma-
tory fibrosis, drain positioning, and recurrence. This may be
explained by the fact that post-inflammatory fibrosis, persis-
tence of a foreign body in the skin, and reoperation may inter-
fere with wound healing, resulting in worse scarring.
Knowledge of these factors can help in the correct manage-
ment of TDC. Prompt indication to surgery allows better heal-
ing and reduces recurrence rates, because it avoids fibrosis
due to chronic inflammation of the cyst if left untreated. The
decision of whether a drain is truly necessary is important,
because this interferes with skin integrity and may further hin-
der wound healing, with some authors considering it altogether
unnecessary and useless in preventing complications associ-
ated with Sistrunk procedure.26
In conclusion, the Sistrunk procedure should be performed in
a timely manner in children, as soon as TDC is detected, thus
avoiding post-inflammatory fibrosis, which may result in a
higher recurrence rate and worse cosmetic outcome.
The risk factors that negatively influence scar cosmetic
outcome were identified as post-inflammatory fibrosis,
positioning of a drain after surgery, and development of
recurrence.
Given that the cosmetic appearance of the scar after sur-
gery is of paramount importance, a proper knowledge and
management of these risk factors are necessary to achieve a
better cosmetic outcome.
Disclosure
The authors declare no conflict of interest.
Author contribution
A.B. performed the statistical analysis and drafted the manu-
script; S.L. critically reviewed the manuscript; M.G. and
A.Ma. designed the study; R.L.P., F.F., and A.Me. collected
data. All authors read and approved the final manuscript.
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