1171784

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CPJXXX10.1177/00099228231171784Clinical PediatricsChen et al

Article
Clinical Pediatrics

Clinical Characteristics and Prognosis 1­–7

© The Author(s) 2023
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DOI: 10.1177/00099228231171784
https://doi.org/10.1177/00099228231171784

Age With Thyroglossal Duct Cyst journals.sagepub.com/home/cpj

Complicated With a Parapharyngeal

Mass: A Case Series Study

Caidi Chen1*, Fanli Liu1*, Jianjing Fang2*, Linyuan Sima1, Liyan Ni1,
Songjie Xiang1, and Siwen Xia, MD1

Abstract
Thyroglossal duct cysts (TGDCs) are congenital and developmental abnormalities in infants and young children.
This retrospective case series study examined the characteristics of 7 patients <3 years (mean age, 1.9 years)
with TGDC complicated with a parapharyngeal mass treated at one hospital between January 2019 and 2022. Four
patients had a painless mass around the neck, 2 had a painless mass associated with snoring, and 1 presented repeated
swelling and pain. B-ultrasound suggested 6 cases of TGDC and 1 possible lymphangioma. All patients were treated
with Sistrunk surgery to remove the TGDC. Six patients had no cyst recurrence during follow-up (6 months to 2
years). In conclusion, TGDC complicated with a parapharyngeal mass has complex and variable clinical manifestations.
Completely removing the cyst while sparing thyroid cartilage and surrounding vascular and neuroanatomical structures
is important to avoid complications. The patients are likely to be free from recurrence after surgery.

Keywords
infant, children, thyroglossal cyst, laryngoscopy, paracentesis, case series

Introduction can be displaced unilaterally to form a parapharyngeal

Thyroglossal duct cysts (TGDCs) are congenital and
developmental abnormalities that can occur at any age
but are most common in infants and children between
the ages of 1 and 10,1 with rare cases <1 year of age.2
Clinically, the patients often present a painless neck
mass, and the most common location is near the hyoid
bone, which is the midline neck in infants.3 In adults,
TGDC has the potential to develop associated malig-
nancy.4 It is associated with a mortality rate of 30% to
40% in neonates due to preterm delivery or sudden
infant death.5 Studies reporting the surgical treatment of
TGDCs complicated with a parapharyngeal mass in
infants and young children are scarce.
The TGDC is a cyst in the middle of the neck that
protrudes inward during growth due to the small size of
the laryngopharyngeal cavity. The resulting parapharyn-
geal mass can cause laryngeal obstruction, ranging from
snoring and stridor in mild cases to inspiratory dyspnea
in severe cases.6 In addition, the connective tissue of the
larynx is loose, and the TGDC in the middle of the neck
cyst.7 Sistrunk surgery is a common method selected for
removing a TGDC, especially for low-risk patients. The
procedure consists of the excision of the TGDC, the
middle part of the hyoid bone, and the surrounding tis-
sue around the thyroglossal tract.8,9 It involves a trans-
verse incision over the cyst or a fusiform incision over
an external fistula opening. All abnormal tissue, includ-
ing the cyst, fistula, body of the hyoid bone (preferably
1
Department of Otolaryngology Head & Neck Surgery, The Second
Affiliated Hospital and Yuying Children’s Hospital, Wenzhou Medical
University, Wenzhou, China
2
Department of Ultrasonography, Wuma Street Community Health
Service Center of Lucheng District, Wenzhou, China
*These authors contributed equally to this research.
Corresponding Author:
Siwen Xia, Department of Otolaryngology Head & Neck Surgery,
The Second Affiliated Hospital and Yuying Children’s Hospital,
Wenzhou Medical University, 109 Xueyuan West Road, Wenzhou
325027, Zhejiang, China.
Email: 199069@wzhealth.com
2 Clinical Pediatrics 00(0)
more than 15 mm), and the fibrous cord extending to the
foramen cecum, is resected. Sistrunk’s procedure is still
considered the gold standard in treating TGDCs in
infants and young children, but marsupialization and
simple cyst excision can also be performed.10,11
The surgery is usually successful in older children
and adults if inflammation is controlled,12,13 but due to
the physiological and structural differences, the failure
rate of surgery in infants and children is significantly
higher than in adults.14 Interestingly, the surgery’s suc-
cess rate and the disease’s recurrence are associated with
the anatomical structure and location of the cyst identi-
fied during the surgery.15,16 Clinical diagnosis, surgical
treatment, and postoperative precautions need in-depth
exploration.
TGDCs are uncommon in infants and young chil-
dren, and TGDCs with respiratory obstruction due to a
parapharyngeal neck mass are even rarer. Therefore, this
study aimed to examine the clinical characteristics and
prognosis of infants and children <3 years of age with
TGDC complicated with a parapharyngeal mass.
Methods
Study Design and Patients
This retrospective case series study included infants and
children <3 years of age with TGDC complicated with
parapharyngeal masses admitted to the Department of
Otolaryngology of our Hospital between January 2019
and 2022.
The inclusion criteria were (1) <3 years old, (2)
underwent classic Sistrunk surgery9,17 because conser-
vative treatment was ineffective, and (3) TGDC con-
firmed by postoperative pathological examination.
Patients with a physical examination that showed a mid-
line mass in the neck rather than a unilateral parapharyn-
geal mass were excluded.
The Ethics Committee of our Hospital approved the
study protocol. The requirement for individual consent
was waived by the committee because of the retrospec-
tive nature of the study.
Data Collection
The demographic and baseline clinical characteristics
(including age, sex, symptoms, and imaging examina-
tion results), treatment, and prognosis during follow-up
were collected from the medical records.
Statistical Analysis
SPSS 22.0 (IBM, Armonk, New York) was used for sta-
tistical analysis. Only descriptive statistics were used in
this study. The continuous data were expressed as means
± standard deviations (SD) or medians (ranges). The
categorical data were expressed as n (%).
Results
Clinical and Pathological Characteristics
Seven patients (5 males and 2 females), aged 1.9 years
(range, 10 months to 2 years 7 months), were included in
this study (Table 1). Four patients had a painless mass
around the neck, 2 had a painless mass around the neck
associated with snoring, and 1 had repeated swelling and
pain. All patients underwent B-ultrasound examination
before surgery; TGDC was confirmed in 6 patients,
while lymphangioma was suggested in 1 patient.
Preoperative computed tomography (CT) examination
was conducted on the patient with suspected lymphan-
gioma to distinguish the mass boundary; imaging sug-
gested a left anterior cervical intramuscular cystic
lesion.
All included patients underwent surgery due to con-
servative treatment failure. Six of the 7 patients under-
went surgery more than 3 months after diagnosis. One
patient was suffering from repeated neck infection and
underwent surgery 1 month after diagnosis once the
infection was under control.
Strikingly, all patients had simple TGDC without
anterior cervical fistula. Preoperative electronic laryn-
goscopy in 1 patient showed a right aryepiglottic fissure
mucosa bulge with a smooth surface. In 3 patients, mag-
netic resonance imaging (MRI) of the neck showed a
cystic mass in the neck with a clear boundary. Magnetic
resonance imaging suggested that the airway was nar-
rowed due to airway compression by the mass in 1
patient.
All patients underwent surgery because conservative
treatment was ineffective. The TGDC mass in the rear of
the tongue was removed using the traditional Sistrunk
surgery. The drainage was removed after about 3 days,
and the sutures were removed 7 days after surgery.
The patients were followed up (6 months to 2 years)
after the surgery. The neck incision recovered well, the
scars hidden in the necklines were not detected easily,
and the family members were satisfied with the results.
Only in 1 case (repeated infection of the neck mass
before surgery) did a small cyst in the middle of the
hyoid bone recur 6 months after surgery, while no cysts
were detected in the other 6 patients.
Typical Case
An 11-month-old male patient was admitted to the Second
Affiliated Hospital of Wenzhou Medical University on
 Table 1. Detailed Clinical Characteristics of the Included Cases.
Imaging examination

Electronic Cervical regional Neck computed Neck magnetic Neck mass

Characteristics Age Gender Symptoms laryngoscope B-mode ultrasonic tomography resonance imaging puncture Therapy Follow-up Outcomes

Case 1 (typical 1Y1M Female 6 months after the neck mass found √ √ √ √ √ July 15, 2021-July 20, 2021 11M No recurrence
case) with snoring
Case 2 2Y7M Male 1 month after the neck mass was / √ / √ √ December 15, 2021-December 6M No recurrence
found 19, 2021
Case 3 2Y6M Female 11 months after the neck mass was / √ / √ / September 27, 2021-September 9M No recurrence
found 30, 2021
Case 4 2Y1M Male 1 year after the neck mass was found / √ / / √ May 4, 2021-May 10, 2021 12M No recurrence
with snoring
Case 5 10M Male Half a month after neck swelling and / √ √ / √ January 3, 2021-January 7, 2021 18M Recurrence of small cyst
aggravation for 1 day in the median of hyoid
bone half a year after
surgery
Case 6 2Y5M Male 1 year after the neck mass was found / √ / / / June 2, 2020-June 8, 2020 2Y No recurrence
Case 7 2Y1M Male 2 years after the neck mass was found / √ √ / √ March 11, 2019-March 18, 2019 2Y No recurrence

Abbreviations: Y, years; M, months.

3
4 Clinical Pediatrics 00(0)

Figure 1. Typical case presentation. (A) Laryngoscopy results demonstrate a right parapharyngeal mass protruding into the
larynx. (B) Ultrasound of the neck shows a subcutaneous cystic mass of about 35 × 20 mm2 on the right side of the neck
with a distinct boundary. (C) Neck MRI (T2) showing a lobulated cystic mass of about 32 × 32 × 23 mm3 in size in the right
parapharynx with a clear boundary. The airway is narrowed due to mass-induced compression of the trachea, and the parotid
and submandibular glands are enlarged. (D) Cyst mass after complete excision. (E) Postoperative pathology examination
(hematoxylin-eosin [HE], ×200) shows a cyst wall covered with squamous epithelium and no prominent atypia; differentiated
and mature thyroid cells beside the cyst wall. (F) Neck MRI shows postoperative results.
Abbreviation: MRI, magnetic resonance imaging.

June 2021 due to a right neck mass observed for 6 months,
with aggravation and dyspnea for 1 month. Six months
ago, the family members observed a mass (about the size
of a peanut) on the right side of the neck, accompanied by
snoring and stridor, but without fever and cough. The
diagnosis in the pediatric outpatient clinic was a right
neck cyst by a combination of B-mode ultrasound, MRI,
and other auxiliary examinations. The right neck cyst was
treated with puncture and fluid extraction, which relieved
the symptoms, but the mass in the back of the neck
increased repeatedly. Thus, multiple punctures were per-
formed. During the last month before admission, the mass
had grown, and the child suffered from aggravated snor-
ing, dyspnea, and frequent awakenings.
Physical examination revealed a movable cystic mass
of about 35 × 20 × 20 mm3 in the right parapharynx,
with clear boundaries. The mass moved with swallow-
ing. Three concave signs without tenderness and other
abnormalities were observed during inhalation.
Electronic fiber laryngoscopy detected a mucosal bulge
with a smooth surface in the right parapharynx, com-
pressing the airway (Figure 1A).
A neck ultrasound at the outpatient clinic showed a
subcutaneous cystic mass of about 35 × 20 mm2 with
clear boundaries on the right side of the neck, compress-
ing the back of the trachea (Figure 1B). The neck MRI
showed a lobulated cystic mass of about 32 × 32 × 23
mm3 with a clear boundary in the front of the right
carotid artery, compressing the glottis area, leading to
the narrowing of the airway and enlargement of the
parotid and submandibular glands (Figure 1C). The
admission diagnosis was “parapharyngeal mass: sus-
pected thyroglossal cyst, Laryngeal obstruction (grade
III).”
After excluding the surgery and anesthesia contrain-
dications, the resection of the parapharyngeal mass was
Chen et al
performed under general anesthesia. During surgery,
blunt dissection was performed around the cyst. The
cyst adhered to the surrounding tissue, and its root was
around the back of the hyoid bone, indicating a TGDC.
Thus, about 1.0 cm of the hyoid bone and the root of the
cyst were removed (Figure 1D). After hemostasis, drain-
age was placed, and the incision was sutured layer by
layer. Three days later, the patient was discharged from
the hospital after removing the drainage. The postopera-
tive pathology examination confirmed the TGDC
(Figure 1E). Three months later, the clinical symptoms
were relieved, and the neck MRI showed no residual
mass (Figure 1F). No recurrence was reported during the
1-year follow-up.
Discussion
The present study showed that TGDC complicated with
parapharyngeal masses in infants and young children
has complex and variable clinical manifestations. The
mass can be punctured and aspirated when parapharyn-
geal TGDC is considered after B-mode ultrasound, CT,
or MRI. Surgery might be an alternative after conserva-
tive treatment failure. Thyroglossal duct cysts are
uncommon in infants and young children, and TGDCs
with respiratory obstruction due to a parapharyngeal
neck mass are even rarer. Their main symptoms include
dyspnea,18 laryngeal stridor,19,20 or asphyxia.6,21,22
Furthermore, the mass in the patients included in previ-
ous studies was in the midline neck,6,18,19,21,22 and unilat-
eral neck mass is even rarer, leading to a high risk for
misdiagnosis. The patients reported here underwent sur-
gery after conservative treatment failure and were con-
firmed with atypical TGDCs according to the
pathological examination. The result suggests that
patients with such characteristics and conservative treat-
ment failure should be considered for TGDC.
Thyroglossal duct cyst might exhibit no clinical
symptoms, and patients often seek medical attention due
to a neck mass.23 In this study, a physical examination
revealed a smooth, movable cystic mass in the middle of
the neck that had mobility when swallowing. In some
patients, a cord was described along the direction of the
hyoid bone, as reported previously,24 but if the structures
of the hyoid bone and other neck bones are immature,
the correlation between the hyoid bone and the mass
cannot be determined by palpation. The cases included
in this series confirmed that in infants and young chil-
dren with a unilateral parapharyngeal mass and symp-
toms of laryngeal obstruction, the possibility of a TGDC
should be considered to avoid misdiagnosis. The rela-
tively long time between diagnosis and surgical removal
could also explain why the TGDCs caused airway
5
obstruction. In the included patients, the masses were
located in the parapharyngeal space, compression of the
airway, leading to respiratory obstruction. Furthermore,
patients under 3 years old have a small airway, the con-
nective tissue in the neck is loose, and the airway is
more likely to develop edema in the presence of infec-
tion or oppression. Therefore, all 7 patients suffered
from respiratory obstruction.
In addition to the clinical manifestations and physical
examination, TGDC in infants and young children can
be diagnosed through auxiliary examinations. Neck
ultrasound, CT, and MRI are commonly used for diag-
nosing TGDC in infants and young children and for
determining the location, size, shape, nature, blood sup-
ply, and the correlation between the mass and surround-
ing tissues.11,25 In patients undergoing electronic
fiberoptic laryngoscopy, round-like bulged unilateral
laryngopharyngeal mucosa can be identified as one of
the warning signs. Still, TGDC complicated by parapha-
ryngeal masses in infants and young children lacks char-
acteristic manifestations, leading to confusion with
liquid cystic tumors that can also occur in infants and
young children. Therefore, puncture and resection are
recommended when a parapharyngeal TGDC is sus-
pected after B-mode ultrasound, CT, or MRI.
Sistrunk surgery in the external cervical approach is
one of the most used surgical approaches for TGDCs.26
Given the short neck of infants and young children and
the fact that the bone structure has not been developed
fully and the body surface landmarks are lacking, the
cervical stripe is considered the marking line of the sur-
gical incision. Typically, the first cervical stripe is
located at the level of the hyoid bone, the second cervi-
cal stripe is between the thyroid cartilage and the cricoid
cartilage, and the third cervical stripe is at the level of
the tracheal ring 3-4 above the sternum in infants and
young children. Surgery along the cervical stripe can
avoid the intraoperative loss of direction to increase the
safety of neck surgery for infants and young children
with no severe surgical scar or incision infection and
reduce the impact on facial esthetics, thus making it
acceptable to the families of patients.
Furthermore, the development speed of each system
of infants and young children is different, with the ner-
vous system developing early.27 During surgery, the
diameter of the nerves of the infants is often found to be
like that of adults, but the structures of the neck and
other tissues are not fully developed, especially the
hyoid bone, which is an anatomical marker. The hyoid
bone of the infants is slender and should be identified
carefully to locate the cyst accurately.28 Thus, the corre-
lation between the fistula and the hyoid bone should be
judged cautiously. When the boundary between the
6 Clinical Pediatrics 00(0)
fistula and the hyoid bone is distinct, the hyoid bone can
be preserved; otherwise, a part of the hyoid bone needs
to be removed.29 After the excision of the cyst, hyoid
bone resection does not affect the probability of the
postoperative recurrence of TGDC.30 In contrast, pre-
serving the hyoid bone can reduce complications and
preserve the anatomical markers. Strikingly, when the
fistula passes through the hyoid bone, the resection of
the bone should reach at least 1.0 cm, or else it can easily
rupture the cyst wall or the fistula and its branches,
resulting in recurrence. In addition, the surrounding tis-
sues should be removed along the cyst wall during the
surgery, except for patients with repeated infection
before surgery that may lead to severe adhesion.
The complications of TGDC resection mainly include
wound infection or hematoma, nerve paralysis, hypo-
thyroidism, and TGDC recurrence.31 In our patients,
postoperative complications were fewer, which was in
line with previous reports wherein TGDC resection in
infants and young children ≤3 years old did not increase
the risk of complications.32 However, infants and young
children must be prepared for the perioperative period.
This study included all cases admitted to our Hospital
between January 2019 and January 2022, but only 7
cases were operated on during this time, confirming that
few infants and young children manifested TGDC with
parapharyngeal masses. Thus, it is necessary to extend
the duration of follow-up and include additional cases to
obtain reliable conclusions in future studies.
Atypical cases characterized by a unilateral parapha-
ryngeal mass are rare with TGDC and even rarer with
respiratory obstruction. Patients with such characteris-
tics can easily be misdiagnosed as cystic hygroma or
branchial cleft cysts, which would mislead the treatment
plan. As the analysis of the cyst fluid can lead to a defin-
itive diagnosis, and since puncture and fluid extraction
would relieve the symptoms, ultrasound-guided punc-
ture extraction should be performed. Infants under 3
years of age have a risk of dyspnea or even asphyxia
because of small immature airways. If dyspnea >II
degree occurs, it can be quickly relieved by timely punc-
ture and drainage, atomization, systemic hormone, and
other symptomatic treatment. Children who fail those
conservative treatments should be treated with surgery
as soon as possible. Preoperative inflammation control
is key. Although the external cervical approach Sistrunk
surgery is the classic choice, neck localization of infants
under 3 years of age remains one of the challenges for
head and neck surgeons. In this study, it was found that
the intraoperative projection of the 3 cervical lines of
infants should be strictly followed. Carefully looking for
the hyoid bone while sparing thyroid cartilage and sur-
rounding vascular and neuroanatomical structures that
have not been fully ossified is important to completely
remove cysts and avoid complications.
In conclusion, infants and young children <3 years
of age with TGDC complicated with parapharyngeal
mass have complex and variable clinical manifestations.
Clearly establishing the diagnosis of pharyngeal mass
due to TGDC is essential for proper management, and
imaging examinations can be helpful in diagnosis.
Inflammation control is the key to management.
Children who fail conservative treatments should be
treated with surgery as soon as possible. Completely
removing the cyst while sparing thyroid cartilage and
surrounding vascular and neuroanatomical structures is
important to avoid complications. Infants and young
children <3 years of age are likely to be free from recur-
rence after surgery.
Author Contributions
CC and SX: Carried out the studies, participated in collecting
data, and drafted the manuscript. FL and JF: Performed the
statistical analysis and participated in its design. LS, LN, and
SX: Participated in acquisition, analysis, or interpretation of
data and draft the manuscript. All authors read and approved
the final manuscript.
Availability of Data and Materials
All data generated or analyzed during this study are included
in this published article.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with
respect to the research, authorship, and/or publication of this
article.
Funding
The author(s) disclosed receipt of the following financial sup-
port for the research, authorship, and/or publication of this
article: The study was supported by Fundamental Research
Project of Wenzhou City (Y20210015, Y20211064).
Ethics Approval and Consent to Participate
The Ethics Committee approved the study protocol of the
Second Affiliated Hospital of Wenzhou Medical University.
All the included infants’ parents or legal guardians signed
informed consent. All methods were performed in accordance
with the relevant guidelines and regulations.
Consent for Publication
Not applicable.
ORCID iD
Siwen Xia https://orcid.org/0000-0002-6179-7418
Chen et al
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