The Laryngoscope
Lippincott Williams & Wilkins, Inc.
© 2007 The American Laryngological,
Rhinological and Otological Society, Inc.
How I Do It
Management of Thyroglossal Duct Cysts
After Failed Sistrunk Procedure
Kim Hewitt, MD; Ted Pysher, MD; Albert Park, MD
INTRODUCTION
Dr. Walter Sistrunk introduced the surgical tech-
nique of resection of the central hyoid bone and central
core of the tongue base for the treatment of thyroglossal
duct (TGD) cysts.1 This technique has greatly reduced the
recurrence rate of these lesions from greater than 50% to
3% to 5%.2 The current challenge is the management of
the occasional recurrent lesion.
Significant morbidity will result when these cysts
recur. Persistent cysts will cause neck swelling, mucopu-
rulent drainage, associated fever and pain, and potentially
carcinoma.3 We report on our experience in treating four
patients with recurrent TGD cysts (Table I).
CASE REPORTS
Patient 1 developed a tender mass at the midline of
the neck approximately 10 days prior to presentation. A
computed tomography scan of the neck revealed a 4 !
4-cm mass just to the left of the midline. A presumptive
diagnosis of an infected TGD cyst was made, and he was
admitted to the hospital for intravenous antibiotics. He
did not require an incision and drainage procedure. One
month later, he underwent a Sistrunk procedure in which
the central hyoid and a 1-cm cuff of the base of the tongue
were removed. Within 1 month, he developed a recurrent
neck mass with mucoid drainage on aspiration. A revision
procedure was performed in which a 2- to 3-cm cyst was
noted in close approximation to residual hyoid bone. The
mass was carefully dissected with a small ("0.5 cm) cuff of
normal tissue from the surrounding cervical contents. He-
mostasis was meticulously maintained to optimize visual-
From The University of Utah Medical Center, Division of Otolaryn-
gology, Head and Neck Surgery (K.H., A.P.); Primary Children’s Medical
Center, Department of Pathology (T.P.), Salt Lake City, UT, U.S.A.
Editor’s Note: This Manuscript was accepted for publication Decem-
ber 11, 2006.
This material was presented as a poster at the Combined Otolaryn-
gology Spring Meeting (COSM), American Society of Pediatric Otolaryn-
gology (ASPO) Section in Chicago, IL, USA, May 20, 2006.
Send correspondence to Albert Park, MD, Division of Otolaryngol-
ogy, 50 N Medical Drive, 3C120, Salt Lake City, UT 84132. E-mail:
Albert.Park@intermountainmail.org
Laryngoscope 117: April 2007
756
ization of the surgical site. Since granulation tissue may
result from an inflammatory reaction to retained epi-
thelium, this tissue was resected with the mass. As the
dissection proceeded superiorly, the hypoglossal nerve
had to be identified and followed into the genioglossus
muscle to facilitate removal of the cyst. Additional hyoid
bone was removed in continuity with the cyst. A frozen
section of the base of the tongue showed only skeletal
muscle without any respiratory epithelium. This pa-
tient has not had another recurrence with a follow-up of
3 years.
Patient 2 was first seen at 5 months of age and had
an anterior neck mass just slightly to the right of the
midline of the neck for 1 month without signs of infec-
tion. He underwent a Sistrunk procedure in which a
1.5-cm segment of the central hyoid and a 1-cm segment
of the base of the tongue were removed. Approximately
1 month postoperatively, he developed drainage and
recurrent swelling of the neck. At reexploration, a pal-
pable mass was carefully dissected away from any sur-
rounding normal tissue. Granulation tissue was again
left on the specimen, and meticulous hemostasis was
followed. Superior dissection resulted in identification
of the left hypoglossal nerve and an epithelial tract just
inferior to the nerve. This tract was traced into the base
of the tongue. A sample of the base of tongue deep to the
tract was taken and sent for frozen section analysis. It
was positive for respiratory epithelium; thus, additional
tissue was taken from the base of the tongue. The pa-
tient has had no further recurrence after 2 years.
DISCUSSION
The original Sistrunk procedure, introduced in the
1920s, involved dissecting the cyst to the hyoid, removing
the central portion of the hyoid and a core of the tongue
base. A finger is placed transorally onto the foramen ce-
cum to facilitate removal of the tongue base component.
Thus, portions of the hyoid, mylohyoid, geniohyoid, and
genioglossus are removed with the thyroglossal duct.1
Despite an adequately performed Sistrunk proce-
dure, recurrence of a TGD cyst may occur. One possible
Hewitt et al.: Management of Thyroglossal Duct Cysts
 TABLE I.
Patients With Recurrent Thyroglossal Duct (TGD) Cysts.
Patient No. 1 2 3 4

Age/sex 8-year-old M 5-month-old M 11-year-old F 9-year-old M

Presentation 10 days of tender, erythematous
neck mass at midline
Initial treatment Sistrunk done 1 month later
Outcome Recurred within 1 month,
revision Sistrunk done
Area of recurrence In close approximation to
residual hyoid
Revision Left hypoglossal nerve
technique dissected, frozen section of
BOT negative
Outcome No recurrence at 3 years
CT # computed tomography; BOT # base of tongue.
1-month history of neck mass
at midline
Sistrunk procedure
Recurred within 1 month,
revision Sistrunk done
Tract just below hypoglossal
nerve and into BOT
Wide excision including thyroid
isthmus, dissecting
hypoglossal nerve into BOT.
Frozen section of BOT
positive, more tissue taken
into oropharynx
No recurrence at 2 years
Draining neck mass at Draining anterior neck
midline, CT scan mass
report “atypical” for
TGD cyst
Simple excision Sistrunk procedure
including skin
Recurred within 2 Recurred within 1
months, Sistrunk week, revision
done Sistrunk done
BOT Tract into the BOT and
granulation tissue
around hyoid
After Sistrunk, frozen Bilateral hypoglossal
section of BOT was nerve dissections.
positive, further Frozen section of
BOT taken into BOT negative
oropharynx
No recurrence at 3 No recurrence at 1
years year

reason is that the initial procedure was not wide enough to
include multiple, laterally directed, or branching tracts.
Horisawa and colleagues performed anatomical recon-
structions of thyroglossal duct cysts from 10 patients
and reported multiple branches and secretory glands
merging into a single duct at the hyoid.4 They also found
that the duct might run posterior to the hyoid but not
through the bone (Fig. 1). We will typically palpate the
lesion and dissect a cuff of normal tissue with the spec-
imen. Any granulation tissue will be kept with the
specimen, since it may represent an inflammatory reac-
tion to adjacent retained epithelium. Strict hemostasis
of the surgical field with loupe magnification helps to
optimize visualization.
Several descriptions of extending the Sistrunk proce-
dure—taking an additional core of the tongue base and
performing a central neck dissection— have been sug-
gested to prevent or treat recurrent lesions.5,6,7 We found
identification of the hypoglossal nerve to be an important
step in three patients (Fig. 2). The nerve was found to be
in close proximity to the recurrent mass as it entered into
the genioglossus muscle. Unless the nerve is dissected
along this region, one may leave residual tumor or inad-
vertently sacrifice this important structure.

Fig. 1. This drawing illustrates the po-

tential branching of ductuli off the main
duct as it ascends from the hyoid be-
tween the two hypoglossal nerves to
the base of the tongue.

Laryngoscope 117: April 2007 Hewitt et al.: Management of Thyroglossal Duct Cysts
757

Fig. 2. A photograph taken after a revision thyroglossal duct cyst
excision. The white arrows are pointing to the hypoglossal nerves.
We found that intraoperative evaluation of suspi-
cious regions via frozen section to be useful. In two
patients, residual respiratory epithelium was identified
and further tissue was resected (Figs. 3 and 4). In the
other two, no remnant tissue was identified, which pro-
vided more confidence in an adequate resection.
Unfortunately, TGD cysts may recur despite appar-
ent adequate removal of the tongue base and hyoid as
advocated by Sistrunk. We have had success utilizing
intraoperative frozen section analysis and, in some situa-
tions, dissection of the hypoglossal nerve for preservation
Fig. 3. Slide of a frozen section from patient #2, stained with hema-
toxylin and eosin (H&E) and photographed at 40!. The slide shows
a tract lined by pseudostratified ciliated columnar epithelium with
chronic inflammation in the wall.
Laryngoscope 117: April 2007
758
Fig. 4. Another frozen section sample, photographed at 40! and
stained with H&E showing the adjacent mucous glands with appar-
ent connection of the glands to the tract.
TABLE II.
Approach to a Recurrent Cyst of the Thyroglossal Duct.
1. Excise prior skin incision including fistula, if present.
2. Elevate superior and inferior skin flaps.
3. Dissect lesion from surrounding normal tissue by maintaining
meticulous hemostasis. Keep a cuff of normal tissue ("0.5 cm)
and any granulation tissue on the specimen for optimal
resection.
4. Inferior dissection may require removal of the thyroid isthmus.
5. Superior dissection may require identification of the
hypoglossal nerve as it enters the genioglossus.
6. Remove a central 1 cm core of tissue from the base of tongue.
7. Clamp and transect the base of tongue tissue within a cm of
the foramen cecum. It is not necessary to resect mucosa.
8. Take a frozen section sample of the base of tongue deep to
your transaction site to confirm absence of epithelial tissue.
of this important structure and for adequate tumor resec-
tion near the genioglossus muscle. Table II summarizes
our approach to these difficult-to-treat lesions.
BIBLIOGRAPHY
1. Sistrunk WE. Technique of removal of cysts and sinuses of the
thyroglossal duct. Ann Surg 1920;71:121–124.
2. Pelausa ME, Forte V. Sistrunk revisited: a 10-year review of
revision thyroglossal duct surgery at Toronto’s Hospital for
Sick Children. J Otolaryngol 1989;18:325–333.
3. Ashurst AP, White CY. Carcinoma in an aberrant thyroid at
the base of the tongue. JAMA 1925;85:1219 –1220.
4. Horisawa M, Niinomi N, Ito T. Anatomical reconstruction of
the thyroglossal duct. J Pediatr Surg 1991;26:766 –769.
5. Sattar AK, McRae R, Mangray S, Hansen K, Luks FI. Core
excision of the foramen cecum for recurrent thyroglossal
duct cyst after Sistrunk operation. J Pediatr Surg 2004;39:
e3– e5.
6. Kim MK, Pawel BR, Isaacson G. Central neck dissection for
the treatment of recurrent thyroglossal duct cysts in child-
hood. Otolaryngol Head Neck Surg 1999;121:543–547.
7. Patel NN, Hartley BE, Howard DJ. Management of thyroglos-
sal tract disease after failed Sistrunk’s procedure. J Lar-
yngol Otol 2003;117:710 –712.
Hewitt et al.: Management of Thyroglossal Duct Cysts