Original Article

Page 1 of 6

Recurrent thyroglossal duct cysts: a 15-year review of

presentation, management and outcomes from a tertiary
paediatric institution
Luke M. O’Neil1,2, Alan T. Cheng1,2,3
1
Department of Otolaryngology, Head and Neck Surgery, Children’s Hospital at Westmead, Sydney, Australia; 2Sydney Medical School, University
of Sydney, Sydney, Australia; 3Discipline of Child and Adolescent Health, University of Sydney, Sydney, Australia
Contributions: (I) Conception and design: All authors; (II) Administrative support: LM O’Neil; (III) Provision of study materials or patients: AT
Cheng; (IV) Collection and assembly of data: LM O’Neil; (V) Data analysis and interpretation: All authors; (VI) Manuscript writing: All authors; (VII)
Final approval of manuscript: All authors.
Correspondence to: Dr. Luke M. O’Neil, MBBS, MS. Department of Otolaryngology, Head and Neck Surgery, Children’s Hospital at Westmead,
Hawkesbury Road, Westmead, NSW 2145, Australia. Email: [email protected].

Background: Primary management of thyroglossal duct cysts (TGDC) is the Sistrunk procedure, which
aims to completely excise the cyst and associated duct. Recurrences are attributed to variable histopathology
and inadequate excision of the duct and its branching ductules. We present a review of the presentation and
management of recurrent TGDC over a 15-year period to assess trends and outcomes.
Methods: Retrospective review of patients who underwent surgery for a TGDC at a tertiary paediatric
hospital over a 15-year period. The following data was collected: age, sex, primary and subsequent
presentations, histopathology, primary and subsequent operation reports and speciality of the primary operator.
Results: Sixteen patients had a recurrent TGDC over the 15-year period. Six (37%) were male and 10 (63%)
were female, with a mean age of 5.3±3.1 years. Initial management included Sistrunk procedure (11 cases,
68.75%), cystectomy (4 cases, 25%) and Schlange procedure (1 case, 6.25%). The mean time to recurrence
was 20.7±26.9 months. Management of recurrences were Sistrunk (5 cases), revision Sistrunk (9 cases) or
conservative management (2 cases). Two patients had three operations, without cure.
Conclusions: Sistrunk procedure should be the primary management for TGDC. Patients who recurred
after an initial limited surgical resection may be cured with subsequent Sistrunk procedure. Revision Sistrunk
to manage recurrences following initial Sistrunk, had a high recurrence rate and two patients required two
further operative interventions, both without cure. Research into the use of en bloc anterior neck dissection
for the management of recurrent TGDC following Sistrunk in the paediatric population is suggested.

Keywords: Thyroglossal duct cyst (TGDC); thyroglossal duct sinus; recurrence; paediatric; management

Received: 09 February 2018; Accepted: 14 May 2018; Published: 02 July 2018.

doi: 10.21037/ajo.2018.05.02
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Introduction TGDC are the most common congenital midline neck

swelling in the paediatric population, with over half of all
Thyroglossal duct cysts (TGDC) arise from the incomplete cases presenting before 20 years of age (3,4).
closure of the thyroglossal tract, an integral structure The management of TGDC has evolved through a
in the embryological development of the thyroid gland. greater understanding of the embryological and histological
Thyroglossal tract remnants are reported to be present in basis of the disease. Gold standard management is the
7% of the population, however not all develop a cyst (1,2). Sistrunk procedure, which involves en bloc resection

© Australian Journal of Otolaryngology. All rights reserved. www.TheAJO.com Aust J Otolaryngol 2018;1:17
Page 2 of 6 Australian Journal of Otolaryngology, 2018

of the cyst and the thyroglossal tract, with the superior operation report review identified 191 (84%) cases of
excision encompassing the central aspect of the hyoid TGDC, 16 (7%) cases of recurrent TGDC, 13 (6%) cases
bone and continuing up to the foramen caecum, of dermoid cysts and 1 (0.4%) each for epidermal cyst, neck
without breaching the oral mucosa (5). The recurrence abscess and lymph nodes. Patient notes were unable to be
rate after Sistrunk is between 3% and 10% (6-8), located for 4 subjects.
which is a vast improvement of the recurrence rate of
more than 50% after performing an incision and drainage
Recurrent TGDC
(I&D) (9). Recurrence is attributed to an inadequate initial
resection along and/or variability in histological anatomy of Demographics
the thyroglossal tract, with small ductules extending from Sixteen patients had a recurrence after initial management
the primary thyroglossal duct. of their TGDC, of which 6 (38%) were male and 10 (62%)
Typically, patients who recur present in a similar fashion female. The patients had a mean age of 5.3±3.1 years.
to their primary presentation, however, there can be an Table 1 outlines patient information.
increased incidence of cutaneous involvement (9). The
majority of patients recur within the first postoperative Initial presentation and management
year (10). The operative goal of recurrence surgery is The initial TGDC presentation included non-infected cysts
to excise the remaining thyroglossal duct and associated (9 cases, 56%), infected cyst (2, 12%), infected cyst and
ductules. sinus (1, 6%), infected sinus (1, 6%), sinus (1, 6%) and the
In this paper, we present a review of the presentation and presentation was not documented in 2 cases (12%). The
management of recurrent TGDC over a 15-year period at a first surgical procedure of these patients included a Sistrunk
tertiary paediatric hospital to assess trends and outcomes. procedure (11 cases, 68.75%), cystectomy (4 cases, 25%)
and Schlange procedure (1 case, 6.25%). Four patients had
an incision and drainage of an infected cyst, prior to initial
Methods
cyst excision.
Institutional board review was obtained. A retrospective
chart review was performed to include all patients who Presentation of recurrence
presented to a tertiary paediatric hospital with a TGDC The mean time to recurrence was 20.7±26.9 months.
over a 15-year period. Patients were screened for Patients presented with a non-infected cyst in 7 cases (44%),
recurrences of their TGDC through additional admissions draining sinus in 7 cases (44%), cyst and sinus in 1 case
to the tertiary hospital, clinic notes, primary care (6%) and an infected cyst in 1 case (6%). Two patients had
practitioner referrals and requests for admission sent from a third recurrence after operative intervention, 1 presented
private consultant rooms. with a non-infected cyst and the other a draining sinus.
Electronic and paper records were reviewed to obtain: Eight patients (50%) presented with a sinus at recurrence,
age, sex, recurrence, primary presentation, primary of these, 3 (19%) had cutaneous involvement at initial
management, presentation of recurrence/s, length of follow presentation.
up, postoperative complications and further management.
If patients were noted to have a recurrence they were sent Management of recurrence
a questionnaire with a single question, ‘Have you/your The management of recurrences was dependent on the
child had another recurrence of a thyroglossal duct cyst or primary operative intervention. Two operations were
sinus since the last operation performed at The Children’s required for cure in 88% of patients. Eleven patients
Hospital at Westmead?’. If patients did not respond in recurred after primary Sistrunk and of these, 9 were
2 months, they were phoned. managed with revision Sistrunk and 2 were managed
conservatively. The revision Sistrunk comprised of further
dissection along the primary resection, with an extended
Results
excision of the remaining hyoid. Two patients (22%) had
Two hundred and twenty-seven operations were performed a further recurrence following revision Sistrunk, 1 was
for suspected TGDC or recurrent TGDC in paediatric managed with a further revision Sistrunk, however recurred
patients over a 15-year period. Histopathology and again, while the other had a cystectomy and also recurred.

© Australian Journal of Otolaryngology. All rights reserved. www.TheAJO.com Aust J Otolaryngol 2018;1:17
 Table 1 Presentation, management and outcomes for patients with a recurrent thyroglossal duct cyst

Complication/ Incision and

Initial 1st recurrence 2nd recurrence 3rd recurrence
Case recurrence after drainage prior Response to Total follow up
ID/Sex last operative to primary questionnaire (months)
Presentation Operation Presentation Operation Presentation Operation Presentation Operation
intervention excision

Revision
1/F Infected cyst Sistrunk Cyst Yes No recurrence 140
Sistrunk

Revision
2/M Cyst Sistrunk Cyst No No recurrence 180
Sistrunk

Revision
3/F Cyst Sistrunk Cyst No – 0.5
Sistrunk

Revision –
4/F Cyst Sistrunk Infected cyst Cyst Cystectomy Cyst Monitored No 1
Sistrunk
Australian Journal of Otolaryngology, 2018

5/M Unknown Cystectomy Cyst Sistrunk No – 0.5

Revision Revision –
6/M Cyst Sistrunk Sinus Sinus Sinus Monitored Yes 3

© Australian Journal of Otolaryngology. All rights reserved.

Sistrunk Sistrunk

7/M Unknown Cystectomy Sinus Sistrunk No No recurrence 180

8/M Sinus Sistrunk Sinus Monitored Yes No recurrence 36

Revision
9/F Cyst Sistrunk Sinus No No recurrence 120
Sistrunk

Infected Revision Post-operative

10/F Sistrunk Cyst & sinus No – 6
sinus Sistrunk wound infection

Infected Revision Post-operative

11/F Sistrunk Sinus No No recurrence 84
cyst & sinus Sistrunk wound infection

12/F Cyst Cystectomy Sinus Sistrunk No – 0.5

www.TheAJO.com
13/M Infected cyst Sistrunk Sinus Monitored Yes – 0.5

14/F Cyst Schlange Cyst Sistrunk No No recurrence 12

Revision
15/F Cyst Sistrunk Cyst No – 0.5
Sistrunk

16/F Cyst Cystectomy Cyst Sistrunk No No recurrence 12

Aust J Otolaryngol 2018;1:17

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Page 4 of 6 Australian Journal of Otolaryngology, 2018

To our knowledge, they were both subsequently managed 28 patients by Mickel et al. (9), found a similar increase
conservatively. Four patients managed with a cystectomy in risk of cutaneous involvement following failed initial
recurred and in all cases, management with Sistrunk operative interventions. The majority of patients with
procedure provided cure. One patient recurred following a recurrence have been reported to present in the first
Schlange procedure, which was successfully managed with postoperative year (10), however in our series, the majority
Sistrunk procedure. presented in the second postoperative year. Younger
children have a higher risk of recurrence, which has been
Response to questionnaire suggested to be due to a conservative initial surgical
Eight patients (50%) responded to the questionnaire, with approach, thus increasing the risk of residual thyroglossal
no recurrences reported. tract (12,13).
The gold standard surgical management of TGDC is
Follow up the Sistrunk procedure, as it offers the best chance of a
Follow up ranged from 2 weeks to 15 years, with a mean of complete excision of the thyroglossal tract and cyst. In our
48.5±68.2 months. institution, there were 11 patients who had a recurrence
following a primary Sistrunk procedure, 9 of whom
Surgical speciality directing management underwent a revision Sistrunk, with the other 2 monitored.
All patients who recurred had their initial intervention Seven (78%) of these patients had no further recurrence
performed by a paediatric General Surgeon. Fifteen patients over a mean follow up of 21.8±29.2 months. Two (22%)
(94%) had their recurrence managed by a paediatric patients required further operative interventions without
General Surgeon, while 1 patient (6%) was managed by a cure. Patients who recurred after being managed with
paediatric Otolaryngologist. a cystectomy or Schlange procedure (5 cases, 31%),
had a subsequent Sistrunk procedure with no further
recurrences. Thus, the Sistrunk procedure is a safe and
Discussion
effective operative intervention for patients who have a
TGDC recurrences are attributed to the incomplete TGDC recurrence following inadequate initial resection.
excision of the thyroglossal tract and its branching ductules. The management of recurrent TGDC following
The residual tract is present postoperatively due to variable Sistrunk procedure in children has been discussed, however
histology or a limited surgical resection. Histologically, papers are limited by patient numbers. The revision
the thyroglossal duct can have multiple ductules extending Sistrunk procedure, whereby the surgeon removes further
from the main ventral duct which may not be detected tissue along the previous resection, has been well described
intraoperatively, complicating the initial management and in the literature. A systematic review by Ibrahim et al. (14)
increasing the risk of recurrence due to residual thyroglossal documented that the Sistrunk procedure was performed
tract. Historically, incision and drainage, cystectomy and on 83 cases of recurrent TDGC in children, with 25 (30%)
Schlange procedure were performed to manage TGDC, having further recurrences. In our series, 22% of patients
however they are associated with high recurrence rates as managed with a revision Sistrunk had a further recurrence.
they do not completely excise the thyroglossal tract, thus Variations of an en block neck dissection have also been
are no longer recommended treatment options. I&D prior described in the literature, with an increased width of
to Sistrunk procedure has been associated with the need for anterolateral dissection compared to the revision Sistrunk.
multiple operations for cure (11). In our series, there were Papers are limited by patient numbers and documentation
4 patients who had an I&D prior to Sistrunk procedure. of patient details, however the results are promising
Three patients had no recurrence, however 1 patient has (9,15,16). A wide anterior neck dissection excising the
persistent disease despite 3 further operative interventions. central compartment between the lateral borders of the
Failed surgical intervention alters the presentation sternohyoid muscles has been performed in adults with
of TGDC, with recurrences having a greater cutaneous recurrent TGDC, demonstrating good outcomes with
involvement. In our series, 19% of initial TGDC minimal morbidity (17).
presentation had a draining sinus, however with recurrent The subspecialty fellowship training of the primary
TGDC, the incidence rose to 50%. A previous review of operator has been reported to impact on the recurrence

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Australian Journal of Otolaryngology, 2018 Page 5 of 6

rate, with fellowship trained otorhinolaryngologists having References

a lower recurrence rate than fellowship trained paediatric
1. Ellis PD, van Nostrand AW. The applied anatomy of
and plastic surgeons (18). In our review, all recurrences had
thyroglossal tract remnants. Laryngoscope 1977;87:765-70.
their initial intervention performed by fellowship trained
2. Mondin V, Ferlito A, Muzzi E, et al. Thyroglossal duct
paediatric general surgeons and in similar fashion to Geller
cyst: personal experience and literature review. Auris
et al. (18), a proportion of these cases had an inadequate
Nasus Larynx 2008;35:11-25.
primary resection.
3. Allard RH. The thyroglossal cyst. Head Neck Surg
Our research is limited by small patient numbers, though
1982;5:134-46.
this is expected given the low recurrence rate of TGDC
4. Maddalozzo J, Venkatesan TK, Gupta P. Complications
after Sistrunk procedure and the relatively low incidence of
associated with the Sistrunk procedure. Laryngoscope
the disease. Our patient numbers are in keeping with other
2001;111:119-23.
published articles on the subject. We are also limited by the
5. Sistrunk WE. Technique of removal of cysts and
response of patients to the questionnaire, with half of the
sinuses of the thyroglossal duct. Surg Gynecol Obstet
patients not contactable or declined participation. Given
1928;46:109-12.
the long period of time between admission and research for
6. Galluzzi F, Pignataro L, Gaini RM, et al. Risk of
this paper, many patients had changed address and contact
details. recurrence in children operated for thyroglossal duct cysts:
A systematic review. J Pediatr Surg 2013;48:222-7.
7. Hirshoren N, Neuman T, Udassin R, et al. The imperative
Conclusions of the Sistrunk operation: review of 160 thyroglossal
TGDC should be managed with Sistrunk procedure tract remnant operations. Otolaryngol Head Neck Surg
to minimse the risk of recurrence. The average age of 2009;140:338-42.
patients in our series was 5.3 years old and they typically 8. Athow AC, Fagg NL, Drake DP. Management of
recurred within the second postoperative year. Patients thyroglossal cysts in children. Br J Surg 1989;76:811-4.
had a higher risk of cutaneous involvement when a TGDC 9. Mickel RA, Calcaterra TC. Management of recurrent
recurred. Sistrunk procedure should be considered in the thyroglossal duct cysts. Arch Otolaryngol 1983;109:34-6.
management of recurrences after a limited initial surgical 10. Rohof D, Honings J, Theunisse HJ, et al. Recurrences
excision, as there were no reported recurrences in our after thyroglossal duct cyst surgery: Results in 207
series. Management of recurrence following initial Sistrunk consecutive cases and review of the literature. Head Neck
is challenging, as revision Sistrunk had a high recurrence 2015;37:1699-704.
rate and 2 (22%) patients weren’t cured despite 2 11. Perkins JA, Inglis AF, Sie KC, et al. Recurrent
subsequent excisions. Further research into en bloc anterior thyroglossal duct cysts: a 23-year experience and a new
neck dissections after failed Sistrunk in the paediatric method for management. Ann Otol Rhinol Laryngol
population is required. 2006;115:850-6.
12. Ducic Y, Chou S, Drkulec J, et al. Recurrent thyroglossal
duct cysts: a clinical and pathologic analysis. Int J Pediatr
Acknowledgements
Otorhinolaryngol 1998;44:47-50.
None. 13. Marianowski R, Ait Amer JL, Morisseau-Durand MP,
et al. Risk factors for thyroglossal duct remnants after
Sistrunk procedure in a pediatric population. Int J Pediatr
Footnote
Otorhinolaryngol.2003;67:19-23.
Conflicts of Interest: The authors have no conflicts of interest 14. Ibrahim FF, Alnoury MK, Varma N, et al. Surgical
to declare. management outcomes of recurrent thyroglossal duct
cyst in children--A systematic review. Int J Pediatr
Ethical Statement: The study was approved by the Sydney Otorhinolaryngol 2015;79:863-7.
Children’s Hospitals Network Human Research Ethics 15. Howard DJ, Lund VJ. Thyroglossal ducts, cysts and
Committee, approval number LNR/14/SCHN/181. sinuses: a recurrent problem. Ann R Coll Surg Engl

© Australian Journal of Otolaryngology. All rights reserved. www.TheAJO.com Aust J Otolaryngol 2018;1:17
Page 6 of 6 Australian Journal of Otolaryngology, 2018

1986;68:137-8. thyroglossal duct cysts in adults. J Laryngol Otol 2016;130

16. Patel NN, Hartley BE, Howard DJ. Management of Suppl 4:S41-4.
thyroglossal tract disease after failed Sistrunk's procedure. 18. Geller KA, Cohen D, Koempel JA. Thyroglossal duct
J Laryngol Otol 2003;117:710-2. cyst and sinuses: a 20-year Los Angeles experience
17. O'Neil LM, Gunaratne DA, Cheng AT, et al. Wide and lessons learned. Int J Pediatr Otorhinolaryngol
anterior neck dissection for management of recurrent 2014;78:264-7.

doi: 10.21037/ajo.2018.05.02
Cite this article as: O’Neil LM, Cheng AT. Recurrent
thyroglossal duct cysts: a 15-year review of presentation,
management and outcomes from a tertiary paediatric
institution. Aust J Otolaryngol 2018;1:17.

© Australian Journal of Otolaryngology. All rights reserved. www.TheAJO.com Aust J Otolaryngol 2018;1:17