Mucoepidermoid carcinoma in a thyroglossal duct remnant

Abstract

Thyroglossal duct cysts (TDC) are common midline neck swellings resulting from embryological remnants of the thyroglossal duct. They often contain ectopic thyroid tissue and malignant transformation has been reported, most commonly to papillary thyroid carcinoma. Mucoepidermoid carcinoma (MEC) usually occurs in the salivary glands and only rarely in the thyroid. This is the first case of a MEC occurring within a thyroglossal duct remnant.
A 73 year old lady presented with a thyroglossal duct cyst. She declined surgical excision, as she was adamant she wanted to avoid surgery. The neck mass rapidly enlarged at two years following initial diagnosis. Fine needle aspiration cytology was suspicious for carcinoma. She underwent total thyroidectomy and selective central compartment neck dissection with adjuvant radiotherapy. She remains alive and well two years post treatment.
Mucoepidermoid carcinoma is the most common malignant neoplasm of salivary glands, although it has rarely been reported in diverse locations including the thyroid, lung and pancreas. To the best of our knowledge, this is the first reported case of mucoepidermoid carcinoma arising from a thyroglossal duct remnant.
This case adds weight to the literature favouring surgical excision of thyroglossal duct remnants due to the risk of malignant transformation.
Copyright © 2015 The Authors. Published by Elsevier Ltd.. All rights reserved.

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E.
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Mucoepidermoid
carcinoma
in
a
thyroglossal
duct
remnant,
Int
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Surg
Case
Rep
(2015),
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Mucoepidermoid
carcinoma
in
a
thyroglossal
duct
remnant
E.
Warnera,∗,
E.
Ofoa,
S.
Connorb,
E.
Odellc,
J.P.
Jeannona
Q2
aDepartment
of
Otolaryngology
and
Head
and
Neck
Surgery,
Guy’s
Hospital,
Great
Maze
Pond,
London,
SE1
9RT
England,
UK
bHead
and
Neck
Radiology,
Guy’s
Hospital,
Great
Maze
Pond,
London,
SE1
9RT
England,
UK
cHead
and
Neck
Pathology,
Guy’s
Hospital,
Great
Maze
Pond,
London,
SE1
9RT
England,
UK
a
r
t
i
c
l
e
i
n
f
o
Article
history:
Received
10
February
2015
Received
in
revised
form
20
April
2015
Accepted
26
April
2015
Available
online
xxx
Keywords:
Mucoepidermoid
carcinoma
Thyroglossal
duct
Head
and
neck
neoplasm
Salivary
gland
a
b
s
t
r
a
c
t
INTRODUCTION:
Thyroglossal
duct
cysts
(TDC)
are
common
midline
neck
swellings
resulting
from
embry-
ological
remnants
of
the
thyroglossal
duct.
They
often
contain
ectopic
thyroid
tissue
and
malignant
transformation
has
been
reported,
most
commonly
to
papillary
thyroid
carcinoma.
Mucoepidermoid
carcinoma
(MEC)
usually
occurs
in
the
salivary
glands
and
only
rarely
in
the
thyroid.
This
is
the
first
case
of
a
MEC
occurring
within
a
thyroglossal
duct
remnant.
PRESENTATION
OF
A
CASE:
A
73-year-old
lady
presented
with
a
thyroglossal
duct
cyst,
and
declined
sur-
Q3
gical
management.
The
neck
mass
rapidly
enlarged
at
two
years
following
initial
diagnosis.
Fine
needle
aspiration
cytology
was
suspicious
for
carcinoma.
She
underwent
total
thyroidectomy
and
selective
cen-
tral
compartment
neck
dissection
with
adjuvant
radiotherapy.
She
remains
alive
and
well
two
years
post
treatment.
DISCUSSION:
Mucoepidermoid
carcinoma
is
the
most
common
malignant
neoplasm
of
salivary
glands,
although
it
has
rarely
been
reported
in
diverse
locations
including
the
thyroid,
lung
and
pancreas.
To
the
best
of
our
knowledge,
this
is
the
first
reported
case
of
mucoepidermoid
carcinoma
arising
from
a
thyroglossal
duct
remnant.
CONCLUSION:
This
case
adds
weight
to
the
literature
favouring
surgical
excision
of
thyroglossal
duct
remnants
due
to
the
risk
of
malignant
transformation.
©
2015
Published
by
Elsevier
Ltd.
on
behalf
of
Surgical
Associates
Ltd.
This
is
an
open
access
article
under
the
CC
BY-NC-ND
license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
1.
Introduction
Thyroglossal
duct
cysts
(TDC)
are
common
midline
neckQ4
swellings.
Usually
presenting
in
childhood,
they
can
be
also
be
found
in
up
to
7%
of
the
adult
population
[1].
They
develop
in
rests
of
epithelium
sequestered
in
migration
during
the
formation
and
disappearance
of
the
thyroglossal
duct.
Islands
of
epithelium
may
persist
throughout
life
as
ectopic
thyroid
tissue
but
often
undergo
cystic
degeneration
to
form
thyroglossal
duct
cysts
[2].
The
com-
monest
site
for
such
cysts
is
around
the
body
of
the
hyoid
bone.
Malignant
transformation
occurs
in
1%
of
TDC
and
is
usually
of
the
papillary
carcinoma
type
[3].
There
are
two
theories
for
how
this
occurs,
either
the
tumour
arises
from
the
malignant
transformation
of
ectopic
thyroid
tissue
retained
within
the
duct,
or
arises
de-novo
from
cells
lining
the
duct
[4].
Mucoepidermoid
carcinomas
are
best
characterised
in
salivary
glands,
rarely
occurring
in
other
glandu-
lar
tissue
including
lung,
lacrimal
glands,
and
thyroid.
Of
the
40
cases
of
thyroid
MEC
in
the
literature
to
date
the
majority
are
low
∗Corresponding
author.
Current
address:
National
hospital
for
Neurology
and
Neurosurgery,
33
Queen
Square,
London,
WC1N
3BG
England,
UK.
Tel.:
+44
0
7956446486.
E-mail
address:
Elinor.warner87@gmail.com
(E.
Warner).
grade
variants,
for
example,
thyroid
sclerosing
mucoepidermoid
carcinoma
with
stromal
eosinophilia.
High-grade
mucoepidermoid
carcinomas
are
found
even
less
frequently
in
the
thyroid
gland.
This
is
the
first
reported
case
of
a
mucoepidermoid
carcinoma
occurring
within
a
thyroglossal
duct
cyst.
2.
Case
report
A
73-year-old
lady
of
Bangladeshi
origin
initially
presented
to
the
endocrine
surgeons
with
a
two-year
history
of
a
gradually
enlarging
midline
neck
lump,
which
moved
on
tongue
protrusion.
There
was
no
lymphadenopathy
or
abnormality
within
the
thyroid
gland
itself,
and
thyroid
function
tests
were
normal.
Additionally
there
were
no
suspicious
clinical
features;
she
was
a
non-smoker,
with
no
history
of
alcohol
consumption.
Clinically,
the
neck
lump
was
felt
to
be
an
uncomplicated
thyroglossal
duct
cyst.
Ultrasound
scan
showed
a
cystic
lesion
of
27
mm
in
maximal
dimension,
supe-
rior
to
and
separate
from
the
thyroid
gland
(Fig.
1),
whilst
fine
needle
aspiration
cytology
(FNAC),
revealed
features
consistent
with
a
thyroglossal
duct
cyst.
Surgical
excision
was
recommended
but
the
patient
declined.
She
agreed
to
regular
follow-up.
The
neck
mass
gradually
enlarged
over
the
course
of
12
months,
but
the
patient
still
declined
surgery.
Two
years
following
her
initial
presentation,
the
neck
mass
rapidly
http://dx.doi.org/10.1016/j.ijscr.2015.04.028
2210-2612/©
2015
Published
by
Elsevier
Ltd.
on
behalf
of
Surgical
Associates
Ltd.
This
is
an
open
access
article
under
the
CC
BY-NC-ND
license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
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Please
cite
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E.
Warner,
et
al.,
Mucoepidermoid
carcinoma
in
a
thyroglossal
duct
remnant,
Int
J
Surg
Case
Rep
(2015),
http://dx.doi.org/10.1016/j.ijscr.2015.04.028
G
Model
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xxx
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xxx–xxx
Fig.
1.
Ultrasound
images
from
initial
clinic
presentation.
A:
Midline
view
showing
central
cystic
lesion.
B:
Doppler
showing
no
evidence
of
increased
vascularity.
C:
Right
lobe
of
the
thyroid
showing
evidence
of
colloid
cyst,
otherwise
normal
appearances.
D:
Left
lobe
of
thyroid
showing
normal
appearances
of
the
gland.
Fig.
2.
CT
scan
of
the
neck
(sagittal
view).
A:
Sagittal
(non-contrast)
view
of
the
neck
showing
anterior
midline
neck
mass
extending
from
the
suprasternal
notch
to
the
hyoid
bone.
expanded
in
size
and
the
patient
was
assessed
urgently
in
the
head
and
neck
cancer
clinic.
At
this
time,
a
large
hard
mass
with
maxi-
mal
dimension
of
70
mm
was
palpable
in
the
region
of
the
thyroid
gland.
Clinically,
there
was
no
stridor
or
cervical
lymphadenopathy.
FNAC
was
highly
suspicious
for
malignancy.
A
computed
tomogra-
phy
(CT)
scan
of
the
neck
confirmed
the
presence
of
a
large
anterior
midline
neck
mass
measuring,
50
mm
×
70
mm
×
66
mm,
extend-
ing
from
the
level
of
the
thyroid
to
the
hyoid
bone
(Fig.
2).
CT
scans
of
chest,
abdomen
and
pelvis,
did
not
show
any
evidence
of
distant
metastases.
T2
weighted
MRI
axial
(A)
and
coronal
STIR
(B)
images
demonstrating
a
68
mm
×
47
mm
×
93
mm
mixed
T2
isoin-
tense/hyperintense
infrahyoid
lesion.
It
indents
the
superior
thyroid
gland
and
displaces
the
larynx
and
trachea
posteriorly.
A
T1/T2
weighted
magnetic
resonance
imaging
(MRI)
of
the
face
and
neck
with
STIR
and
diffusion
imaging
was
performed
to
evaluate
the
lesion
further
(Fig.
3).
The
mass
measured
68
mm
(long
axial
dimension)
×
47
mm
(short
axial
dimension)
×
93
mm
(cranio–caudal),
extending
inferiorly
to
the
suprasternal
notch,
and
superiorly
to
the
level
of
the
hyoid
bone.
A
small
volume
of
thyroid
tissue
was
noted
postero-inferior
to
the
mass,
and
some
slightly
enlarged
level
IV
lymph
nodes
(up
to
13
mm).
Following
discussion
at
the
head
and
neck
cancer
multidisci-
plinary
meeting,
she
underwent
total
thyroidectomy
and
level
VI
neck
dissection,
during
which
a
partially
necrotic
tumour
mass
was
excised
(Fig.
4).
Histological
examination
revealed
a
high-
grade
mucoepidermoid
carcinoma
above
the
gland
consistent
with
origin
in
a
thyroglossal
duct
remnant
(Fig.
5).
Six
central
compartment
neck
nodes
were
retrieved,
with
no
evidence
of
metastasis.
The
carcinoma
was
investigated
for
MECT1/MAML2
(CRTC1/MAML2)
translocation
typical
of
mucoepidermoid
car-
cinoma
by
fluorescence
in-situ
hybridisation
using
a
MAML2
dual
colour
break
apart
probe
(Zytovision
GmBH,
Bremerhaven,
Germany).
The
carcinoma
cells
were
negative
for
the
translo-
cation
but
showed
a
slightly
increased
copy
number
2–4
throughout.
Apart
from
a
return
to
theatre
for
evacuation
of
neck
haematoma
at
one-week
post
surgery,
she
had
an
uneventful
post-operative
recovery.
She
underwent
adjuvant
radiotherapy
(60
Gy
in
30
fractions)
in
view
of
close
surgical
margins.
She
experienced
radiotherapy
related
side
effects
including
mucositis,
which
were
managed
conservatively
with
analgesia
and
steroids
and
is
alive
two
years
following
completion
of
oncological
treatment.
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94
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96
97
98
99
100
101

Please
cite
this
article
in
press
as:
E.
Warner,
et
al.,
Mucoepidermoid
carcinoma
in
a
thyroglossal
duct
remnant,
Int
J
Surg
Case
Rep
(2015),
http://dx.doi.org/10.1016/j.ijscr.2015.04.028
G
Model
IJSCR
1356
1–5 CASE
REPORT
–
OPEN
ACCESS
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Warner
et
al.
/
International
Journal
of
Surgery
Case
Reports
xxx
(2015)
xxx–xxx
3
Fig.
3.
T2
weighted
MRI
axial
(A)
and
coronal
STIR
(B)
images
demonstrating
a
68
mm
×
47
mm
×
93
mm
mixed
T2
isointense/hyperintense
infrahyoid
lesion.
It
indents
the
superior
thyroid
gland
and
displaces
the
larynx
and
trachea
posteriorly.
To
the
best
of
our
knowledge,
this
is
the
first
reported
case
of
MEC
arising
within
an
existing
thyroglossal
duct
remnant.
We
present
a
review
of
the
literature
on
mucoepidermoid
carcinoma
of
the
thyroid
and
recommend
that
surgical
excision
of
these
lesions
should
be
performed
due
to
the
risk
of
malignant
transformation.
3.
Discussion
MEC
are
one
of
the
most
common
malignant
neoplasms
of
sali-
vary
glands
in
adults,
but
also
arise
in
sites
as
diverse
as
the
lung,
breast,
pancreas,
oesophagus,
lacrimal
ducts
and
thyroid
[5–10],
where
a
high
index
of
suspicion
is
needed
for
accurate
diagnosis.
In
the
medical
literature
to
date
there
are
only
40
reported
cases
of
thyroid
MEC,
and
no
previous
cases
of
mucoepidermoid
carcinoma
arising
in
a
thyroglossal
duct
cyst
[9,13].
MEC
was
first
described
by
Stewart
in
1945
[11].
Onset
is
usually
in
the
5th
decade
of
life,
and
it
usually
presents
with
a
fixed
painless
enlarging
swelling
[1].
Women
are
more
commonly
affected
than
men
[2].
Typical
histopathological
features
include
the
presence
of
epidermoid,
mucinous
and
clear
cells
(see
Fig.
5).
Mucoepidermoid
carcinoma
is
consistently
associated
with
a
t(11;19)(q21;p13)
translocation
that
produces
a
fusion
pro-
tein
combining
the
CREB-regulated
transcription
coactivator
1
(CRTC1/MECT1)
with
Notch
coactivator
mastermind-like
gene
2
(MAML2)
that
constitutively
activates
the
Notch
signalling
path-
way
important
in
development
and
oncogenesis
[14,16,17].
This
change
was
thought
to
be
a
causative
oncogenic
event,
though
it
has
since
also
been
found
in
a
minority
of
Warthin’s
tumours
and
hidradenocarcinomas
[15].
In
mucoepidermoid
carcinomas
of
salivary
glands
the
translocation
is
associated
with
low
and
intermediate
grade
carcinomas
and
a
lower
risk
of
recurrence,
Fig.
4.
Macroscopic
histopathology.
A:
Macroscopic
specimen
from
total
thyroidectomy:
a
single
spherical
mass
89mm
×
80mm
×
55
mm
without
identifiable
left
or
right
lobes,
or
obvious
thyroid
capsule.
B:
Cut
surface
demonstrates
well
circumscribed,
partly
necrotic
tumour
mass
102
103
104
105
106
107
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111
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126
127
128
129
130

Please
cite
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in
press
as:
E.
Warner,
et
al.,
Mucoepidermoid
carcinoma
in
a
thyroglossal
duct
remnant,
Int
J
Surg
Case
Rep
(2015),
http://dx.doi.org/10.1016/j.ijscr.2015.04.028
G
Model
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xxx
(2015)
xxx–xxx
Fig.
5.
Microscopic
histopathology
demonstrating
mucoepidermoid
tumour.
A:
High
power
image
showing
epidermoid,
clear
and
mucinous
cells.
B:
Cytokeratin
5/6
immunohistochemistry
confirms
epidermoid
differentiation
(brown
stain).
C:
B/PAS
stain
reveals
mucin
secretion
(blue).
metastasis
and
death
than
those
without
[16].
However,
approx-
imately
only
a
quarter
of
high
grade
mucoepidermoid
carcinomas
contain
the
translocation,
and
those
that
are
positive
are
thought
to
arise
by
high
grade
transformation
from
low
grade
carcinomas
[17].
Our
presented
case
did
not
contain
the
translocation,
suggest-
ing
that,
despite
its
mucoepidermoid
differentiation,
it
did
not
arise
through
translocation
as
most
low
grade
salivary
gland
carcinomas
do.
There
is
paucity
of
knowledge
on
thyroid
MEC,
but
three
cases
have
been
shown
to
be
positive
for
the
t(11;19)(q21;p13)
translo-
cation
[16–18].
As
the
majority
of
thyroid
MEC
are
low
grade,
the
finding
that
this
unusual
high
grade
tumour
lacks
the
translocation
is
interesting
and
suggests
that,
as
in
salivary
gland
neoplasms,
the
high
grade
carcinomas
are
a
biologically
and
molecularly
different
group.
There
are
two
main
theories
for
the
histogenesis
of
thyroid
MEC;
that
it
develops
from
solid
cell
nests
(SCN)
or
thyroid
follicu-
lar
epithelial
cells
[10].
SCN
share
many
histopathological
features
with
MEC,
including
ductal
structures
lined
by
ciliated
epithelium,
and
multipotent
cells.
These
multipotent
cells
have
been
linked
to
several
thyroid
cancers,
including
papillary
and
MEC.
Alterna-
tively,
it
is
thought
that
papillary
carcinoma
(the
most
common
malignancy
found
in
thyroglossal
duct
remnants),
may
undergo
squamous
and
mucinous
metaplasia
resulting
in
MEC
[21,22,13].
Prichard
et
al.
report
three
cases
where
de-differentiation
appears
to
have
occurred
from
other
thyroid
neoplasms
[13].
In
addition,
thyroid
specific
mRNAs
including
TTF-1
and
PAX-8,
which
are
exclusively
found
together
in
thyroid
follicular
cells
were
both
detected
in
a
metastatic
axillary
lymph
node
of
MEC
[21].
It
is
not
clear,
whether
MEC
in
a
thyroglossal
duct
remnant
has
arisen
via
the
same
means
as
a
thyroid
MEC,
or
via
another
as
yet
unspecified
mechanism.
The
sudden
onset
enlargement
of
our
patient’s
presumed
thy-
roglossal
duct
cyst
and
suspicion
of
nodal
disease
on
pre-operative
imaging
lead
to
the
decision
for
aggressive
surgical
management
with
total
thyroidectomy
and
selective
level
VI
neck
dissection
rather
than
the
more
conservative
Sistrunk
procedure.
The
avail-
able
literature
supports
the
use
of
adjuvant
radiotherapy
in
cases
of
high
grade
tumours,
which
generally
carry
a
poor
prognosis
[23–25].
In
addition,
the
posterior
extension
of
the
tumour
to
the
trachea,
necessitated
incomplete
resection
margins
that
would
have
potentially
dictated
a
poorer
outcome
without
additional
treatment.
Pritchard
et
al.
[13]
support
the
use
of
radioiodine
ablation
due
to
the
possible
origins
of
these
tumours
having
de-differentiated
from
other
thyroid
neoplasms.
Thankfully,
our
patient
is
alive
at
two
years
following
the
completion
of
oncological
treatment.
4.
Conclusion
In
conclusion,
we
report,
to
the
best
of
our
knowledge,
the
first
case
of
mucoepidermoid
carcinoma
arising
from
a
thyroglossal
duct
remnant.
Whilst
the
majority
of
thyroglossal
duct
carcinomas
are
of
papillary
type,
a
high
index
of
suspicion
must
be
kept
in
case
of
more
aggressive
carcinomas.
This
case
highlights
the
importance
of
removal
of
thyroglossal
duct
cysts
at
an
early
stage,
and
lends
weight
to
the
argument
for
a
more
radical
surgical
approach,
and
adjuvant
treatment
particularly
for
large
carcinomas,
or
when
clear
margins
cannot
be
assured.
Conflict
of
interest
None
declared.
Funding
None.
Ethical
approval
Not
required.
Consent
Written
informed
consent
was
obtained
from
the
patient
for
publication
of
this
case
report
and
accompanying
images.
A
copy
of
the
written
consent
is
available
for
review
by
the
Editor-in-Chief
of
this
journal
on
request.
Author
contribution
Elinor
Warner:
Data
collection,
literature
review,
presentation
at
international
and
national
meetings,
writing
paper.
Enyi
Ofo:
Writing
paper
and
reviewing
and
editing
drafts.
Steve
Connor:
Radiological
review
of
imaging,
editing
drafts.
Edward
Odell:
Pathological
review
of
histology,
editing
drafts.
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204

Please
cite
this
article
in
press
as:
E.
Warner,
et
al.,
Mucoepidermoid
carcinoma
in
a
thyroglossal
duct
remnant,
Int
J
Surg
Case
Rep
(2015),
http://dx.doi.org/10.1016/j.ijscr.2015.04.028
G
Model
IJSCR
1356
1–5 CASE
REPORT
–
OPEN
ACCESS
E.
Warner
et
al.
/
International
Journal
of
Surgery
Case
Reports
xxx
(2015)
xxx–xxx
5
Jean-Pierre
Jeannon:
Lead
supervisor,
recruitment
of
patient,
consenting
patient,
review
and
editing
of
draft
manuscript.
Guarantor
Mr
Jean-Pierre
Jeannon.
Uncited
referencesQ5
[12,19,20].
Acknowledgements
and
disclosures
Presented
at
the
Irish
Otolaryngological
and
Head
and
Neck
Soci-
ety
and
awarded
“Highly
Commended”
poster
presentation.
(Oct.
2013)
Presented
at
Semon
Club
(Nov.
2013).
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