ORIGINAL ARTICLE
Papillary carcinoma in a lingual
thyroid: An unusual presentation
Kiran M. Bhojwani, MS; Mahesh Chandra Hegde, MS; Arathi Alva, MS;
K,Y: Vishwas, MBBS, MS
Abstract
A lingual thyroidisa massofectopic thyroidtissue located
in the midline of the base of the tongue. Its estimated
prevalence rangesfrom 1 in 3,000to 1 in 10,000 popula-
tion. Wereport theinterestingcase ofa28-year-old woman
who presented with a primary papillary carcinoma in a
lingual thyroidand a histologically normalthyroidgland.
To thebestofourknowledge, thiscase probably represents
only the second reported case of a follicular variant of a
papillarycarcinoma arising in a lingual thyroid.
Introduction
Lingual thyroid is the term applied to a mass of ectopic
thyroid tissue that is located in the base of the tongue
at the midline. Its prevalence is variably estimated at 1
in 3,000 to 1 in 10,000 population.' The primary thy-
roid tissue is absent in 70% of cases.' Lingual thyroids
usually contain histologically normal tissue; reports of
a carcinoma arising within this form of ectopic thyroid
are rare. In this article, we report an extremely rare case
of a follicular variant of a papillary carcinoma arising
in a lingual thyroid.
Case report
A 28-year-old woman presented to our outpatient unit
with a complaint of blood-tinged saliva for the previous
2 weeks. She said that she had experienced a similar
episode 3 years earlier; the previous episode had lasted
3 days and subsided without any treatment. She also
reported that she had felt a lump in her throat for the
From the Department of Otolaryngology-Head and Neck Surgery. Kas-
turba Medical College. Mangalore, India (Dr. Bhojwani, Dr. Hegde,
and Dr. Alva); and the Department ofENT. RajaRajeshwari Medical
College & Hospital. Mangalore (Dr. Vishwas). The case described in
this article occurred at the Kasturba Medical College.
Corresponding author: Dr. Kiran M. Bhojwani, Department ofOto!aryn-
gology-Head and Neck Surgery. Kasturba Medical College Hospital,
Attavar, Mangalore-575002, India. Email: sunbeamis@yahoo.co.in
Volume 91, Number 7
previous 2 weeks. She had no associated dysphagia or
dyspnea, and no other relevant history.
A general physical examination revealed no abnor-
malities. However, the oropharyngeal examination
revealed the presence of a globular, fleshy, 2-cm mass
arising from the midline on the base of the tongue. On
intraoral palpation, the mass was limited to the orophar-
ynx without supraglottic extension. The mass was firm
and sensitive to touch, but it did not bleed on touch. No
other swellingwasfound in the upper aerodigestive tract.
External examination of the neck revealed bilateral
0.5 x 1.0-cm level IB nodes that were nontender, mo-
bile, and firm. On further palpation, a firm, nontender,
solitary, 1.0 x 1.0-cm nodule was found in the superior
pole of the left thyroid lobe. This mass moved with de-
glutition but not with protrusion of the tongue.
Findings on computed tomography (CT) of the oro-
pharynx were suggestive of a vascular malformation
(figure). The mass measured 2.5 x 2.3 x 3.6 em. Fine-
needle aspiration cytology (FNAC) ofthe oropharyngeal
mass revealed only blood and skeletal muscle bundles,
which suggested ahemangioma of the tongue base. FNAC
of the solitary thyroid mass demonstrated evidence of
colloid changes in the nodule.
Laboratory testing revealed a total triiodothyronine
(T 3) level of 104 ng/dl, a total thyroxine (T 4) level of 6.3
figl dl, and a thyroid-stimulating hormone (TSH) levelof
2.09 mIU/L, all of which were within the normal refer-
ence range. The patient's hemoglobin concentration was
low at 10.2g/dl, and she therefore received a transfusion
of 3 units of a-positive, cross-matched whole blood.
Following preoperative preparation, the lesion was
excised via a median pharyngotomyapproach. Complete
exposure of the tongue base was achieved by entering
the pre-epiglottic space and vallecula. Intraoperatively,
the globular vascular mass was found to be arising from
the left side of the tongue base on a stalk. The pedicled
fleshy mass extended anteriorly up to the circumvallate
www.entjournal.com • 289
 BHOJWANI, HEGDE, ALVA, VISHWAS
line; posteriorly, it stopped short of the
vallecula and crossed the midline to the
right. The mass was removed in toto,
and a wedge excision of the tongue base
was made. The patient's recovery was
uneventful, and she was discharged on
postoperative day 7.
On histopathology, we were surprised
to learn that the specimen contained
ulcerated squamous epithelium with
underlying infiltrating thyroid papillary
tumor cells within the lingual thyroid.
On repeat sectioning and microscopic Figure. Thepedunculatedlingual mass is seenon pre- (A) and postcontrast (B) CTs.
evaluation by experienced pathologists,
the lesion was confirmed as the follicular variant of
papillary thyroid cancer within a lingual thyroid.
In light of the pathology findings, the patient was
counseled on the need for thyroid surgery. Within 15
days ofthe first surgery, she underwent a total thyroidec-
tomywith central neck node clearance. Intraoperatively,
nodular tissues were found in the superior pole of the
left lobe and the inferior pole of the right lobe. Her
postoperative recovery was aided by levothyroxine and
calcium supplementation. The cervical thyroid specimen
showed no histologic evidence of papillary carcinoma,
and it was reported as benign, even after multiplanar
sections were studied.
The patient underwent a whole-body radioiodine scan,
and she was discharged with advice for regular follow-up
every 3 months with clinical evaluation, thyroglobulin
assays, and nuclear scintigraphy. Three years later, the
patient was asymptomatic and disease-free.
Discussion
The origin of a lingual thyroid is the result of a failure
ofthe thyroid primordium to descend into the neck. As
a consequence, a focus of thyroid tissue remains at the
foramen cecum. Sometimes a lingual thyroid is the only
functioning thyroid tissue in the body. On the other hand,
an overdescent of the thyroid primordium can result in
the formation ofectopic thyroid tissue in the mediastinal
area or even within the cardiac endothelium.'
Lingual thyroid tissue has been well documented by
many independent reports in the literature. lAS These
reports include a series of 4 cases in our city of Manga-
lore, India.' Lingual thyroid is known to present as an
asymptomatic, smooth-surfaced lump in the midline of
the posterior one- third ofthe tongue between the sulcus
terminalis and the epiglottis. In most cases it is detected
incidentally on radioiodine scanning, or it presents with
290 • www.entjournal.com
dysphagia or airway compromise in infancy or child-
hood.Y In rare cases it presents with acute bleeding.
Our patient presented with chiefcomplaints ofblood-
tinged saliva and a feeling of a lump in the throat. She had
not experienced any dysphagia or dyspnea, even though
the size of her lingual thyroid on CT was measured as
2.5 x 2.3 x 3.6 em. The most logical explanation for her
clinical picture is that the attachment of the mass by a
pedicle made it more susceptible to bleeding and that
the mass's overt mobility might not have caused any
static obstruction Significant enough to cause dysphagia.
Lingual thyroid tissue is known to be associated with
an absence ofcervical thyroid tissue in 70% ofpatients. 2
It has also been reported that hypothyroidism is more
common than hyperthyroidism in patients with a lingual
thyroid as a consequence ofthe failure ofthe lateral lobes
to fuse to the median lobes, which results in an insuffi-
cient number of cells being made available to adequately
produce thyroid hormones." However our patient had
a fully developed thyroid gland in the euthyroid state.
Standard CT has been found to adequately demon-
strate a lingual thyroid in the oropharynx. However,
the lesion in our patient was mistakenly believed to
be a vascular malformation because the precontrast
scan showed a homogeneous, well-defined soft-tissue
attenuation of 40 Hounsfield units (HU) with intense
postcontrast enhancement of 80 to 100 HU.
In light of the fact that our patient presented with a
bleeding mass in the base of the tongue, a conventional
diagnosis of a malignancy or a benign lesion such as a
hemangioma was suggested. The intraoral FNAC had
failed to reveal any definitive diagnosis. Therefore, surgi-
cal exploration was planned, with adequate precautions
being taken for airway and hemodynamic support.
In our group's limited experience with lingual thyroid
(5 cases), the present case is the first that involved a
ENT-Ear, Nose & Throat Journal • July 2012
 PAPILLARY CARCINOMA IN A LINGUAL THYROID: AN UNUSUAL PRESENTATION

carcinoma. Our review of the literature found fewer
than 50 cases of a malignant change in an ectopic lin-
gual thyroid, with follicular cancer being the dominant
variety,":" In only 5 of these reported cases":" did the
patient develop papillary cancer, and in only I case "
was it the follicular variant of papillary carcinoma. To
the best of our knowledge, our case is probably only the
second reported case of the follicular variant of a papil-
lary carcinoma arising in a lingual thyroid.
Patients with a lingual thyroid and a nonfunctioning
primary thyroid can be treated with levothyroxine to
keep their TSH level between 0.2 and 0.6 mIU/L, which
should result in shrinkage of the tissue and a return of.
general health to normal. When a lingual mass does
not shrink or when it bleeds or becomes ulcerated, a
biopsy is advisable. In the event that ectopic thyroid
tissue harbors a malignancy, surgery at the base of the
tongue followed by a total thyroidectomy is advisable,
provided that the cervical thyroid gland is present on
preoperative ultrasonography.
In terms of the surgical approach to a lingual tumor,
an oral, lateral cervical, or even a transhyoid incision
may be best. A temporary tracheotomy may be advis-
able in anticipation of postoperative edema in the base
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case reporta nd reviewo fthe literature.Thyroid2001;1 1(12):1191-6.
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G t
of the tongue and upper pharynx. In cases of advanced
and invasive cancer, substantial damage to the tongue
can be expected.
Following surgical clearance, radioiodine scanning
is necessary; iodine-I3I ablation should be performed
only when the pathologic characteristics so demand. The
patient should then receive adequate doses of lifelong
levothyroxine and should be followed for intrathyroidal

o e
cancer by scanning and serial assays of thyroglobulin.
In conclusion, we emphasize the importance of
thoroughly investigating all patients who present with
a midline tongue swelling. The diagnosis ofa malignant
lingual thyroid may be easily missed, as it initially was
Ii
in our patient.

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