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 previous 2 weeks. She had no associated dysphagia or
in the midline of the base of the tongue. Its estimated dyspnea, and no other relevant history.
prevalence rangesfrom 1 in 3,000to 1 in 10,000 popula- A general physical examination revealed no abnor-
tion. Wereport theinterestingcase ofa28-year-old woman malities. However, the oropharyngeal examination
who presented with a primary papillary carcinoma in a revealed the presence of a globular, fleshy, 2-cm mass
lingual thyroidand a histologically normalthyroidgland. arising from the midline on the base of the tongue. On
To thebestofourknowledge, thiscase probably represents intraoral palpation, the mass was limited to the orophar-
only the second reported case of a follicular variant of a ynx without supraglottic extension. The mass was firm
papillarycarcinoma arising in a lingual thyroid. and sensitive to touch, but it did not bleed on touch. No
other swellingwasfound in the upper aerodigestive tract.
Introduction External examination of the neck revealed bilateral
Lingual thyroid is the term applied to a mass of ectopic 0.5 x 1.0-cm level IB nodes that were nontender, mo-
thyroid tissue that is located in the base of the tongue bile, and firm. On further palpation, a firm, nontender,
at the midline. Its prevalence is variably estimated at 1 solitary, 1.0 x 1.0-cm nodule was found in the superior
in 3,000 to 1 in 10,000 population.' The primary thy- pole of the left thyroid lobe. This mass moved with de-
roid tissue is absent in 70% of cases.' Lingual thyroids glutition but not with protrusion of the tongue.
usually contain histologically normal tissue; reports of Findings on computed tomography (CT) of the oro-
a carcinoma arising within this form of ectopic thyroid pharynx were suggestive of a vascular malformation
are rare. In this article, we report an extremely rare case (figure). The mass measured 2.5 x 2.3 x 3.6 em. Fine-
of a follicular variant of a papillary carcinoma arising needle aspiration cytology (FNAC) ofthe oropharyngeal
in a lingual thyroid. mass revealed only blood and skeletal muscle bundles,
which suggested ahemangioma of the tongue base. FNAC
Case report of the solitary thyroid mass demonstrated evidence of
A 28-year-old woman presented to our outpatient unit colloid changes in the nodule.
with a complaint of blood-tinged saliva for the previous Laboratory testing revealed a total triiodothyronine
2 weeks. She said that she had experienced a similar (T 3) level of 104 ng/dl, a total thyroxine (T 4) level of 6.3
episode 3 years earlier; the previous episode had lasted figl dl, and a thyroid-stimulating hormone (TSH) levelof
3 days and subsided without any treatment. She also 2.09 mIU/L, all of which were within the normal refer-
reported that she had felt a lump in her throat for the 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
From the Department of Otolaryngology-Head and Neck Surgery. Kas- excised via a median pharyngotomyapproach. Complete
turba Medical College. Mangalore, India (Dr. Bhojwani, Dr. Hegde,
exposure of the tongue base was achieved by entering
and Dr. Alva); and the Department ofENT. RajaRajeshwari Medical
College & Hospital. Mangalore (Dr. Vishwas). The case described in the pre-epiglottic space and vallecula. Intraoperatively,
this article occurred at the Kasturba Medical College. the globular vascular mass was found to be arising from
Corresponding author: Dr. Kiran M. Bhojwani, Department ofOto!aryn-
gology-Head and Neck Surgery. Kasturba Medical College Hospital,
the left side of the tongue base on a stalk. The pedicled
Attavar, Mangalore-575002, India. Email: sunbeamis@yahoo.co.in fleshy mass extended anteriorly up to the circumvallate

Volume 91, Number 7 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 dysphagia or airway compromise in infancy or child-
papillary thyroid cancer within a lingual thyroid. hood.Y In rare cases it presents with acute bleeding.
In light of the pathology findings, the patient was Our patient presented with chiefcomplaints ofblood-
counseled on the need for thyroid surgery. Within 15 tinged saliva and a feeling of a lump in the throat. She had
days ofthe first surgery, she underwent a total thyroidec- not experienced any dysphagia or dyspnea, even though
tomywith central neck node clearance. Intraoperatively, the size of her lingual thyroid on CT was measured as
nodular tissues were found in the superior pole of the 2.5 x 2.3 x 3.6 em. The most logical explanation for her
left lobe and the inferior pole of the right lobe. Her clinical picture is that the attachment of the mass by a
postoperative recovery was aided by levothyroxine and pedicle made it more susceptible to bleeding and that
calcium supplementation. The cervical thyroid specimen the mass's overt mobility might not have caused any
showed no histologic evidence of papillary carcinoma, static obstruction Significant enough to cause dysphagia.
and it was reported as benign, even after multiplanar Lingual thyroid tissue is known to be associated with
sections were studied. an absence ofcervical thyroid tissue in 70% ofpatients. 2
The patient underwent a whole-body radioiodine scan, It has also been reported that hypothyroidism is more
and she was discharged with advice for regular follow-up common than hyperthyroidism in patients with a lingual
every 3 months with clinical evaluation, thyroglobulin thyroid as a consequence ofthe failure ofthe lateral lobes
assays, and nuclear scintigraphy. Three years later, the to fuse to the median lobes, which results in an insuffi-
patient was asymptomatic and disease-free. cient number of cells being made available to adequately
produce thyroid hormones." However our patient had
Discussion a fully developed thyroid gland in the euthyroid state.
The origin of a lingual thyroid is the result of a failure Standard CT has been found to adequately demon-
ofthe thyroid primordium to descend into the neck. As strate a lingual thyroid in the oropharynx. However,
a consequence, a focus of thyroid tissue remains at the the lesion in our patient was mistakenly believed to
foramen cecum. Sometimes a lingual thyroid is the only be a vascular malformation because the precontrast
functioning thyroid tissue in the body. On the other hand, scan showed a homogeneous, well-defined soft-tissue
an overdescent of the thyroid primordium can result in attenuation of 40 Hounsfield units (HU) with intense
the formation ofectopic thyroid tissue in the mediastinal postcontrast enhancement of 80 to 100 HU.
area or even within the cardiac endothelium.' In light of the fact that our patient presented with a
Lingual thyroid tissue has been well documented by bleeding mass in the base of the tongue, a conventional
many independent reports in the literature. lAS These diagnosis of a malignancy or a benign lesion such as a
reports include a series of 4 cases in our city of Manga- hemangioma was suggested. The intraoral FNAC had
lore, India.' Lingual thyroid is known to present as an failed to reveal any definitive diagnosis. Therefore, surgi-
asymptomatic, smooth-surfaced lump in the midline of cal exploration was planned, with adequate precautions
the posterior one- third ofthe tongue between the sulcus being taken for airway and hemodynamic support.
terminalis and the epiglottis. In most cases it is detected In our group's limited experience with lingual thyroid
incidentally on radioiodine scanning, or it presents with (5 cases), the present case is the first that involved a

290 • www.entjournal.com ENT-Ear, Nose & Throat Journal • July 2012

 PAPILLARY CARCINOMA IN A LINGUAL THYROID: AN UNUSUAL PRESENTATION

carcinoma. Our review of the literature found fewer 5. Benhammou A. Benche ikh R. Benbouzid MA. et aI. Ectopic lingual
thyroid. B-ENT 2006;2(3):121-2.
than 50 cases of a malignant change in an ectopic lin-
6. Gallo A, Leonett i F. Torri E, et al. Ectop ic lingual thyroid as unusual
gual thyroid, with follicular cancer being the dominant cause of severe dysphag ia. Dysph agia 200 1;16(3):220-3.
variety,":" In only 5 of these reported cases":" did the 7. Bayram F, Kiilahli I, Yiice I. et al. Fun ctional lingual thyroid as
unusual cause of progressive dysphagia . Thyroid 2004;14(4):321-4.
patient develop papillary cancer, and in only I case "
8. Neinas H V,Gorman CA. Devine KD. Woolner LB.Lingual thyroid.
was it the follicular variant of papillary carcinoma. To Clinic al characteristics of 15 cases. Ann Intern Med 1973;79(2):
the best of our knowledge, our case is probably only the 205-10.
9. Massine RE. Durning SI. KoroscilTM. Lingual thyroid carcinoma:A
second reported case of the follicular variant of a papil- case reporta nd reviewo fthe literature.Thyroid2001;1 1(12):1191-6.
lary carcinoma arising in a lingual thyroid. 10. BigottiG. Coli A. Follicular carcin oma in lingual thyroid presenting
Patients with a lingual thyroid and a nonfunctioning as a latero -cervical mass. Case report and review of the literature.
I Oral Pathol Med 1997;26(3):142-6.
primary thyroid can be treated with levothyroxine to II . Casella A, Pisano R, Navarro Cuellar C, et al. Papillary carcinoma of
keep their TSH level between 0.2 and 0.6 mIU/L, which the base of the tongue. Case clinic. Minerva Stomato11999;48( II):
should result in shrinkage of the tissue and a return of. 535-8.
12. Goldstein B, Westra WH, Califano J. Multifocal papillary thyroid
general health to normal. When a lingual mass does carcinoma arising in a lingual thyroid: A case report. Arch Otolar-
not shrink or when it bleeds or becomes ulcerated, a yngol Head Neck Surg 2002;128(10) :1198-1200.
biopsy is advisable. In the event that ectopic thyroid 13. Perez JS. Munoz M, Naval L. et al. Papillary carcinoma arising in
lingual thyroid. J Craniofac Surg 2003;31(3):179-82.
tissue harbors a malignancy, surgery at the base of the 14. Basaria S. Westra WH, Cooper DS. Ectopic lingual thyroid mas-
tongue followed by a total thyroidectomy is advisable, querading as thyroid cancer metastases. I Clin Endocrinol Metab
2001;86(1):392-5.
provided that the cervical thyroid gland is present on
15. Hari CK, Kumar M. Abo-Khatwa MM , et al. Follicular variant of
preoperative ultrasonography. papillary carcinoma arising from lingu al thyroid. Ear Nose Throat
In terms of the surgical approach to a lingual tumor, 12009;88(6):E7.
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

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.

References Our site offers: Online Exclusives (full-

1. Pra sad KC, Bhat V. Surgical management of lingual thyroid: A
report of four cases. J Oral Maxillofac Surg 2000;58(2):223-7.
length articles vou 'll find only online).
2. Montgomery ML. Lingual thyro id-a comprehensive review. The
Western Journal of Surgery 1935;43:661.1936 ;44:122.189, 237. 303.
full-text content of the current print
373. 442. issue. ENT Clinics. News & Events.
3. Pintar JE. Normal development ofhypothal amic-pituitary-thyroid
axis. In: Braverman LE, Utiger RD, eds. Werner & Ingber's The reader polls. subscription information.
Thyroid . 8th ed. Philadelphia: Lippincott Williams & Wilkins; 2000:
7· 43. and Instructions for Authors.
4. Williams ED, Toyn CEo Har ach HR. The ultimobran chial gland

www.entjournal.com
and congenital th yroid abnormalities in man. J PathoI1989;159(2):
135-41.

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