Lingual Thyroid Case

Report

Jeffrey

B.

and Review of the Literature

Monroe, MD, Daniel Fahey,

MD

review the world literature on lingual thyroid. One of our cases of lingual thyroid is added to this body of information specifically because of the patient's unusual lack of any response to hormonal suppression treatment. This behavior was suggestive of a malignant neoplasm and was, in our opinion, indication for excisional biopsy, even without any of the usual symptoms associated with lingual thyroid. To our knowledge, this behavior has not been previously reported. \s=b\ We

(Arch Otolaryngol 101:574-576, 1975)

thyroid is an unusual i lesion that must be considered in any patient who has a mass at the base of the tongue. By 1969, 373 cases of lingual thyroid had been reported in the world literature.1 Montgomery, in an extensive re¬ view of the subject, established cer¬ tain criteria necessary for making the diagnosis of lingual thyroid.2 These criteria are as follows: (1) the pres¬ ence of a lingual mass between the epiglottis and the circumvallate pa¬

Lingual

pillae; (2) biopsy specimen revealing thyroid tissue; and (3) hypothyroid manifestations following excision. Accepted

for publication May 13, 1975. From the Department of Otolaryngology, State University of New York at Buffalo. Reprints not available.

Ward et al' added a fourth criterion as follows: (4) radioactive iodine uptake (RAIU) in the mass at the base of the

tongue.

complaint of a patient lingual thyroid is likely to be one of dysphagia, dyspnea, dys¬ phonia, or intraoral mass. Seventyfive percent of these patients will be The chief

with

a

female.1 The initial manifestation of this disease may be a rapidly expand¬ ing mass in the mouth of a previously healthy child. An explanation for the sudden rapid growth may be that the child outstrips the hormone-produc¬ ing capacity of his abnormal thyroid gland due to his own rapidly increas¬ ing growth and subsequent metabolic demands. This hypothyroid state trig¬ gers an increased release of thyroidstimulating hormone (TSH). Since the dysfunctional lingual thyroid can¬ not meet the increased demand, it merely becomes hypertrophie. If other normal thyroid tissue is present this will not occur, but 70% or more of lingual thyroid patients have no thy¬ roid tissue other than that present in their oral cavity.4 It can be readily ap¬ preciated that a lingual thyroid pa¬ tient may have clinical manifesta¬ tions of hypothyroidism since this is the metabolic state most likely to oc¬ cur. Cretinism may be present in as many as 10% of these patients."

REPORT OF A CASE A 17-year-old girl was referred to us with the diagnosis of lingual thyroid. This patient was unusual in that she had no symptoms. She first became aware of her anomaly while examining her throat dur¬ ing an attack of pharyngitis. At that time she noted a large pink mass on the dorsum of her tongue. A scintiscan showed uptake at the base of the tongue in the area of the lesion (Fig 1). On physical examination, a pinkish 3-cm mass was found at the base of the tongue. The mass was solid, not tender with promi¬ nent surface vascularity, and no pulsations (Fig 2). A summary of the patient's endo¬ crine workup and management is shown in the Table. Throughout treatment, the size of the patient's lingual thyroid did not decrease substantially. It was thought that ade¬ quate replacement and suppressive ther¬ apy had been given, and with an RAIU of almost zero and a low TSH level, it was ob¬ vious that the patient did indeed take her medicine. Since there were no thyroid anti¬ bodies, thyroiditis was not considered. The question of carcinoma arose, and for this reason, an excisional biopsy was done. An oral approach was used since the lesion was small. The pathologic diagnosis on the operative specimen indicated apparently normal thyroid tissue without evidence of carcinoma.

TREATMENT With

a

symptomatic lingual thyroid that

represents

no

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emergency, medical treat-

should be tried. By instituting ther¬ apy with liothyronine sodium, TSH release should be suppressed.5 With the pituitarythyroid axis inhibited, maintenance thy¬ roid replacement with thyroid extract in full doses should be begun. In the majority of patients, this regimen will decrease the size and vascularity of the gland remark¬ ably. The offending symptoms may be en¬ tirely alleviated, thus obviating the need for surgery. Even if surgery should subse¬ quently prove necessary, the decreased size and vascularity of the gland will cer¬ tainly facilitate the operation. Radioactive iodine therapy for glandular ablation may be helpful in certain cases. However, since the majority of these glands are hypoactive, the amount of ra¬ dioactivity necessary for gland destruction may prove too toxic for the patient to tol¬ erate.5 In addition, many authors feel that the carcinogenic potential of RAIU is still undefined. Therefore, its safe use in the young is questionable.34e Fish and Moore advise using radioactive iodine only in pa¬ tients refusing or unable to undergo surgi¬ cal intervention, and in whom medical management with thyroid hormones has been ineffective.4 The surgical approach to the problem of lingual thyroid is discussed ment

by Ward et al." Pathologic diagnosis of thyroid tissue is difficult, but the microscopic evaluation of lingual thyroid is worsened by some of its innate characteristics. Lingual thyroid tis¬ sue has no distinct capsule and it is usually found infiltrating the musculature of the tongue. In the past, many lingual thyroids have retrospectively been diagnosed as being carcinomatous on the basis of post¬ operative recurrence. In all probability, the majority of these recurrences represent mere hypertrophy of the thyroid tissue left

Fig

1— Scintiscan

Fig 2.—Intraoral

showing positive uptake

at base of

tongue.

lesion.

behind at surgery.

LINGUAL THYROID CARCINOMAS

The question of carcinoma in lin¬ gual thyroid becomes of major con¬ cern in deciding for or against autotransplantation and in the extent of extirpative surgery necessary in re¬ moving this lesion. The earliest men¬ tion of cancer in lingual thyroid was reported in 1910 by two observers, Gunn and Rutgers. In 1936, Mont¬ gomery reported an additional seven cases,2 and Buckman reported seven carcinomas and two sarcomas. Wat¬ son and Pool, in 1940, found three lin¬ gual thyroid carcinomas in 167 pa¬ tients with malignant neoplasms of the thyroid. Eight cases of carcinoma were reported by Cromartie and Wei-

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in 1941. Wapshaw reported 11 in 1942 and Mill reported 15 in 1958. In reviewing the literature, Fish and Moore found 21 cases of lingual thyroid carcinoma.4 Twelve were fe¬ male and seven were male (the sex of two patients was not given). The ages ranged from 18 to 86, with an average age of 41. Ward et al noted that five of six lin¬ gual thyroid carcinomas occurred in male patients.3 Hung found no re¬ ports of lingual thyroid carcinoma in children.6 It would appear that the develop¬ ment of cancer in lingual thyroid tis¬ sue is a definite clinical entity that must be considered when dealing son

Patient's Endocrine Workup and Management Date

11/11/71

RAIU 11% at base

Treatment With Desiccated Thyroid 128 mg

of tongue

12/13/71 4/10/72 12/5/72

Serum T4, ng/ml TSH, mil/ml (Normal, 50-130) (Normal,