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Lingual Thyroid Presenting As Acquired Hypothyroidism in the Adulthood Katsuyoshi Tojo

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Lingual Thyroid Presenting As Acquired Hypothyroidism in the Adulthood Katsuyoshi Tojo CASE REPORT Lingual Thyroid Presenting as Acquired Hypothyroidism in the Adulthood Katsuyoshi Tojo There are numerous reported cases of lingual thyroid with an obvious prevalence in pediatric age. Such ectopic thyroid glands are probably quantitatively deficient and thyroid function may be low or at a low normal level. Apparently, most cases of ectopic thyroid tissue develop congenital hypothyroidism, the so-called cretinism. In this report, wedescribe a very rare adult malecase of lingual thyroid whodeveloped hypothyroidism in adulthood; the anomaly remained undiscovered, being without local commonsymptoms, and permitted a normal life. (Internal Medicine 37: 381-384, 1998) Key words: ectopic thyroid gland, thyroid function, cretinism Introduction Methods Thyroid dysplasia (ectopy, hypoplasia, or aplasia) is a com- Thyroid function assessment moncause of congenital hypothyroidism. Essentially, all cases Commerciallyavailable kits were used to measureserum of thyroid aplasia and most cases of thyroid ectopy become concentrations of thyroid hormones as follows: For TSH, free clinically evident in infancy or childhood. A lingual thyroid, a triiodothyronine (FT3), free thyroxine (FT4), T3 and T4, an rare congenital anomaly first describedby Hickman in 1 869 ( 1), enzyme immunoassay (EIA) (E test [TOSOH] II (TSH), (FT3), is defined as the presence of thyroid tissue at the base of the (FT4), (TT3) and (TT4), TOSOH, Tokyo), respectively. Serum tongue. Actually, the thyroid gland develops as the thyroglossal thyroxine binding globulin (TBG) was determined by radioim- duct which grows at the base of the thyroid gland within the munoassay (RIA) (RIAgnost TBG kit, Hoechst Japan, Tokyo) foramen caecum and finally localizes in the lower neck inferior and thyroglobulin was determined by immuno-radiometric to the thyroid cartilage and anterior to the first few tracheal assay (IRMA) (Ab Bead Thyroglobulin 'EIKEN', EIKEN rings. These steps are usually completed by the 10th fetal week. Kagaku Tokyo). Anti-thyroglobulin antibody (TgAb) and anti- Anyarrest or irregularity in the normal descent of the thyroid thyroid microsomal antibody were determined by commer- gland will result in ectopy. Depending upon the anatomic cially available hemagglutination methods (TGHA:Thyroid location, an ectopic thyroid gland may be classified as lingual test and MCHA:Microsome test, Fuji Rebio, Tokyo). Titers of (at the base of the tongue), sublingual (below the tongue), TGHAand MCHA^1: 102 were defined as positive. TgAb was prelaryngeal (in front of the larynx) or substernal (in the also determined by RIA (TgAb Cosmic kit, Cosmic Co., To- mediastinum) (2). In the hypothyroid condition, oversecretion kyo). Anti-thyroid peroxidase antibody (TPOAb) was deter- of thyrotropin (TSH) causes hypertrophy of the lingual thyroid mined by RIA (TPOAb Cosmic kit, Cosmic Co., Tokyo). The producing local symptoms and in younger patients it may thyroid uptake of 123I was determined by the standard proce- induce dangerous airway obstruction. In some cases, however, dure. T3 uptake was measured by RIA (Spac T3 uptake kit, a lingual thyroid secretes adequate quantities of thyroid hor- Daiichi Radioisotope Lab., Tokyo). TSH-binding inhibitor mone during childhood but is associated with hypothyroidism immunoglobulins (TBII) were measured by the inhibition of in adolescence or adulthood. Wereport here a male adult case 125I-TSH binding to its receptor (TRAb kit, Baxter Travenol of lingual thyroid presenting as acquired hypothyroidism in Co., Tokyo) (normal range: <15%). Thyroid-stimulating anti- adulthood without any local symptoms. bodies (TSAb) were measured using cultured rat FRTL-5 cells. Bovine TSH (SigmaChemical Co., St. Louis, MO) was used as the standard for thyroid stimulation. TSAbactivity was ex- From the Division of Endocrinology and Metabolism, the Second Department of Internal Medicine, The Jikei University School of Medicine, Tokyo Received for publication May 23, 1997; Accepted for publication December 22, 1997 Reprint requests should be addressed to Dr. Katsuyoshi Tojo, the Second Department of Internal Medicine, the Jikei University School of Medicine, 3-25-8 Nishi- Shinbashi, Minato-ku, Tokyo 105-8461 Internal Medicine Vol. 37, No. 4 (April 1998) 381 Tojo pressed as the percent stimulation of bTSH(100 mU//)-stimu- In February 1 997, his home doctor checked his thyroid function lated CAMPincrease (normal range: <180%). because of the past history of lingual thyroid, and hypothy- roidism was first pointed out. On physical examination, his general condition appeared almost normal. His height was 168 Case Report cm and weight was 67.8 kg. Bloodpressure was 1 16/72 mmHg A 32-year-old manwas referred to the endocrine clinic for and pulse rate was 64/min andregular. His skin was slightly dry. evaluation of thyroid function in February 1997. He showed His voice was normal. The thyroid gland was not palpable in the normal growth and development. His fertility is normal. He has normal pretracheal location. a son and a daughter. He had been in good health except for Since the past medical information including his laboratory, duodenal ulcer. There was no history of thyroid disease in his radiological and pathological data was not available unfortu- family. He had never taken any medication which would affect nately, a series of evaluation focused on thyroid function was thyroid function. He first noticed left otalgia in March 1994 and performed to confirm previous diagnosis. Laboratory data on consulted an otolaryngist of another hospital. During a series of admission revealed no particular abnormalities (Table 1 ). Thy- screening exploration for otalgia, a round reddish and rubbery roid function on admission is shown in Table 2. Serum levels of mass, approximately 2.5 cmin diameter was seen at the base of FT3, FT4, T3, T4 and TSH were 1.80 pg/ml, 0.41 ng/dl, 0.92 ng/ the tongue by laryngoscopy. A final diagnosis ofectopic thyroid ml, 3.9 [ig/dl and 379.6 plU/ml, respectively. TGHAand gland was obtained by incision biopsy. At that time his thyroid MCHAwere both weakly positive (1: 102 and l:102, respec- function was normal. Since that time, he was free from any tively. TBII and TSAb were negative (0.8 and 155%, respec- subjective complaints, and had not received any medications. tively). T3 uptake was low or low normal (T3-index: 0.84, T3- Table 1. Laboratory Data on Admission Hematology Urea nitrogen 21 mg/dl White blood cells 5.5X10V Creatinine 1.1 mg/dl Red blood cells 4.53xl0V Na 142 mmol// Hemoglobin 13.7 g/dl K 4.4 mmol// Hematocrit 40.6% Ca 9.7 mg/dl Platelet 215x103/ll1 Pi 3.9 mg/dl Mg 2.0 mg/dl Chemistry Fe 107 jag/dl Asparate aminotransferase 18 IU/Z Unsaturated iron binding Alanine aminotransferase 16 iu// capacity 1 64 |ig/dl Lactic dehydrogenase 118 IU/Z Total cholesterol 202 mg/dl Cholinesterase 474 mU/ml Triglyceride 130 mg/dl Alkaline phosphatase 199 IU// High density lipoprotein Leucin aminopeptidase 65 IU// cholesterol 73 mg/dl y-Glutamyl transferase 18 mU/ml Creactive protein 0.0 mg/dl Creatine phosphokinase 104 IU// Aldolase 3.1 IU///37°C Urine 7.4 g/dl Protein (-) TotalAlbumin protein 4.6 g/dl Sugar (-) A/G 1.6 Occult blood (-) Table 2. Thyroid Function Thyrotropin379.6 (normal: 0.34-4.04 jilU/ml) Triiodothyronine (T3) Free triiodothyronine (Free T3) Thyroxine (T4) Free thyroxine (Free T4) T3 uptake: T3-index T3-percent 123I-uptake: 3 hour 6hour Thyroxine binding globulin 0.921.803.90.410.84 (normal:0.86-1.622.36-5.004.7-11.10.88-1.670.80-1.20)ng/ml)pg/ml)jug/dl)ng/dl) 23.217780 (normal:21.5-38.1%)6.5%8.1%12-30(Hg/dl)<30ng/ml)ThyroglobulinThyroidTSHreceptorantibodyperoxidasestimulatingantibody<0.3(normal:0.8155 <0.3(normal:<0.5%)U/ml)<0.3<180%) ThyroglobulinMicrosomehemagglutination1OOxlOOx (normal:<1OOx)<1 00x) 382 Internal Medicine Vol. 37, No. 4 (April 1998) Lingual Thyroid and Hypothyroidism percent: 23.2%). Serum thyroglobulin level was extremely high thyroid with homogeneous uptake of radiolabeled iodine (Fig. (780 ng/ml), and remained at a high level during hospitalization 2). The uptake was 6.5% at 3 hours and 8.1% at 6 hours. (between 400 and 600 ng/ml). Basal metabolic rate (BMR) was -13%. Computed tomography (CT) of the pharynx revealed a Discussion round mass with high intensity in the mid region at the base of the tongue (Fig. 1). Ultrasonography of the neck revealed a Lingual thyroid is a relatively rare developmental anomaly. mass with echogenicity similar to that of thyroid tissue. No Montgomery (3) first suggested the basic diagnostic criteria thyroid tissue was found in the typical location in the lower for establishing the diagnosis: (i) a lingual mass between the neck. Diagnosis of ectopic thyroid was confirmed by 123I epiglottis and circumvallate papillae, (ii) identification of thy- scanning. 123I thyroid scintigraphy demonstrated a lingual roid tissue by biopsy, and (iii) hypothyroidism after surgical removal. Wardet al (4) later amendedthis to include uptake of radioactive iodine at the base of the tongue. It has a prevalence of approximately 1 per 100,000 to 300,000 patients (5, 6) and 1 in 4,000 cases of thyroid disease (7). Buckman and Pa (8) reported that of 140 female patients with lingual thyroid, 55% became symptomatic between the ages of 18 and 40 years, and commonpresenting symptoms are dysphagia, dyspnea, and dysphonia (9-12). Occasionally respiratory difficulty or hemorrhage can occur (13). Female patients predominated in all reported series, ranging from 75% to 89% of the cases (3, 6, 9, 14). Although there may be other thyroid tissue present, in approximately 70% of cases, the lingual thyroid is the only functioning thyroid tissue (3, 15-17). Ectopic thyroid tissue may be the site of development of a colloid goiter, hypo or hyperthyroidism, adenoma, or even carcinoma. An ectopic thyroid tissue not clinically apparent maybecomeevident when such a pathological condition develops in it.
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