ABERRANT
LINGUAL THYROID
REPORT
OF A CASE*
HERMAN RUBIN, BROOKLYN,
B
S., male, age thirty years, was admitted to the hospita1 on January 16th, 1933, l with a Iive year history of a graduaIIy increasing Iump on the back of his tongue which interfered with degIutition. OnIy Iiquids and semi-solids couId be swallowed, and this was occasionaIIy foIIowed by choking and coughing. His speech was impaired, his voice being thick and non-resonant. During this period he had gained about thirty pounds in weight. Examination revealed a maIe aduIt presenting a11 the signs of myxedema, such as sIuggish mentaIity, swoIIen face, thickened Iips and eyelids, edema of the hands and feet, Ioss of hair, dryness and roughness of the skin. No evidence of a thyroid gIand couId be found upon examination of the Iower part of the neck. The trachea could be readiIy paIpated. The tongue presented a Iarge, In-m, soIid duI1 red mass, posteriorIy in the midIine, extending to the entire undersurface of the tongue above the hyoid bone. Mirror examination showed this mass extended downward and backward toward the pharyngea1 waI1 obscuring the view of the epigIottis and the remainder of the larynx. Direct Iaryngoscopy demonstrated that the tumor mass was central and attached to the posterior surface of the tongue, with many Iarge veins seen traversing it. It was impossible to eIevate the tongue with the tip of the Iaryngoscope, so that a view of the Iarynx was unobtainable. A smaI1 piece of tissue was removed for biopsy. This was folIowed by a severe hemorrhage which was finaIIy controIIed by the electric cautery. LateraI x-ray picture of the neck reveaIed a large mass extending downward from the tongue into the pharynx, depressing the epigIottis and narrowing the Iumen of the oropharynx. The tumefaction reached into the subIingua1 region and appeared to be pushing the hyoid bone downward. The basa1 metaboIic rate on admission was minus 26. Under thyroid therapy this rose and * Front the OtoIaryngoIogical
M.D.
N. Y.
became normaI, and the symptoms of myxedema disappeared within two weeks. The patient became more aIert mentaIIy, the edema of the face and legs was no longer present whiIe the skin became smooth and moist. The x-ray study (Fig. I) would indicate that the patient had both a IinguaI and a subIingua1 thyroid tumor. AIthough this mass was causing difficuIty in swaIIowing, it was feIt that surgery shouId not be done, because of the myxedma. Even if this were not so, the severe hemorrhage that foIIowed attempted remova of the IinguaI thyroid tissue, might be repeated and endangered the patient’s Iife. In addition, no thyroid tissue couId be detected in the neck. UnfortunateIy, the biopsy specimen was Iost. However, it was feIt that we were deaIing with some form of degeneration of the thyroid tissue, which originaIIy made up the mass. Of the aberrant tumors of the thyroid, those in the IinguaI region are probabIy the rarest. In 7600 cases of thyroid disease at the Lahey Clinic only 2 were encountered and only 2 in 4000 cases at the University Lingua1 thyroids can of PennsyIvania. cause asphyxia in the newborn, troubIe-
some hemorrhage, myxedema and on rare occasion may be the seat of carcinoma. Approximately 150 cases have been reported in the Iiterature. This condition occurs about eight times as often in maIes as in femaIes. The average age is thirty years. What was probably the first case of lingua1 thvroid was described by Hickman9 in 1869. &s patient was a newborn infant who appeared to be asphyxiated, and palpation reveaIed a mass Ming the oral cavity. Death occurred before a life-saving tracheotomy couId be done. Postmortem examination reveaIed a IinguaI goitre com-
Service of Dr. Louis C. Menger, Bushwick HospitaI.
150
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No. I
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Lingual
Thyroid
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edema in which the tumor was removed pletely fiIling the pharynx. Meixner” because of attacks of bIeeding and difficulty reported a case in which the chiId never breathed, postmortem examination re- in swallowing. Smythe2* reported an intervealed a Iarge growth pressing upon the epigIottis. Perkins,21 quoting Aschoff, reported the case of a six months oId infant, who died of myxedema, in which a Iingual thyroid was found at postmortem. New’s18 patient was probably the youngest in the Iiterature. A male infant of three and one-half months had had diffIcuIty in breathing and swaIIowing since he was two weeks oId, because of a mass on the dorsum of the tongue. New has seen the Iargest number I0 cases, probabIy observed by any individua1. In none of his cases was the mass large enough to cause alarming symptoms so no treatment was required. Bernays2 in 1888, described the first of IinguaI thyroid. The operated case patient, a gir1 of seventeen years, had both a sublingua and a Iingual thyroid. Both tumors were removed by means of an incision extending from the inferior maxiIIa to the hyoid bone, which divided a11 the roentgenogram showing irreguIar FIG. 1. Lateral structures down to the tumor. tumor mass at the tongue base and large rounded Spontaneous hemorrhage from the Iarge tumor mass filling sublingual region. vesseIs coursing across the tumor is nto uncommon. According to Grace and Weeks6 esting case in a female of seventy-nine this occurred in 8 of the 81 cases which years, who choked whiIe eating canned Dore collected. Hemorrhage is frequentIy corn. Her son attempted to remove the encountered when tissue remova for biopsy foreign body with his finger and caused a is done. This occurred in the writer’s case. severe hemorrhage. Kramer’s’l patient Rabinowitz23 reported a case in a femaIe, suffered with occasional profuse, hemorage forty-seven years, who for four months rhages from the mouth over a period of ten had a mass on the posterior surface of the years. Her basa1 metaboIic rate before tongue which occupied aImost the entire operation was normal. Ten days after thickness of the tongue and extended we11 removal of the tumor she showed signs of into the suprahyoid region. FoIIowing a myxedema. section for biopsy severe bIeeding occurred Two cases of a IinguaI thyroid attached which required severa mattress sutures for by a pedicIe have been described. Bishop3 its control. The tumor was not removed bereported a case in which the Iingual thyroid cause of the hemorrhage and the presence was connected to the base of the tongue by of myxedema. There were no symptoms of a pedicIe and was removed under IocaI pharyngea1 or IaryngeaI obstruction and no anesthesia with a tonsi snare. Harvey’ had thyroid tissue was to be found in the neck. a similar experience with a female of PearIman20 and Porter22 described hemtwenty-six years, who was a cretin and orrhage as a symptom. Monroe and Iooked Iike a chiId of eight years. She had Taylor’s” patient had symptoms of myxno paIpabIe thyroid tissue in the neck. The
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Rubin-Aberrant
growth was removed because of severe hemorrhages, dyspnoea and dysphagia. Myxedema is frequentIy encountered in Iingual thyroids. A basal metabohsm shouId aIways be done in this condition before contempIating any operative procedure. The percentage of cases of myxedema occurring after operations on the IinguaI thyroid, has been estimated by various authors to range from 15 to more than 50 per cent. In the absence of other demonstrabIe thyroid tissue, compIete removal of the IinguaI thyroid is definiteIy contraindicated. The frequency of myxedema is reIativeIy great foIIowing its removal The experiences of Lahey,12 MiIIer, l6 ZiegeIman, 26 Bisi, 4 CatteII and Hoover,5 Harvey,’ Haynes,s Lenzi,13 and Kramer-l’ prove this to be so. CatteII and Hoover5 report the case of a woman of sixty-two years, who had a growth on the tongue for forty years. She deveIoped smaI1 masses in the skin of her back and which Iater became scattered over her body. The thyroid gIand couId not be paIpated. At postmortem it was thought that death resuIted from melanosarcoma with widespread metastases. Microscopic examination however showed that these growths were Ieukemic noduIes. SimiIar deposits were found in the trachea, Iymph nodes and other organs. Laheyl2 reported a femaIe of twenty-five years, who had had a tumor at the base of the tongue since birth. Her basal metaboIism before operation was normaI. TweIve days after operation it was minus 18. After two months thyroid extract medication it became pIus rg. Lenzi13 myxedema reported a case in which deveIoped foIIowing the removal of a tumor in the submaxiIIary region and another at the base of the tongue. Examination showed both tumors to contain thyroid tissue. HiIIlO reported a case in a Negress, nine years oId, who had a Iump on the tongue for three years. No thyroid couId be feIt in the neck. Goiters were present in both the mother and grandmother. WhaIen25 reported a case in a femaIe of
Lingua1
Thyroid
APRIL,
1936
twenty-four years, in which the size of the lingua1 thyroid was reduced by puncture with the endothermy eIectrode without deveIoping symptoms of myxedema. ZiegeIman26 states that St. CIair Thomson was the first to suggest the use of eIectrocoagulation in the treatment of IinguaI goitre. Owenslg describes the case of a femaIe of thirty-six years who did not deveIop myxedema nine months after remova of a IinguaI tumor. Although he couId not find any thyroid in the neck, he feIt that she probabIy had thyroid tissue elsewhere. Carcinoma occurring in a Iingual thyroid is a rarity, onIy 2 cases having been reported in the Iiterature. Ashhurst and White’ report the case of a maIe, fifty-six years of age, who had a Iump on the back of his tongue for more than twenty years. Five years after this was lirst noticed, he began to complain of pain. The mass was considered as IinguaI tonsi tissue and therefore removed. BilateraIIy enIarged gIands of the neck appeared, three years after operation. The tongue growth graduaIIy returned. The mass was hard and sIightIy uIcerated, and was removed with the eIectric cautery knife, under intratrachea1 ether anesthesia. This proved to be adenocarcinoma of a Iingual thyroid. Levi and Hankins14 reported the second case in a femaIe of twenty-one years, who had a mass on the posterior portion of the tongue. SwaIIowing produced pain, choking and coughing. A smaI1 mass in the region of the posterior surface of the tongue, was removed by actua1 cautery under ether anesthesia. Biopsy reveaIed a Iow grade carcinoma arising in the thyroid tissue. She deveIoped an edema of the Iarynx on the second day foIlowing operation which necessitated a tracheotomy. The tube was removed two weeks Iater. CONCLUSIONS
One must aIways bear in mind the possibiIity of the presence of an aberrant thyroid tumor on the posterior dorsum of the tongue. The diagnosis is estabIished
Naw SERIESVOL. XxX11, No.
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Rubin-Aberrant
when no thyroid tissue is detectabIe in the neck and unusuaI vascuIarity is noted. The usua1 picture presented is that of a smooth, rounded mass in the preepiglottic region with reIativeIy Iarge blood vesseIs traversing its surface. Sectioning for biopsy may and at times, seriousIy mean severe, troubIesome hemorrhage. AIso the thyroid tissue at the tongue base may be a11 that the patient possesses, so that its remova wiI1 cause myxedema and necessitate taking thyroid extract for the remainder of one’s Iife. Carcinoma of the IinguaI thyroid is unusuaIIy rare. SUMMARY
A case is reported in which a patient had an extensive tumor in both the IinguaI and subIingua1 regions, with myxedema as the predominant picture. The condition cIeared after the administration of thyroid extract. The importance of knowing the patient’s metaboIic rate is stressed. The hemorrhage incidenta to biopsy was so severe as to require eIectrocautery for its control. The value of x-ray films in the Iatera1 pIane of the tongue base region, is demonstrated. REFERENCES I. ASHHURST, A. P. C. and WHITE, C. Y. Carcinoma in aberrant thyroid at base of tongue. Jour. Am. Med. Assn., 85: 1219-1220 (Oct. 17) x925. 2. BERNAYS, A. C. Operation of a rare tumor of the root of the tongue. St. Louis Med. and Surg. Jour., 55: 201 (Oct.) 1888. 3. BISHOP, F. J. Lingual thyroid. Ann. Otol. Rbin. Lary., 43: 294-298, (March) 1934. 4. BISI, H. Aberrant thyroid at base of tongue., Revue Ass. Med. Argentine, 46: 1467-1468 (Nov.) 1932.
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Thyroid
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5. CATTELL, R. B. and HOOVER, W. B. Lingua1 thyroid, report of case. Surg. Clin. N. Amer., g: 1355-1362 (Dec.) 1929. 6. GRACE, R. V. and WEEKS, C. Lingua1 goitre, Ann. Surg., 96: 973-978, (Dec.) 1932. _ 7. HARVEY. F. Pedunculated thvroid tumor at base of tongue. Brit. Jour. Surg.: 13: 746-748 (April) 1926. 8. HAYNES, I. S. SubIinguaI thyroid. Ann. Surg., 56: 465-467, 1912. g. HICKMAN, W. CongenitaI tumor of the base of the tongue, causing death by suffocation. Trans. Path. Sot., London, 20: 160, 1869. IO. HIL.L, R. C. Lingua1 thyroid. Jour. Med. Ass. Alabama, 3: 103-104. Sept., 1933. I I. KRAMER, R. Lingua1 Thyroid. Laryngoscope, 39: 546 (Aug.) 1929. 12. LAHEY, F. H. Lingual Goitre. Surg. Gynec. Obst., 36: 395, 1923. 13. LENZI. I. Due strume a sede rara; contribute a110 studio de1 grozzo IuiguaIe. Sperimentale. Arch. di. biol., 59: 852, 890, 1905. 14. LEVI, L. M. and HANKINS, F. D. Carcinoma of the lingual thyroid. Amer. Jour. Cancer, 23: 328-333. Feb.1 1935. 15. MEIXNER, K. Zwei FaIIe von struma baseos Iinguae. Deutscb. Ztscbr. f. Cbir., 88: I-19, 1905. 16. MILLER, R. H. Lingua1 goitre. New England Med. Jour., 208: 480-484, (Mar. 2) 1933. 17. MONRO, T. K. and TAYLOR, M. L. Lingua1 goitre, remova foIlowed by myxedema. Glasgow Med. Jour., 105: 213-215 (Mar.) 1926. 18. NEW, G. B. CongitaI obstruction of Iarynx and pharynx. Jour. Am. Med. Assn., 81: 363 (Aug. 4) ’ 923. 19. OWENS, M. J. Lingua1 thyroid. Jour. Kansas Med. Sot., 26: 160, 1926. 20. PEARLMAN, H. B. Lingua1 thyroid. Arch. Oto-
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19: 594-599
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2 I. PERKINS, C. E. Accessorv thvroid at base of tonaue. Laryng. 18: 292-296, igo8. 22. PORTER, L. B. Lingual thyroid. Rbode Is. Med. Jour., 17: 103-104 (June) 1934. 23. RABINOWITZ, M. A. Aberrant thyroid tumor of the tongue. Surg., Gynec. Obst., 27: rgI-194, 1918. 24. SMYTHE, H. E. Accessory thyroid tumors of the tongue. Ann. Otol., Rbin., Laryng., 20: 367, Igrr. 25. WHALEK, E. J. Lingua1 goitre. Laryngoscope. 44: 555-567 (JuIy) ‘934. 26. ZIEGELMAN, E. F. Lingua1 goitre. Arch. Otolaryng., 16: 496505, Oct., 1932.