Br. J. Surg. Vol. 62 (1975) 689491
Carcinoma arising in thyroglossal duct remnant : case reports and review of the literature P R A K A S H C. S A H A R I A * SUMMARY
Two cases of thyroglossal duct cyst carcinoma are presented and the world literature is reviewed. There are only 74 cases reported to date. In nearly all the cases the clinical diagnosis was thyroglossal cyst. Although the great majority of the tumours were papillary adenocarcinomas, 13.15 per cent were of other histological types. The treatment has been quite variable, but the most common initial treatment was Sistrunk‘s (1928) operation. Following the establishment o j the diagnosis of malignancy, thyroidectomy was done in several cases, but it failed to show any evidence of malignancy in most of the thyroids removed, which establishes firmly the de novo origin of these tumours from the thyroglossal duct remnarit. In only one case had tumour disseminated to distant organs, the lung, liver, etc. The scepticism as to whether some of the carcinomas associated with thyroglossal duct remnants may not in fact represent metastases from a small primary tumour of thyroidgland is examined. It appears that, though a possibility of primary or metastatic tumour in the thyroid does exist, the probability does not appear to be high. From our own experience it is suggested that local excision followed by radiotherapy, irrespective of recurrence, may be worth considering for the treatment of such carcinomas.
was a well-differentiated squamous cell carcinoma invading the surrounding tissue and strap muscles. The postoperative course was uneventful and the patient has remained free of any evidence of recurrence for 3 years following surgery. Case 2: A 54-year-old white male was admitted to Wordsley Hospital, Worcester, in August 1970 with a neck mass of 3 months’ duration. Physical examination revealed a smooth, cystic, spherical, non-tender, 5 x 4-cm mass overlying the left thyroid cartilage. The mass moved with swallowing and protrusion of the tongue. The thyroid gland felt normal in size and consistency. There were no enlarged cervical lymph nodes. The mass was removed through a collar incision along with the adherent strap muscles, left hyoid bone, part of the thyroid cartilage and the thyroglossal duct en bloc. The thyroid gland was exposed and was found to be normal. No enlarged cervical glands were detected. Examination of the specimen revealed a cyst 5 x 4 x 2 c m with a thick fibrous wall and an irregular fibrotic plaque in the wall of the cyst. Microscopically, the wall was fibrotic with foci of active and chronic inflammation. In parts of the cyst, groups of papillary elements were found which consisted of orderly, well-differentiated neoplastic cells consistent with papillary adenocarcinorna. Some follicular neoplastic elements containing colloid were also present. Tumour cells were observed in the overlying strap muscles (Fig. 1). The patient’s postoperative recovery was uneventful. In view of the muscle invasion, the possibility of incomplete excision and the fact that the patient was a poor surgical risk, a course of radiotherapy was given. There is no evidence of recurrence after 3 f years’ follow-up.
CARCINOMA arising in a thyroglossal duct remnant is rare. Only 74 cases have been reported in the world literature to date. Most have been papillary adenocarcinomas, but squamous cell lesions have also been documented. The present report adds 2 cases to the literature and documents common clinical data for this group of unusual tumours. Case reports Case 1 : An 81-year-old white female was admitted to Derby City Hospital (Eng.) in February 1971 with a mass in her neck of 4h months’ duration. I t was painless but increasing in size rapidly. There were no related symptoms. Physical examination revealed a 4 x 5-cm mass at the level of the thyroid cartilage, slightly to the left of the midline. It was smooth, spherical in shape and firm in consistency. It was adherent to the hyoid bone, but was not attached to the overlying skin. The mass moved with swallowing and protrusion of the tongue. There were no palpable cervical lymph nodes, and the thyroid gland felt normal in size and consistency. X-ray of the chest showed no abnormality. Excision of the mass, including the left hyoid bone, strap muscles and part of the thyroid cartilage and thyroglossal duct up to the foramen caecum, was carried out en bloc through a collar incision. The thyroid gland was exposed and was found to be normal. Exploration of the neck for enlarged cervical glands was negative. Examination of the specimen revealed a cyst, 6 c m in diameter, containing a nodule in the wall. The wall of the cyst was lined by squamous cell epithelium and the nodule itself
50
Fig. 1. Papillary adenocarcinoma with follicular neoplastic elements containing colloid. ( x 180.)
Discussion The thyroid gland forms at about the fourth week of embryonic life from a median diverticulum of the ventral pharyngeal wall at a position marked in adult life by the foramen caecum. As the thyroid gland migrates downwards, it forms a duct behind it in its path. The duct can be lined by squamous, columnar or transitional epithelium. Ordinarily the duct disappears at the sixth or seventh week of fetal life.
* Department of Surgery, The Johns Hopkins University, Baltimore, Maryland, USA. 689
Prakash C. Saharia thyroglossal duct or sinus origin they would be expected to be of the squamous cell type. Thyroid tissue has been found with thyroglossal duct remnants by others, however, in incidences ranging from 3 to Horizontal plane Midline 27 36.5 per cent (Ward et al., 1949; Choy et al., 1964). Left of midline 9 In addition, 4 cases of squamous cell carcinoma 4 Right of midline (Owen and Inglesby, 1927; Nachlas, 1950; Shepard Not mentioned 36 and Rosenfeld, 1968; Mobini et al., 1974) arising from Vertical plane the thyroglossal duct have been reported. Further13 Suprahyoid more, it is certainly possible that the stomodeal 9 Level of hvoid epithelium of the thyroglossal duct with its ernbryoInfrahyoid 16 Not mentioned 31 logical potential of thyroidogenesis could produce a neoplasm resembling that of thyroid origin. There is no unanimity of opinion regarding the Table 11: HISTOLOGY OF THE TUMOURS surgical approach to such tumours. In most cases Histology % __ No. Sistrunk’s (1928) operation was carried out. Following Papillary adenocarcinoma 66 86.84 histological diagnosis of malignancy, further treatment 2 2.6 Adenocarcinoma has included re-excision of local tissues, bilateral block 5 6.57 Squafious cell carcinoma neck dissection, thyroid administration and radio3 3.94 Mixed follicular and DaDillarv carcinoma iodine therapy (Zamrazil et al., 1971). Although thyroglossal duct cyst presents as a swelling slightly lateral to the midline, developmentally the thyroglossal Table 111: FOLLOW-UP OF 76 CASES duct cyst is a midline structure. Malignancy arising No. of cases: 47 Time: Less than 5 years within it, therefore, can metastasize to lymph nodes 13 5-10 years on either side of the neck. Unilateral dissection has no 5 11-15 years 2 16-20 years place in the prophylaxis of spread, since it is impossible 2 Over 20 years to predict which side (if either) will be the site of 7 No follow-up further metastasis. The follow-up of the reported cases ranges from 3 months to 28 years. Only 13 cases of Incomplete obliteration, however, may lead to the recurrence have been reported. Twenty-nine cases development of a thyroglossal duct cyst which can have been followed up for more than 5 years (Table III). appear in the midline anywhere between the foramen In only 1 case (Maxwell and Marchetta, 1960) were caecum and the suprasternal notch. In addition, there distant metastases. The remaining instances thyroid rests can appear along this tract. were local recurrences treated by repeated local A survey of literature reveals 74 cases, and with the excisions or block dissection. addition of the present 2 cases a total of 76 cases Because of the paucity of cases the long term becomes available for analysis. prognosis is not clearly known. However, there is no Clinically, the presenting symptom has generally reason to believe that this tumour should have been a gradually increasing cervical mass with variable different biological behaviour from carcinoma of the location (Table I ) , in some cases associated with thyroid gland. hoarseness and dysphagia. The known duration of the mass prior to surgery ranges from 10 days to 15 Acknowledgements years. In the majority of cases the thyroid gland was I would like to thank Mr A. W. Anderson, Consultant, reported to be normal on clinical and gross examina- City Hospital, Derby, and Mr M. Hershman, Contion. Only 2 out of the 76 cases were associated with sultant, Wordsley Hospital, near Stourbridge, for carcinoma of the isthmus and the right lobe of the permitting me to publish their cases. I am also grateful thyroid (Rees and Brown, 1953; Mobini et al., 1974). to Dr John L. Cameron, Associate Professor of The pathological diagnoses were papillary adeno- Surgery at the Johns Hopkins Hospital, Baltimore, carcinoma in 66 cases, adenocarcinoma in 2, for his help and guidance in preparing this manuscript. squamous cell carcinoma in 5 and mixed follicular and papillary carcinoma in 3 (Table ZZ). The case reported References* by Dalgaard and Wetteland (1956) appears to have AKBARI Y.,RICHTER R . M. and PAPADAKIS L. K . (1967) been a solid undifferentiated carcinoma and bears Thyroid carcinoma arising in thyroglossal duct resemblance to the recently recognized medullary remnants. Arch. Surg. 94, 235-239. carcinoma with amyloid stroma. ARONOFF B. L. (1952) Papillary thyroid cancer originatJudd (1963) has stated that thyroid tissue in and ing in thyroglossal cyst. Am. Surg. 18, 362around the thyroglossal duct is rarely encountered 371. and that the occasional recording of papillary BERGERA J. I. and HORWITZ A. (1961) Papillary adenocarcinoma of a thyroglossal cyst or sinus is erroneous. carcinoma arising in a thyroglossal duct cyst. He contends that when a papillary carcinoma is Am. Surg. 27, 759-761. encountered in this region the thyroid gland is always the source, and that if these were true carcinomas of * Including the references of all the cases reported to date.
Table 1: LOCATION OF THE PRESENTING MASS IN THE NECK IN 76 CASES No. of cases Location of mass
690
Carcinoma in thyroglossal duct remnant BHAGAWAN B. S., GORWIDA RAO D. R.
and WEINBERG
T.
(1 970) Carcinoma of thyroglossal duct cyst: case
report and review of literature. Surgery 67, 28 1-292. BOCHETTO J. F., MOUTOYE A. and SUNDE E. A. (1962) Papillary carcinoma of the thyroid in thyroglossal duct cysts. Am. J . Surg. 104, 773-776. BUTLER E. c., DICKEY J. R., SHILL 0.s. and SHALAK E. (1969) Carcinoma of the thyroglossal duct remnant. Laryngoscope 79, 264271. BYRNE J. J . (1952) Carcinoma in a thyroglossal cyst: report of a case. Bost. Med. Q. 3, 86-89. CHOY F. J., WARD R. and RICHARDSON R. (1964) Carcinoma of the thyroglossal duct. Am. J . Surg. 108 361-369. CLAWSON J. and KIMBALL H. (1962) Papillary adenocarcinoma of the thyroglossal duct cyst. Ariz. Med. 19, 80-8 I . CLUTE H. M. and CATTELL R. B. (1930) Thyroglossal cysts and sinuses. Ann. Surg. 92, 57-66. COON w. w. (1 965) Thyroid carcinoma-three unusual problems in diagnosis and management. Am. J. Surg. 109, 629-633. DALGAARD J. B. and WETTELAND P. (1956) Thyroglossal anomalies. Acta Chir. Scand. 111, 444455. FALKINBURG L. w., HOEY w. 0.and STUART J. R. (1966) Papillary adenocarcinoma arising in a thyroglossal duct cyst. Oral Surg. 21, 358-360. FISH J. and MOORE R. M. (1963) Ectopic thyroid tissue and ectopic thyroid carcinoma: a review of the literature and report of a case. Ann. Surg. 157, 2 12-222. GHENT w. R. and WAUGH D. (1960) Carcinoma in a thyroglossal remnant. Can. J. Surg. 4, 79-81. GURAIEB s. R. and VIETA L. J. (1962) Carcinoma in a thyroglossal cyst. Report of a case. Arch. Otolaryngol. 75, 457459. HAYS L. L. and MARLOWE J. F. (1968) Papillary adenocarcinoma arising in thyroglossal duct cyst case report and literature review. Laryngoscope 78, 2189-2203. HILL D. P. (1961) Papillary carcinoma arising in a thyroglossal tract. Can. Med. Assoc. J. 85, 791-793. JAQUES D. A., CHAMBERS R. G. and OERTEL J. E. (1970) Thyroglossal tract carcinoma. Am. J. Surg. 120, 439446. JUDD E. s. (1963) Thyroglossal duct cysts and sinuses. Surg. Clin. North Am. 43, 1023-1032. KALDERON A. E. and COHN J. D. (1966) Papillary adenocarcinoma in a thyroglossal cyst. Case report and review of literature. Cancer 19, 839-843. KEELING J. H. and OCHSNER A. (1959) Carcinoma in thyroglossal duct remnants. Cancer 12, 596-600. LATIMER R. G., SNOW E. and HICKS H. G. (1 968) Papillary adenocarcinoma arising in a thyroglossal duct remnant. Arch. Surg. 97, 161-162. MARSHALL s. F. and BECKER w. F. (1949) Thyroglossal cysts and sinuses. Ann. Surg. 129, 642-651. MAXWELL w. T. and MARCHETTA F. E. (1960) Papillary adenocarcinoma of the thyroglossal duct tract. Arch. Surg. 80, 224-225.
and KLINGHOFFER J. F. (1974) Squamous cell carcinoma arising in a thyroglossal duct cyst. Am. Surg. 40, 290-294. MOULIK B. N., PRAKASH A., KUMAR v. and SAMPATH A. (1974) Carcinoma of thyroglossal duct remnant: case report and review of the literature. Am. Surg. 40, 248-252. NACHLAS N. E. (1950) Thyroglossal duct cysts. Ai7n. Otol. Rhinol. Laryngol. 59, 381-390. NOGUCHI S., NOGUCHI A. and MURAKAMI N. (1970a) Papillary carcinomas of the thyroid. I. Developing pattern of metastasis. Cancer 26, 1053-1060. NOGUCHI S., NOGUCHI A. and MURAKAMI N. (1970b) Papillary carcinoma of the thyroid. 11. Value of prophylactic lymph node excision. Cancer 26, 1061-1 064. NUTTAL F. Q. (1965) Cystic metastases from papillary adenocarcinoma of the thyroid with comments concerning carcinoma associated with thyroglossal remnants. Am. J . Surg. 109, 500-505. O’KANE c. R. and STRAUS F. H. (1953) Papillary adenocarcinoma arising in association with a persistent thyroglossal duct. Ann. Surg. 138, 805-806. OWEN H. R. and INGELSBY H. (1927) Carcinoma of thyroglossal duct. Ann. Surg. 85, 132-136. POWELL L. w., BYERS T. F. and SCIORTINO A. (1965) Thyroglossal duct carcinoma. Arch. Otolaryngol. 82, 189-190. PAGE c. P., KEMMERER w. T., HAFF R. c. and MAZZAFERRI E. L. (I 974) Thyroid carcinoma arising in thyroglossal ducts. Ann. Surg. 180, 799-803. REES c. E. and BROWN M. J. (1953) Cysts of the thyroglossal duct. Am. J. Surg. 85, 597-599. RUPPMANN E. and GEORGSSON G. (1966) Squamous carcinoma of thyroglossal duct. Ger. Med. Mon. 11,442-447. SHEPARD G. H. and ROSENFELD L. (1968) Carcinoma of thyroglossal duct remnants. Am. J. Surg. 116, 125-129. SISTRUNK w. E. (1928) Technique of removal of cysts and sinuses of the thyroglossal duct. Surg. Gynecol. Obstet. 46, 109-1 12. SNEDECOR P. A. and GROSHONG L. E. (1965) Carcinoma of the thyroglossal duct. Surgery 58, 969-978. TANAKE K. and CINN w. H. (1963) Cancer arising in thyroglossal duct remnant. Arch. Surg. 86, 466477. THOMAS c. G. jun. (1971) Thyroid cancer. In: WERNER s. c. and INGBAR s. H. (ed.) The Thyroid, 3rd ed. London, Harper & Row, p. 456. VERONESI u., GENNAIR L. and CASCINELLI N. (1964) Carcinoma papillifero insorto su cisti del dotto tireglosso. Tumori 50, 317-321. WARD G. E., HENDRICK J. w. and CHAMBERS R. G. (1949) Thyroglossal tract abnormalities, cysts and fistulae. Surg. Gynecol. Obsret. 89, 727-734. WEBER A. 0. (1968) Carcinoma of thyroglossal duct. Calif. Med. 108, 127-130. MOBINI J., KROUSE T. B.
ZAMRAZIL V. NEMEC J., PROKS C., NIEDERLE B., MANAK J. and ZEMAN K. (1971) Primary carcinoma of the
thyroglossal duct treated by radio-iodine. Neoplasma 18, 421-426.
691
Carcinoma arising in thyroglossal duct remnant : case reports and review of the literature P R A K A S H C. S A H A R I A * SUMMARY
Two cases of thyroglossal duct cyst carcinoma are presented and the world literature is reviewed. There are only 74 cases reported to date. In nearly all the cases the clinical diagnosis was thyroglossal cyst. Although the great majority of the tumours were papillary adenocarcinomas, 13.15 per cent were of other histological types. The treatment has been quite variable, but the most common initial treatment was Sistrunk‘s (1928) operation. Following the establishment o j the diagnosis of malignancy, thyroidectomy was done in several cases, but it failed to show any evidence of malignancy in most of the thyroids removed, which establishes firmly the de novo origin of these tumours from the thyroglossal duct remnarit. In only one case had tumour disseminated to distant organs, the lung, liver, etc. The scepticism as to whether some of the carcinomas associated with thyroglossal duct remnants may not in fact represent metastases from a small primary tumour of thyroidgland is examined. It appears that, though a possibility of primary or metastatic tumour in the thyroid does exist, the probability does not appear to be high. From our own experience it is suggested that local excision followed by radiotherapy, irrespective of recurrence, may be worth considering for the treatment of such carcinomas.
was a well-differentiated squamous cell carcinoma invading the surrounding tissue and strap muscles. The postoperative course was uneventful and the patient has remained free of any evidence of recurrence for 3 years following surgery. Case 2: A 54-year-old white male was admitted to Wordsley Hospital, Worcester, in August 1970 with a neck mass of 3 months’ duration. Physical examination revealed a smooth, cystic, spherical, non-tender, 5 x 4-cm mass overlying the left thyroid cartilage. The mass moved with swallowing and protrusion of the tongue. The thyroid gland felt normal in size and consistency. There were no enlarged cervical lymph nodes. The mass was removed through a collar incision along with the adherent strap muscles, left hyoid bone, part of the thyroid cartilage and the thyroglossal duct en bloc. The thyroid gland was exposed and was found to be normal. No enlarged cervical glands were detected. Examination of the specimen revealed a cyst 5 x 4 x 2 c m with a thick fibrous wall and an irregular fibrotic plaque in the wall of the cyst. Microscopically, the wall was fibrotic with foci of active and chronic inflammation. In parts of the cyst, groups of papillary elements were found which consisted of orderly, well-differentiated neoplastic cells consistent with papillary adenocarcinorna. Some follicular neoplastic elements containing colloid were also present. Tumour cells were observed in the overlying strap muscles (Fig. 1). The patient’s postoperative recovery was uneventful. In view of the muscle invasion, the possibility of incomplete excision and the fact that the patient was a poor surgical risk, a course of radiotherapy was given. There is no evidence of recurrence after 3 f years’ follow-up.
CARCINOMA arising in a thyroglossal duct remnant is rare. Only 74 cases have been reported in the world literature to date. Most have been papillary adenocarcinomas, but squamous cell lesions have also been documented. The present report adds 2 cases to the literature and documents common clinical data for this group of unusual tumours. Case reports Case 1 : An 81-year-old white female was admitted to Derby City Hospital (Eng.) in February 1971 with a mass in her neck of 4h months’ duration. I t was painless but increasing in size rapidly. There were no related symptoms. Physical examination revealed a 4 x 5-cm mass at the level of the thyroid cartilage, slightly to the left of the midline. It was smooth, spherical in shape and firm in consistency. It was adherent to the hyoid bone, but was not attached to the overlying skin. The mass moved with swallowing and protrusion of the tongue. There were no palpable cervical lymph nodes, and the thyroid gland felt normal in size and consistency. X-ray of the chest showed no abnormality. Excision of the mass, including the left hyoid bone, strap muscles and part of the thyroid cartilage and thyroglossal duct up to the foramen caecum, was carried out en bloc through a collar incision. The thyroid gland was exposed and was found to be normal. Exploration of the neck for enlarged cervical glands was negative. Examination of the specimen revealed a cyst, 6 c m in diameter, containing a nodule in the wall. The wall of the cyst was lined by squamous cell epithelium and the nodule itself
50
Fig. 1. Papillary adenocarcinoma with follicular neoplastic elements containing colloid. ( x 180.)
Discussion The thyroid gland forms at about the fourth week of embryonic life from a median diverticulum of the ventral pharyngeal wall at a position marked in adult life by the foramen caecum. As the thyroid gland migrates downwards, it forms a duct behind it in its path. The duct can be lined by squamous, columnar or transitional epithelium. Ordinarily the duct disappears at the sixth or seventh week of fetal life.
* Department of Surgery, The Johns Hopkins University, Baltimore, Maryland, USA. 689
Prakash C. Saharia thyroglossal duct or sinus origin they would be expected to be of the squamous cell type. Thyroid tissue has been found with thyroglossal duct remnants by others, however, in incidences ranging from 3 to Horizontal plane Midline 27 36.5 per cent (Ward et al., 1949; Choy et al., 1964). Left of midline 9 In addition, 4 cases of squamous cell carcinoma 4 Right of midline (Owen and Inglesby, 1927; Nachlas, 1950; Shepard Not mentioned 36 and Rosenfeld, 1968; Mobini et al., 1974) arising from Vertical plane the thyroglossal duct have been reported. Further13 Suprahyoid more, it is certainly possible that the stomodeal 9 Level of hvoid epithelium of the thyroglossal duct with its ernbryoInfrahyoid 16 Not mentioned 31 logical potential of thyroidogenesis could produce a neoplasm resembling that of thyroid origin. There is no unanimity of opinion regarding the Table 11: HISTOLOGY OF THE TUMOURS surgical approach to such tumours. In most cases Histology % __ No. Sistrunk’s (1928) operation was carried out. Following Papillary adenocarcinoma 66 86.84 histological diagnosis of malignancy, further treatment 2 2.6 Adenocarcinoma has included re-excision of local tissues, bilateral block 5 6.57 Squafious cell carcinoma neck dissection, thyroid administration and radio3 3.94 Mixed follicular and DaDillarv carcinoma iodine therapy (Zamrazil et al., 1971). Although thyroglossal duct cyst presents as a swelling slightly lateral to the midline, developmentally the thyroglossal Table 111: FOLLOW-UP OF 76 CASES duct cyst is a midline structure. Malignancy arising No. of cases: 47 Time: Less than 5 years within it, therefore, can metastasize to lymph nodes 13 5-10 years on either side of the neck. Unilateral dissection has no 5 11-15 years 2 16-20 years place in the prophylaxis of spread, since it is impossible 2 Over 20 years to predict which side (if either) will be the site of 7 No follow-up further metastasis. The follow-up of the reported cases ranges from 3 months to 28 years. Only 13 cases of Incomplete obliteration, however, may lead to the recurrence have been reported. Twenty-nine cases development of a thyroglossal duct cyst which can have been followed up for more than 5 years (Table III). appear in the midline anywhere between the foramen In only 1 case (Maxwell and Marchetta, 1960) were caecum and the suprasternal notch. In addition, there distant metastases. The remaining instances thyroid rests can appear along this tract. were local recurrences treated by repeated local A survey of literature reveals 74 cases, and with the excisions or block dissection. addition of the present 2 cases a total of 76 cases Because of the paucity of cases the long term becomes available for analysis. prognosis is not clearly known. However, there is no Clinically, the presenting symptom has generally reason to believe that this tumour should have been a gradually increasing cervical mass with variable different biological behaviour from carcinoma of the location (Table I ) , in some cases associated with thyroid gland. hoarseness and dysphagia. The known duration of the mass prior to surgery ranges from 10 days to 15 Acknowledgements years. In the majority of cases the thyroid gland was I would like to thank Mr A. W. Anderson, Consultant, reported to be normal on clinical and gross examina- City Hospital, Derby, and Mr M. Hershman, Contion. Only 2 out of the 76 cases were associated with sultant, Wordsley Hospital, near Stourbridge, for carcinoma of the isthmus and the right lobe of the permitting me to publish their cases. I am also grateful thyroid (Rees and Brown, 1953; Mobini et al., 1974). to Dr John L. Cameron, Associate Professor of The pathological diagnoses were papillary adeno- Surgery at the Johns Hopkins Hospital, Baltimore, carcinoma in 66 cases, adenocarcinoma in 2, for his help and guidance in preparing this manuscript. squamous cell carcinoma in 5 and mixed follicular and papillary carcinoma in 3 (Table ZZ). The case reported References* by Dalgaard and Wetteland (1956) appears to have AKBARI Y.,RICHTER R . M. and PAPADAKIS L. K . (1967) been a solid undifferentiated carcinoma and bears Thyroid carcinoma arising in thyroglossal duct resemblance to the recently recognized medullary remnants. Arch. Surg. 94, 235-239. carcinoma with amyloid stroma. ARONOFF B. L. (1952) Papillary thyroid cancer originatJudd (1963) has stated that thyroid tissue in and ing in thyroglossal cyst. Am. Surg. 18, 362around the thyroglossal duct is rarely encountered 371. and that the occasional recording of papillary BERGERA J. I. and HORWITZ A. (1961) Papillary adenocarcinoma of a thyroglossal cyst or sinus is erroneous. carcinoma arising in a thyroglossal duct cyst. He contends that when a papillary carcinoma is Am. Surg. 27, 759-761. encountered in this region the thyroid gland is always the source, and that if these were true carcinomas of * Including the references of all the cases reported to date.
Table 1: LOCATION OF THE PRESENTING MASS IN THE NECK IN 76 CASES No. of cases Location of mass
690
Carcinoma in thyroglossal duct remnant BHAGAWAN B. S., GORWIDA RAO D. R.
and WEINBERG
T.
(1 970) Carcinoma of thyroglossal duct cyst: case
report and review of literature. Surgery 67, 28 1-292. BOCHETTO J. F., MOUTOYE A. and SUNDE E. A. (1962) Papillary carcinoma of the thyroid in thyroglossal duct cysts. Am. J . Surg. 104, 773-776. BUTLER E. c., DICKEY J. R., SHILL 0.s. and SHALAK E. (1969) Carcinoma of the thyroglossal duct remnant. Laryngoscope 79, 264271. BYRNE J. J . (1952) Carcinoma in a thyroglossal cyst: report of a case. Bost. Med. Q. 3, 86-89. CHOY F. J., WARD R. and RICHARDSON R. (1964) Carcinoma of the thyroglossal duct. Am. J . Surg. 108 361-369. CLAWSON J. and KIMBALL H. (1962) Papillary adenocarcinoma of the thyroglossal duct cyst. Ariz. Med. 19, 80-8 I . CLUTE H. M. and CATTELL R. B. (1930) Thyroglossal cysts and sinuses. Ann. Surg. 92, 57-66. COON w. w. (1 965) Thyroid carcinoma-three unusual problems in diagnosis and management. Am. J. Surg. 109, 629-633. DALGAARD J. B. and WETTELAND P. (1956) Thyroglossal anomalies. Acta Chir. Scand. 111, 444455. FALKINBURG L. w., HOEY w. 0.and STUART J. R. (1966) Papillary adenocarcinoma arising in a thyroglossal duct cyst. Oral Surg. 21, 358-360. FISH J. and MOORE R. M. (1963) Ectopic thyroid tissue and ectopic thyroid carcinoma: a review of the literature and report of a case. Ann. Surg. 157, 2 12-222. GHENT w. R. and WAUGH D. (1960) Carcinoma in a thyroglossal remnant. Can. J. Surg. 4, 79-81. GURAIEB s. R. and VIETA L. J. (1962) Carcinoma in a thyroglossal cyst. Report of a case. Arch. Otolaryngol. 75, 457459. HAYS L. L. and MARLOWE J. F. (1968) Papillary adenocarcinoma arising in thyroglossal duct cyst case report and literature review. Laryngoscope 78, 2189-2203. HILL D. P. (1961) Papillary carcinoma arising in a thyroglossal tract. Can. Med. Assoc. J. 85, 791-793. JAQUES D. A., CHAMBERS R. G. and OERTEL J. E. (1970) Thyroglossal tract carcinoma. Am. J. Surg. 120, 439446. JUDD E. s. (1963) Thyroglossal duct cysts and sinuses. Surg. Clin. North Am. 43, 1023-1032. KALDERON A. E. and COHN J. D. (1966) Papillary adenocarcinoma in a thyroglossal cyst. Case report and review of literature. Cancer 19, 839-843. KEELING J. H. and OCHSNER A. (1959) Carcinoma in thyroglossal duct remnants. Cancer 12, 596-600. LATIMER R. G., SNOW E. and HICKS H. G. (1 968) Papillary adenocarcinoma arising in a thyroglossal duct remnant. Arch. Surg. 97, 161-162. MARSHALL s. F. and BECKER w. F. (1949) Thyroglossal cysts and sinuses. Ann. Surg. 129, 642-651. MAXWELL w. T. and MARCHETTA F. E. (1960) Papillary adenocarcinoma of the thyroglossal duct tract. Arch. Surg. 80, 224-225.
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