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Lingual thyroid and neoplastic change: A review of the literature and description of a case
$100 WINSLOW and WEISBERGER
Albright hereditary osteodystrophy with hypothyroidism, normocalcemia, and normal Gs protein activity: a family presenting with congenital osteoma curls. Am J Med Genet 1992;43:764-7. Prendiville JS, Lucky AW, Mallory SB, Mughal Z, Mimouni F, Langman CB. Osteoma curls as a presenting sign of pseudohypoparathyroidism. Pediatr Dermatol 1992;9:11-8.
OtolaryngologyHead and Neck Surgery December 1997
7. Orlow SJ, Watsky KL, Bolognia JL. Skin and bones. I. J Am Acad Dermatol 1991;25:205-22. 8. Nakamura S, Imai T, Nakayama K, Onda S. Primary osteoma curls. J Dermatol 1987;14:85-8. 9. Monroe AB, Burgdoff WHC, Sheward S, Plate-like cutaneous osteoma. J Am Acad Dermatol 1987; 16:481-4.
Lingual thyroid and neoplastic change: A review of the literature and description of a case CATHERINE P.WINSLOW, MD, and EDWARD C. WEISBERGER, MD, FACS,Denver, Colorado, and Indianapolis, Indiana
T h e embryologic development of the thyroid gland allows for anomalies in the ultimate location a!ong the course of its migration. Although ectopic remnants are common, carcinoma in such remnants is rare. Only 26 cases of lingual thyroid carcinoma were found in a review of the literature. This article reviews the literature on ectopic thyroid and associated pathology, and describes a case of carcinoma arising in the lingual thyroid.
CASE REPORT A 23-year-old white man presented on March 6, 1993, for an examination of a mass in the left side of his neck, which had been gradually increasing in size over the course of a few months. His thyroid had been enlarging since 1990, and he had taken levothyroxine for a few months, with symptomatic relief. He was otherwise asymptomatic. It was noted that another family member had hypothyroidism. Examination revealed a 5-cm mass in the region of the superior pole of the left lateral thyroid gland, overlying his jugular vein, which did not move with swallowing. The remainder of the examination was normal, including a fiberoptic examination of the pharynx and larynx. A fine-needle aspiration was taken, which revealed changes consistent with a follicular variant of papillary carcinoma. It was decided to proceed with a left thyroid lobectomy. His calcitonin level was measured to exclude the possibility of medullary carcinoma. It was found to be within normal limits. Because the mass was believed to be a thyroid malignancy, the patient was taken to surgery on March 6, 1993.
From the Department of Otolaryngology-Head and Neck Surgery, University of Colorado Health Sciences Center; Department of Otolaryngology-Head and Neck Surgery, Indiana University School of Medicine. Reprint requests: Catherine R Winslow, MD, University of Colorado Health Sciences Center, Department of Otolaryngology-Head and Neck Surgery, Box B-205, 4200 East Ninth Avenue, Denver, CO 80262. Otolaryngol Head Neck Surg 1997; 117:S 100-S 102. Copyright © 1997 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/97/$5.00 + 0 2314173752
Surgical exploration revealed an encapsulated, well-circumscribed mass anterior to the carotid and immediately inferior to the hyoid bone. The mass was not anatomically related to the thyroid gland and was believed to be consistent with a metastatic node. A modified dissection was performed on the left side of the neck. The visceral compartment was explored; and no thyroid tissue was found. The hyoid bone and larynx were skeletonized to the seventh tracheal ring, and again no evidence of thyroid tissue was found. Suspecting a lingual thyroid, investigators performed a bimanual examination of the tongue, and a 2-cm mass was palpated in the area of the foramen cecum linguae. 123I scan revealed low uptake (5%) at the foramen cecum linguae, consistent with lingual thyroid. The patient returned to surgery on May 19, 1993, for removal of the lingual thyroid. An oral approach was used, splitting the tongue in the midline. The lingual thyroid measured 3 x 2.7 x 1 cm. A tracheostomy was performed to ensure a safe airway. Pathologically, the neck mass had no demonstrable elements of lymphoid tissue. It did not show any capsular or vascular invasion. Histologic examination revealed psanamoma bodies and nuclear grooving, which are typical of papillary carcinoma. The diagnosis was a follicular variant of papillary carcinoma. No other evidence of carcinoma in the lymph nodes was found. The lingual thyroid exhibited areas o f oxyphilic metaplasia and focal perineural invasion. Oxypbilic cells are consistent with the Htirthle cell variety of follicular carcinoma. It contained follicles lined with cuboidal cells and neoplastic follicles lined with columnar cells with hyperchromic nuclei. The overlying epithelium showed unremarkable change, perhaps contributing to the normal appearance on examination. No vascular invasion was evident, even with immunostains. The specimen was also diagnosed as a follicular variant of papillary carcinoma. The patient exhibited mild dysarthria postoperatively, which abated with time. Thyroid replacement was temporarily withheld to facilitate a repeat 123I scan. Follow-up scintiscan revealed focal uptake posterior and lateral to the location of the lingual thyroid. The patient was admitted on June 28, 1993, for 131I ablation, performed with 150 mCi of 13~I. Since then the patient has been on levothyroxine for replacement and suppression. He is currently doing well, 2 years after his lingual thyroidectomy.
OtolaryngologyHead and Neck Surgery
WtNSLOW and WEISBERGER $101
Volume 117 Number 6
DISCUSSION The thyroid gland appears in the fetus from an invagination in the floor of the pharynx at about the fourth week of gestation. From the fourth through the seventh week of fetal life, the gland migrates caudally until it rests anterior to the larynx. As it migrates, its bilobed structure becomes more evident. It remains attached to the foramen cecum linguae by the thyroglossal duct, which later atrophies. The descent occurs before formation of the tongue and hyoid bone. The thyroglossal duct passes in close proximity to the hyoid bone, which later fuses in the midline. Consequently, the duct may pass anteriorly, posteriorly, or through the hyoid bone. The tract is lined with epithelium, which normally degenerates. Toward the end of the migration, the lateral thyroid primordia dissociate from the caudal base of the fourth pharyngeal pouch to migrate medially and join the thyroid gland. 1 This structure is called the ultimobranchial body, and it contains parafollicular, or C, cells. These are neuroectodermal and are responsible for medullary carcinoma of the thyroid. Because these cells do not join the gland until it descends, medullary carcinoma would not be expected in a purely lingual thyroid. Indeed, no such cases were found in a review of current literature. Histologically, the lingual thyroid exhibits a pattern of microfollicular adenoma. Only one fourth of the cases in one series presented with normal thyroid tissue. 2 Carcinoma of the lingual thyroid is exceedingly rare, with only 26 cases reported in the literature. In a thorough review of the literature, 21 cases were described by Fish and Moore 3 in 1963. Since that time, four cases of follicular carcinoma in a lingual thyroid have been described. 47 A case of papillary carcinoma was described by Singh 8 in 1979. One additional case of squamous cell carcinoma found overlying a lingual thyroid was described by Bukachevsky et al., 9 but the lingual thyroid, itself, displayed no evidence of carcinoma. Previous pathologic reports on carcinoma were vague, with two cases of follicular, one of papillary, one undifferentiated, and one similar to Hiirthle cell carcinoma identified. There is no known reason for this apparent follicular predominance, and it presents a contrast to thyroglossal duct carcinomas, in which the majority are papillary. The pathologic variety in 16 cases was not identified, which would make analysis difficult. All the patients, except the case described by Potdar and Desai, 6 were female. The overall incidence is female predominance by a ratio of 2:1. There have been 16 cases noted in females, and 8 in males. Our case would bring that total to 9. Two reports did not identify the sex of the patient. This incidence is similar to that found in a carcinoma of a normally located thyroid gland, in which a 2 to 3:1 female:male ratio is described. Carcinoma is no more likely to occur in a lingual thyroid than in a normally developed gland. 5 Three cases also have been described in dogs. These were treated surgically with good results. Symptoms are the same as those described for the lingual thyroid, including dysphagia, sense of foreign body, change in the quality of speech, hemorrhage, and dyspnea. Rapid growth, hemorrhage, and ulceration are noted frequently.
Otalgia was noted in one patient. It resoived after surgical removal of the carcinoma. 4 Our patient presented with only a gradually enlarging neck mass. Although local metastases are relatively common, it is unusual to have no symptoms related to the lingual thyroid. Metastases were noted in 6 patients, and were not addressed in 12 cases. Diagnosis depends on correct histopathologic evaluation. It has been suggested that a T1-201 scan may be helpful if the diagnosis is suspected clinically, because it was noted to accumulate in a case of ectopic papillary carcinoma, r° This is a potassium analog, which is concentrated in cellular lesions, such as malignancies, but not in benign nodules. All pathologic varieties of thyroid carcinoma, except medullary, can be found. Our patient exhibited a follicular variant of papillary carcinoma. It is worth noting that although the lingual tissue was purely follicular in histologic appearance, the neck metastasis exhibited some papillary elements. All neoplasms containing foci of papillary carcinoma, regardless of proportions, have identical behavior. This includes an indolent clinical course and propensity for lymphatic metastasis, especially to the neck. This differs from pure follicular carcinoma, which typically exhibits hematologic metastasis. Treatment is surgical excision, with a margin of normal tissue. 6 Lateral pharyngotomy, transhyoid incision, or midline split of the tongue are the surgical approaches of choice, which already have been described in the literature. 11 Excisional biopsy attempted in one patient resulted in the need for further surgery. 7 A tracheotomy should be anticipated because significant edema of the tongue may compromise the airway. Our patient required a temporary tracheotomy and was successfully decannulated after surgery. Thyroidectomy and neck dissection are indicated only if additional lesions or metastatic nodules are noted, similar to the approach for thyroglossal duct cyst carcinoma. Follow-up scans should be performed with radioactive iodine to ensure removal of all thyroid tissue in the foramen cecum linguae. A therapeutic dose of ~31I after surgery may be needed, depending on the completeness of the excision. Our patient had positive margins and a scan indicating residual thyroid tissue in the lingual area, and treatment with radioactive iodine was necessary. If the lingual thyroid is th e only glandular tissue in the patient, thyroid replacement therapy will be necessary. In a case of a large carcinoma, where surgery would have been mutilating, treatment with radioactive iodine was successful.
CONCLUSION T1ais article has reviewed existing information regarding ectopic thyroid tissue and carcinoma arising from such ectopia. Also we have added one case of lingual carcinoma to bring the total number of cases described in the English literature to 27.
REFERENCES 1. Welder D, Parker W. Lingual thyroid: review, case reports, and therapeutic guidelines. Ann Otol 1977;86:841--8.
OtolaryngologyHead and Neck Surgery December 1997
$102 CHEN and DONOVAN
2. Sank J. Ectopic lingual thyroid. J Pathol 1970;102:239-43. 3. Fish J, Moore R. Ectopic thyroid tissue and ectopic thyroid carcinoma. Ann Surg 1963;157:212-22. 4. Gooder R Follicular carcinoma in a lingual thyroid. J Laryngol Otol 1980;94:437-9. 5. Kamat MR, Kulkarni JN, Desai PB, Jussawalla DJ. Lingual thyroid: a review of 12 cases. Br J Surg 1979;66:537-9. 6. Potdar GG, Desai PB. Carcinoma of the lingual thyroid. Laryngoscope 1971 ;81:427-9. 7. Diaz-Arias AA, Bickel JT, Loy TS, et al. Follicular carcinoma with clear cell change arising in lingual thyroid. Oral Surg Oral Med Oral Pathol 1992;74:206-11.
8. Singh HB, Joshi HC, Chakravarty M. Carcinoma of the lingual thyroid: review and case report. J Laryngol Otol 1979;93: 839-45. 9. Bukachevsky RP, Casler JD, Oliver J, Conley J. Squamous cell carcinoma and lingual thyroid. Ear Nose Throat J 1991;70: 505-7. 10. Michigishi T, Mizukami Y, Mura T, et al. Papillary carcinoma in ectopic thyroid detected by T1-201 scintigraphy. Clin Nucl Med 1991;16:337-40. 11. Paparella MM, Shumrich DA, Gluckman JL, Meyerhoff WL. Otolaryngology. 3rd ed. Philadelphia: W B Sannders Co., 1991:2177-9.
Impaired ciliary clearance from tracheopathia osteoplastica of the upper respiratory tract AMY Y. CHEN, MD, and DONALD 1".DONOVAN, MD, Houston, Texas
Tracheopathia osteoplastica is a rare clinical and pathologic condition characterized by multiple submucosal cartilaginous and osseous nodules of various sizes lining the upper respiratory tract. Dalgaard I credits Tokitansky with the first description of this entity in 1855. Two years later, Wilks 2 presented a case and described the histologic features of tracheopathia osteoplastica. Since that time, several case reports have helped delineate the clinical features of this disease process. 36 Although many patients are asymptomatic and the characteristic pathologic changes are discovered incidentally at autopsy, 7 progressive growth of these nodules may lead to persistent hoarseness, cough, dyspnea on exertion, and occasionally significant airway obstruction. We report a case of a patient with a cough of longstanding duration (greater than 8 years) and associated pneumonia and chronic bronchitis who proved to have tracheopathia osteoplastica. Histologic changes were found that suggest a pathophysiologic mechanism, which explains the chronicity and severity of the cough seen in this disease process. To our knowledge, this is the first case report presenting evidence to explain the clinical manifestations seen in these patients.
cough with driving in an automobile for more than several minutes' duration. The cough was nonproductive and, at that time, not associated with any fever, chills, or night sweats. Her medical history was significant for hay fever as a young adult but no other significant environmental allergies or medical problems. As time progressed, she began to have coughing episodes in other settings, including in her home, at work in a hospital setting, and in public places both indoors and outdoors. She sought the advice of a pulmonary specialist and underwent pulmonary function tests with a chest roentgenogram; the results of both were interpreted to be within normal limits. She was treated with cough suppressants. Six years after the onset of her cough, she sought further evaluation by an otolaryngologist-head and neck surgeon. Physical examination findings at that time were normal.
CASE REPORT The patient was a 60-year-old woman with an 8-year history of chronic persistent cough. Initially, she correlated the
From the Department of Otorhinolaryngology and Communicative Sciences, Baylor College of Medicine. Presented at the Annual Meeting of the American Academy of Otolaryngology-Head and Neck Surgery, New Orleans, La., Sept. 17-20, 1995. Reprint requests: Donald T. Donovan, MD, Department of Otorhinolaryngology and Communicative Sciences, Baylor College of Medicine, 1 Baylor Plaza, Houston, TX 77030. Otolaryngol Head Neck Surg 1997; 117:S 102-S 104. Copyright © 1997 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/97/$5.00 + 0 23/4/73751
Fig. 1. Rigid bronchoscopic view of trachea through a Odegree Hopkins telescope demonstrating nodules on anterior and lateral surfaces of trachea.
Albright hereditary osteodystrophy with hypothyroidism, normocalcemia, and normal Gs protein activity: a family presenting with congenital osteoma curls. Am J Med Genet 1992;43:764-7. Prendiville JS, Lucky AW, Mallory SB, Mughal Z, Mimouni F, Langman CB. Osteoma curls as a presenting sign of pseudohypoparathyroidism. Pediatr Dermatol 1992;9:11-8.
OtolaryngologyHead and Neck Surgery December 1997
7. Orlow SJ, Watsky KL, Bolognia JL. Skin and bones. I. J Am Acad Dermatol 1991;25:205-22. 8. Nakamura S, Imai T, Nakayama K, Onda S. Primary osteoma curls. J Dermatol 1987;14:85-8. 9. Monroe AB, Burgdoff WHC, Sheward S, Plate-like cutaneous osteoma. J Am Acad Dermatol 1987; 16:481-4.
Lingual thyroid and neoplastic change: A review of the literature and description of a case CATHERINE P.WINSLOW, MD, and EDWARD C. WEISBERGER, MD, FACS,Denver, Colorado, and Indianapolis, Indiana
T h e embryologic development of the thyroid gland allows for anomalies in the ultimate location a!ong the course of its migration. Although ectopic remnants are common, carcinoma in such remnants is rare. Only 26 cases of lingual thyroid carcinoma were found in a review of the literature. This article reviews the literature on ectopic thyroid and associated pathology, and describes a case of carcinoma arising in the lingual thyroid.
CASE REPORT A 23-year-old white man presented on March 6, 1993, for an examination of a mass in the left side of his neck, which had been gradually increasing in size over the course of a few months. His thyroid had been enlarging since 1990, and he had taken levothyroxine for a few months, with symptomatic relief. He was otherwise asymptomatic. It was noted that another family member had hypothyroidism. Examination revealed a 5-cm mass in the region of the superior pole of the left lateral thyroid gland, overlying his jugular vein, which did not move with swallowing. The remainder of the examination was normal, including a fiberoptic examination of the pharynx and larynx. A fine-needle aspiration was taken, which revealed changes consistent with a follicular variant of papillary carcinoma. It was decided to proceed with a left thyroid lobectomy. His calcitonin level was measured to exclude the possibility of medullary carcinoma. It was found to be within normal limits. Because the mass was believed to be a thyroid malignancy, the patient was taken to surgery on March 6, 1993.
From the Department of Otolaryngology-Head and Neck Surgery, University of Colorado Health Sciences Center; Department of Otolaryngology-Head and Neck Surgery, Indiana University School of Medicine. Reprint requests: Catherine R Winslow, MD, University of Colorado Health Sciences Center, Department of Otolaryngology-Head and Neck Surgery, Box B-205, 4200 East Ninth Avenue, Denver, CO 80262. Otolaryngol Head Neck Surg 1997; 117:S 100-S 102. Copyright © 1997 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/97/$5.00 + 0 2314173752
Surgical exploration revealed an encapsulated, well-circumscribed mass anterior to the carotid and immediately inferior to the hyoid bone. The mass was not anatomically related to the thyroid gland and was believed to be consistent with a metastatic node. A modified dissection was performed on the left side of the neck. The visceral compartment was explored; and no thyroid tissue was found. The hyoid bone and larynx were skeletonized to the seventh tracheal ring, and again no evidence of thyroid tissue was found. Suspecting a lingual thyroid, investigators performed a bimanual examination of the tongue, and a 2-cm mass was palpated in the area of the foramen cecum linguae. 123I scan revealed low uptake (5%) at the foramen cecum linguae, consistent with lingual thyroid. The patient returned to surgery on May 19, 1993, for removal of the lingual thyroid. An oral approach was used, splitting the tongue in the midline. The lingual thyroid measured 3 x 2.7 x 1 cm. A tracheostomy was performed to ensure a safe airway. Pathologically, the neck mass had no demonstrable elements of lymphoid tissue. It did not show any capsular or vascular invasion. Histologic examination revealed psanamoma bodies and nuclear grooving, which are typical of papillary carcinoma. The diagnosis was a follicular variant of papillary carcinoma. No other evidence of carcinoma in the lymph nodes was found. The lingual thyroid exhibited areas o f oxyphilic metaplasia and focal perineural invasion. Oxypbilic cells are consistent with the Htirthle cell variety of follicular carcinoma. It contained follicles lined with cuboidal cells and neoplastic follicles lined with columnar cells with hyperchromic nuclei. The overlying epithelium showed unremarkable change, perhaps contributing to the normal appearance on examination. No vascular invasion was evident, even with immunostains. The specimen was also diagnosed as a follicular variant of papillary carcinoma. The patient exhibited mild dysarthria postoperatively, which abated with time. Thyroid replacement was temporarily withheld to facilitate a repeat 123I scan. Follow-up scintiscan revealed focal uptake posterior and lateral to the location of the lingual thyroid. The patient was admitted on June 28, 1993, for 131I ablation, performed with 150 mCi of 13~I. Since then the patient has been on levothyroxine for replacement and suppression. He is currently doing well, 2 years after his lingual thyroidectomy.
OtolaryngologyHead and Neck Surgery
WtNSLOW and WEISBERGER $101
Volume 117 Number 6
DISCUSSION The thyroid gland appears in the fetus from an invagination in the floor of the pharynx at about the fourth week of gestation. From the fourth through the seventh week of fetal life, the gland migrates caudally until it rests anterior to the larynx. As it migrates, its bilobed structure becomes more evident. It remains attached to the foramen cecum linguae by the thyroglossal duct, which later atrophies. The descent occurs before formation of the tongue and hyoid bone. The thyroglossal duct passes in close proximity to the hyoid bone, which later fuses in the midline. Consequently, the duct may pass anteriorly, posteriorly, or through the hyoid bone. The tract is lined with epithelium, which normally degenerates. Toward the end of the migration, the lateral thyroid primordia dissociate from the caudal base of the fourth pharyngeal pouch to migrate medially and join the thyroid gland. 1 This structure is called the ultimobranchial body, and it contains parafollicular, or C, cells. These are neuroectodermal and are responsible for medullary carcinoma of the thyroid. Because these cells do not join the gland until it descends, medullary carcinoma would not be expected in a purely lingual thyroid. Indeed, no such cases were found in a review of current literature. Histologically, the lingual thyroid exhibits a pattern of microfollicular adenoma. Only one fourth of the cases in one series presented with normal thyroid tissue. 2 Carcinoma of the lingual thyroid is exceedingly rare, with only 26 cases reported in the literature. In a thorough review of the literature, 21 cases were described by Fish and Moore 3 in 1963. Since that time, four cases of follicular carcinoma in a lingual thyroid have been described. 47 A case of papillary carcinoma was described by Singh 8 in 1979. One additional case of squamous cell carcinoma found overlying a lingual thyroid was described by Bukachevsky et al., 9 but the lingual thyroid, itself, displayed no evidence of carcinoma. Previous pathologic reports on carcinoma were vague, with two cases of follicular, one of papillary, one undifferentiated, and one similar to Hiirthle cell carcinoma identified. There is no known reason for this apparent follicular predominance, and it presents a contrast to thyroglossal duct carcinomas, in which the majority are papillary. The pathologic variety in 16 cases was not identified, which would make analysis difficult. All the patients, except the case described by Potdar and Desai, 6 were female. The overall incidence is female predominance by a ratio of 2:1. There have been 16 cases noted in females, and 8 in males. Our case would bring that total to 9. Two reports did not identify the sex of the patient. This incidence is similar to that found in a carcinoma of a normally located thyroid gland, in which a 2 to 3:1 female:male ratio is described. Carcinoma is no more likely to occur in a lingual thyroid than in a normally developed gland. 5 Three cases also have been described in dogs. These were treated surgically with good results. Symptoms are the same as those described for the lingual thyroid, including dysphagia, sense of foreign body, change in the quality of speech, hemorrhage, and dyspnea. Rapid growth, hemorrhage, and ulceration are noted frequently.
Otalgia was noted in one patient. It resoived after surgical removal of the carcinoma. 4 Our patient presented with only a gradually enlarging neck mass. Although local metastases are relatively common, it is unusual to have no symptoms related to the lingual thyroid. Metastases were noted in 6 patients, and were not addressed in 12 cases. Diagnosis depends on correct histopathologic evaluation. It has been suggested that a T1-201 scan may be helpful if the diagnosis is suspected clinically, because it was noted to accumulate in a case of ectopic papillary carcinoma, r° This is a potassium analog, which is concentrated in cellular lesions, such as malignancies, but not in benign nodules. All pathologic varieties of thyroid carcinoma, except medullary, can be found. Our patient exhibited a follicular variant of papillary carcinoma. It is worth noting that although the lingual tissue was purely follicular in histologic appearance, the neck metastasis exhibited some papillary elements. All neoplasms containing foci of papillary carcinoma, regardless of proportions, have identical behavior. This includes an indolent clinical course and propensity for lymphatic metastasis, especially to the neck. This differs from pure follicular carcinoma, which typically exhibits hematologic metastasis. Treatment is surgical excision, with a margin of normal tissue. 6 Lateral pharyngotomy, transhyoid incision, or midline split of the tongue are the surgical approaches of choice, which already have been described in the literature. 11 Excisional biopsy attempted in one patient resulted in the need for further surgery. 7 A tracheotomy should be anticipated because significant edema of the tongue may compromise the airway. Our patient required a temporary tracheotomy and was successfully decannulated after surgery. Thyroidectomy and neck dissection are indicated only if additional lesions or metastatic nodules are noted, similar to the approach for thyroglossal duct cyst carcinoma. Follow-up scans should be performed with radioactive iodine to ensure removal of all thyroid tissue in the foramen cecum linguae. A therapeutic dose of ~31I after surgery may be needed, depending on the completeness of the excision. Our patient had positive margins and a scan indicating residual thyroid tissue in the lingual area, and treatment with radioactive iodine was necessary. If the lingual thyroid is th e only glandular tissue in the patient, thyroid replacement therapy will be necessary. In a case of a large carcinoma, where surgery would have been mutilating, treatment with radioactive iodine was successful.
CONCLUSION T1ais article has reviewed existing information regarding ectopic thyroid tissue and carcinoma arising from such ectopia. Also we have added one case of lingual carcinoma to bring the total number of cases described in the English literature to 27.
REFERENCES 1. Welder D, Parker W. Lingual thyroid: review, case reports, and therapeutic guidelines. Ann Otol 1977;86:841--8.
OtolaryngologyHead and Neck Surgery December 1997
$102 CHEN and DONOVAN
2. Sank J. Ectopic lingual thyroid. J Pathol 1970;102:239-43. 3. Fish J, Moore R. Ectopic thyroid tissue and ectopic thyroid carcinoma. Ann Surg 1963;157:212-22. 4. Gooder R Follicular carcinoma in a lingual thyroid. J Laryngol Otol 1980;94:437-9. 5. Kamat MR, Kulkarni JN, Desai PB, Jussawalla DJ. Lingual thyroid: a review of 12 cases. Br J Surg 1979;66:537-9. 6. Potdar GG, Desai PB. Carcinoma of the lingual thyroid. Laryngoscope 1971 ;81:427-9. 7. Diaz-Arias AA, Bickel JT, Loy TS, et al. Follicular carcinoma with clear cell change arising in lingual thyroid. Oral Surg Oral Med Oral Pathol 1992;74:206-11.
8. Singh HB, Joshi HC, Chakravarty M. Carcinoma of the lingual thyroid: review and case report. J Laryngol Otol 1979;93: 839-45. 9. Bukachevsky RP, Casler JD, Oliver J, Conley J. Squamous cell carcinoma and lingual thyroid. Ear Nose Throat J 1991;70: 505-7. 10. Michigishi T, Mizukami Y, Mura T, et al. Papillary carcinoma in ectopic thyroid detected by T1-201 scintigraphy. Clin Nucl Med 1991;16:337-40. 11. Paparella MM, Shumrich DA, Gluckman JL, Meyerhoff WL. Otolaryngology. 3rd ed. Philadelphia: W B Sannders Co., 1991:2177-9.
Impaired ciliary clearance from tracheopathia osteoplastica of the upper respiratory tract AMY Y. CHEN, MD, and DONALD 1".DONOVAN, MD, Houston, Texas
Tracheopathia osteoplastica is a rare clinical and pathologic condition characterized by multiple submucosal cartilaginous and osseous nodules of various sizes lining the upper respiratory tract. Dalgaard I credits Tokitansky with the first description of this entity in 1855. Two years later, Wilks 2 presented a case and described the histologic features of tracheopathia osteoplastica. Since that time, several case reports have helped delineate the clinical features of this disease process. 36 Although many patients are asymptomatic and the characteristic pathologic changes are discovered incidentally at autopsy, 7 progressive growth of these nodules may lead to persistent hoarseness, cough, dyspnea on exertion, and occasionally significant airway obstruction. We report a case of a patient with a cough of longstanding duration (greater than 8 years) and associated pneumonia and chronic bronchitis who proved to have tracheopathia osteoplastica. Histologic changes were found that suggest a pathophysiologic mechanism, which explains the chronicity and severity of the cough seen in this disease process. To our knowledge, this is the first case report presenting evidence to explain the clinical manifestations seen in these patients.
cough with driving in an automobile for more than several minutes' duration. The cough was nonproductive and, at that time, not associated with any fever, chills, or night sweats. Her medical history was significant for hay fever as a young adult but no other significant environmental allergies or medical problems. As time progressed, she began to have coughing episodes in other settings, including in her home, at work in a hospital setting, and in public places both indoors and outdoors. She sought the advice of a pulmonary specialist and underwent pulmonary function tests with a chest roentgenogram; the results of both were interpreted to be within normal limits. She was treated with cough suppressants. Six years after the onset of her cough, she sought further evaluation by an otolaryngologist-head and neck surgeon. Physical examination findings at that time were normal.
CASE REPORT The patient was a 60-year-old woman with an 8-year history of chronic persistent cough. Initially, she correlated the
From the Department of Otorhinolaryngology and Communicative Sciences, Baylor College of Medicine. Presented at the Annual Meeting of the American Academy of Otolaryngology-Head and Neck Surgery, New Orleans, La., Sept. 17-20, 1995. Reprint requests: Donald T. Donovan, MD, Department of Otorhinolaryngology and Communicative Sciences, Baylor College of Medicine, 1 Baylor Plaza, Houston, TX 77030. Otolaryngol Head Neck Surg 1997; 117:S 102-S 104. Copyright © 1997 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/97/$5.00 + 0 23/4/73751
Fig. 1. Rigid bronchoscopic view of trachea through a Odegree Hopkins telescope demonstrating nodules on anterior and lateral surfaces of trachea.