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Ectopic lingual thyroid with a multinodular goiter
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Ectopic lingual thyroid with a multinodular goiter Eugene Poh Hze-Khoong, BDS, MDS,a Liqun Xu, DDS, MD, PhD,b Shukun Shen, DDS, MD, PhD,b Xuelai Yin, DDS, MD,b Lizhen Wang, DDS, PhD,b and Chenping Zhang, DDS, MD, PhD,b Singapore, and Shanghai, China
From the Oral and Maxillofacial Department,a Khoo Teck Puat Hospital, Singapore; and the Department of Oral and Maxillofacial Surgery,b School of Stomatology, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, PR China
CASE REPORT A 60-YEAR OLD CHINESE MAN presented complaining of dysphagia and fullness in his throat. Clinically, a large mass was palpable at the root of his tongue and computed tomography showed its extension to completely occlude the oropharynx (Figs 1 and 2). Although his thyroid gland was notably absent, his thyroid function tests were normal and technetium Tc-99m scans revealed a distinctive uptake in the mass that was subsequently excised via a transcervical, trans-hyoid approach (Figs 3 and 4). Histologic analysis confirmed the diagnosis of a multinodular goiter and he was immediately started on a course of thyroxine supplements. Follow-up visits have been largely unremarkable with significant amelioration of his complaints. DISCUSSION Ectopic thyroids are aberrantly located thyroid tissue anywhere along the embryologic thyroglossal Accepted for publication August 16, 2011. Reprint requests: Liqun Xu, DDS, MD, PhD, Department of Oral and Maxillofacial Surgery, School of Stomatology, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, Zhizaoju Road 639#, Shanghai 200011, PR China. E-mail: [email protected]. Surgery 2013;153:294-6. 0039-6060/$ - see front matter Ó 2013 Mosby, Inc. All rights reserved. doi:10.1016/j.surg.2011.08.014
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Fig 1. Sagittal computed tomograph of the heterogenous, well-delineated mass completely occluding the oropharynx. (Color version of figure is available online.)
tract between the foramen cecum of the tongue to its orthotopic location anterior to the thyroid cartilage. Its pathogenesis is still unclear although Van der Gaag et al1 postulated that maternal antithyroid immunoglobulins inhibited thyroid-stimulating
Surgery Volume 153, Number 2
Hze-Khoong et al 295
Fig 2. Axial view showing the lateral extension of the mass with varying amounts of calcification. Fig 4. Delivery of the large multi-nodular goiter to reveal the posterior wall of the pharynx. (Color version of figure is available online.)
Fig 3. Perioperative picture of the transected hyoid bone to access the mass. (Color version of figure is available online.)
hormone production and impaired thyroid growth and migration. The lingual thyroid, first described by Hickman in 1869,2 is the most common location for thyroid ectopias with a reported prevalence of 1:100,000 to 1:300,000. They have a 4:1 female predilection and are commonly discovered during
childhood, adolescence, or menopause, when physiologic demands for the anabolic thyroid hormones causes surges in thyroid-stimulating hormone levels.3 Presenting complaints include dyspnea, dysphagia, dysphonia, stridor, fullness of throat, and obstructive sleep apnea. Clinical examinations may reveal a mass behind the circumvallate papillae when the tongue is retracted anteriorly, and magnetic resonance imaging, ultrasonography, computed tomography, radionuclide iodine-131, and technetium Tc-99m scans are invaluable in differentiating lingual thyroids from angiomas, adenomas, branchial cysts, fibromas, thyroglossal duct cysts, lymphangiomas, lipomas, and minor salivary gland tumors, as well as to identify the presence of other thyroid tissue.4 Thomas et al5 reported that lingual thyroids were the only functioning thyroid tissue in 70% of the cases with a high incidence of hypothyroidism after its removal, mandating the need for pre- and postoperative thyroid function tests. Because they can function as a normal exocrine gland, they are susceptible to similar thyroid pathologies, such as glandular hyperplasia, malignancies, calcifications, ossifications, inflammation, and multinodular goiters. In such instances, treatment ranges from watchful waiting for asymptomatic cases to hormonal substitution to suppress thyroid-stimulating hormone–induced growths, simple curative biopsies for small lesions, radiofrequency or radioiodine ablation, and transoral
296 Hze-Khoong et al
or transcervical operative excisions. Transplantation of the ectopic thyroid may be unnecessary, because substitutive thyroid hormones can easily maintain a euthyroid state. In the only other documented incidence of a multinodular goiter in a lingual thyroid, a midline mandibular-split technique was performed to excise the mass.6 Although such transoral approaches may seem less invasive, they should be reserved for smaller lesions owing to their reported propensity for diffuse and uncontrollable hemorrhage in a limited surgical field.3 REFERENCES 1. Van der Gaag RD, Drexhage HA, Dussault JH. Role of maternal immunoglobulins blocking TSH-induced thyroid growth
Surgery February 2013
2.
3.
4.
5.
6.
in sporadic forms of congenital hypothyroidism. Lancet 1985;1:245-50. Hickman W. Congenital tumour of the base of the tongue passing down the epiglottis on the larynx and causing death by suffocation sixteen hours after death (sic.). Trans Path Soc Lond 1869;20:160. Toso A, Colombani F, Averono G, Aluffi P, Pia F. Lingual thyroid causing dysphagia and dyspnoea. Case reports and review of the literature. Acta Otorhinolaryngol Ital 2009;29:213-7. Kalan A, Tariq M. Lingual thyroid gland: clinical evaluation and comprehensive management. Ear Nose Throat J 1999; 78:340-9. Thomas G, Hoilat R, Daniels JS, Kalagie W. Ectopic lingual thyroid: a case report. Int J Oral Maxillofac Surg 2003;32: 219-21. Zackaria M, Chisholm EJ, Tolley NS, Rice A, Chhatwani A. Multinodular goitre in lingual thyroid: case report. J Laryngol Otolaryngol 2010;124:349-51.
Ectopic lingual thyroid with a multinodular goiter Eugene Poh Hze-Khoong, BDS, MDS,a Liqun Xu, DDS, MD, PhD,b Shukun Shen, DDS, MD, PhD,b Xuelai Yin, DDS, MD,b Lizhen Wang, DDS, PhD,b and Chenping Zhang, DDS, MD, PhD,b Singapore, and Shanghai, China
From the Oral and Maxillofacial Department,a Khoo Teck Puat Hospital, Singapore; and the Department of Oral and Maxillofacial Surgery,b School of Stomatology, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, PR China
CASE REPORT A 60-YEAR OLD CHINESE MAN presented complaining of dysphagia and fullness in his throat. Clinically, a large mass was palpable at the root of his tongue and computed tomography showed its extension to completely occlude the oropharynx (Figs 1 and 2). Although his thyroid gland was notably absent, his thyroid function tests were normal and technetium Tc-99m scans revealed a distinctive uptake in the mass that was subsequently excised via a transcervical, trans-hyoid approach (Figs 3 and 4). Histologic analysis confirmed the diagnosis of a multinodular goiter and he was immediately started on a course of thyroxine supplements. Follow-up visits have been largely unremarkable with significant amelioration of his complaints. DISCUSSION Ectopic thyroids are aberrantly located thyroid tissue anywhere along the embryologic thyroglossal Accepted for publication August 16, 2011. Reprint requests: Liqun Xu, DDS, MD, PhD, Department of Oral and Maxillofacial Surgery, School of Stomatology, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, Zhizaoju Road 639#, Shanghai 200011, PR China. E-mail: [email protected]. Surgery 2013;153:294-6. 0039-6060/$ - see front matter Ó 2013 Mosby, Inc. All rights reserved. doi:10.1016/j.surg.2011.08.014
294 SURGERY
Fig 1. Sagittal computed tomograph of the heterogenous, well-delineated mass completely occluding the oropharynx. (Color version of figure is available online.)
tract between the foramen cecum of the tongue to its orthotopic location anterior to the thyroid cartilage. Its pathogenesis is still unclear although Van der Gaag et al1 postulated that maternal antithyroid immunoglobulins inhibited thyroid-stimulating
Surgery Volume 153, Number 2
Hze-Khoong et al 295
Fig 2. Axial view showing the lateral extension of the mass with varying amounts of calcification. Fig 4. Delivery of the large multi-nodular goiter to reveal the posterior wall of the pharynx. (Color version of figure is available online.)
Fig 3. Perioperative picture of the transected hyoid bone to access the mass. (Color version of figure is available online.)
hormone production and impaired thyroid growth and migration. The lingual thyroid, first described by Hickman in 1869,2 is the most common location for thyroid ectopias with a reported prevalence of 1:100,000 to 1:300,000. They have a 4:1 female predilection and are commonly discovered during
childhood, adolescence, or menopause, when physiologic demands for the anabolic thyroid hormones causes surges in thyroid-stimulating hormone levels.3 Presenting complaints include dyspnea, dysphagia, dysphonia, stridor, fullness of throat, and obstructive sleep apnea. Clinical examinations may reveal a mass behind the circumvallate papillae when the tongue is retracted anteriorly, and magnetic resonance imaging, ultrasonography, computed tomography, radionuclide iodine-131, and technetium Tc-99m scans are invaluable in differentiating lingual thyroids from angiomas, adenomas, branchial cysts, fibromas, thyroglossal duct cysts, lymphangiomas, lipomas, and minor salivary gland tumors, as well as to identify the presence of other thyroid tissue.4 Thomas et al5 reported that lingual thyroids were the only functioning thyroid tissue in 70% of the cases with a high incidence of hypothyroidism after its removal, mandating the need for pre- and postoperative thyroid function tests. Because they can function as a normal exocrine gland, they are susceptible to similar thyroid pathologies, such as glandular hyperplasia, malignancies, calcifications, ossifications, inflammation, and multinodular goiters. In such instances, treatment ranges from watchful waiting for asymptomatic cases to hormonal substitution to suppress thyroid-stimulating hormone–induced growths, simple curative biopsies for small lesions, radiofrequency or radioiodine ablation, and transoral
296 Hze-Khoong et al
or transcervical operative excisions. Transplantation of the ectopic thyroid may be unnecessary, because substitutive thyroid hormones can easily maintain a euthyroid state. In the only other documented incidence of a multinodular goiter in a lingual thyroid, a midline mandibular-split technique was performed to excise the mass.6 Although such transoral approaches may seem less invasive, they should be reserved for smaller lesions owing to their reported propensity for diffuse and uncontrollable hemorrhage in a limited surgical field.3 REFERENCES 1. Van der Gaag RD, Drexhage HA, Dussault JH. Role of maternal immunoglobulins blocking TSH-induced thyroid growth
Surgery February 2013
2.
3.
4.
5.
6.
in sporadic forms of congenital hypothyroidism. Lancet 1985;1:245-50. Hickman W. Congenital tumour of the base of the tongue passing down the epiglottis on the larynx and causing death by suffocation sixteen hours after death (sic.). Trans Path Soc Lond 1869;20:160. Toso A, Colombani F, Averono G, Aluffi P, Pia F. Lingual thyroid causing dysphagia and dyspnoea. Case reports and review of the literature. Acta Otorhinolaryngol Ital 2009;29:213-7. Kalan A, Tariq M. Lingual thyroid gland: clinical evaluation and comprehensive management. Ear Nose Throat J 1999; 78:340-9. Thomas G, Hoilat R, Daniels JS, Kalagie W. Ectopic lingual thyroid: a case report. Int J Oral Maxillofac Surg 2003;32: 219-21. Zackaria M, Chisholm EJ, Tolley NS, Rice A, Chhatwani A. Multinodular goitre in lingual thyroid: case report. J Laryngol Otolaryngol 2010;124:349-51.