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Dual Ectopic Thyroid Gland Sonography and Scintigraphy of Lingual and Sublingual Thyroid Vinko Markovi(, MD, PhD,* Gordana Glavina, MD,Þ Davor Eterovi(, PhD,* Ante Punda, MD, PhD,* and Dubravka Brdar, MD* Abstract: Dual ectopic lingual and sublingual thyroid gland is an extraordinarily rare condition. We present 1 patient with subclinical hypothyroidism. The clinical examination revealed that the thyroid gland was not palpable in its usual cervical location, whereas ultrasonography confirmed an empty thyroid bed without any ectopic thyroid tissue in the rest of the neck. The final diagnosis of dual ectopic lingual and sublingual thyroid was established by ultrasound examination through the mouth floor and confirmed by scintigraphy and CT thereafter. Key Words: sonography, scintigraphy, dual ectopic, lingual, sublingual, thyroid gland (Clin Nucl Med 2014;39: 556Y558)

Received for publication May 16, 2013; revision accepted January 27, 2014. From the *Department of Nuclear Medicine, and †Clinical Institute of Radiology, University Hospital Split, University of Split Medical Faculty, Split, Croatia. Conflicts of interest and sources of funding: none declared. Reprints: Vinko Markovi(, MD, PhD, Department of Nuclear Medicine, University Hospital Split, University of Split Medical Faculty, Split, Spincˇi(eva 1, 21 000 Split, Croatia. E-mail: [email protected]. Copyright * 2014 by Lippincott Williams & Wilkins ISSN: 0363-9762/14/3906Y0556

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REFERENCES 1. Chawla M, Kumar R, Malhotra A. Dual ectopic thyroid: case series and review of the literature. Clin Nucl Med. 2007;32:1Y5. 2. Sood A, Sood V, Sharma DR, et al. Thyroid scintigraphy in detecting dual ectopic thyroid: a review. Eur J Nucl Med Mol Imaging. 2008;35:843Y846. 3. Harisankar CN, Preethi GR, George M. Hybrid SPECT/CT evaluation of dual ectopia of thyroid in the absence of orthotopic thyroid gland. Clin Nucl Med. 2012;37:602Y603. 4. Yoon JS, Won KC, Cho IH, et al. Clinical characteristics of ectopic thyroid in Korea. Thyroid. 2007;17:1117Y1121. 5. Jain A, Pathak S. Rare developmental abnormalities of thyroid gland, especially multiple ectopia: a review and our experience. Indian J Nucl Med. 2010;25:143Y146. 6. Marinovic D, Garel C, Czernichow P, et al. Ultrasonographic assessment of the ectopic thyroid tissue in children with congenital hypothyroidism. Pediatr Radiol. 2004;34:109Y113. 7. Fujioka K, Fujioka A, Ban Y, et al. A case of dual ectopic thyroid accompanied by positive antithyroid antibodies. J Med Ultrasonics. 2008;35:201Y205. 8. Kumar Choudhury B, Kaimal Saikia U, Sarma D, et al. Dual ectopic thyroid with normally located thyroid: a case report. J Thyroid Res. 2011. doi:10.4061/2011/159703. 9. McCoul ED, de Vries EJ. Concurrent lingual thyroid and undescended thyroglossal duct thyroid without orthotopic thyroid gland. Laryngoscope. 2009;119:1937Y1940. 10. Ohnishi H, Sato H, Noda H, et al. Color Doppler ultrasonography: diagnosis of ectopic thyroid gland in patients with congenital hypothyroidism caused by thyroid dysgenesis. J Clin Endocrinol Metab. 2003;88:5145Y5149. 11. Choi JH, Kim DH, Kang HJ, et al. Dual ectopic thyroid: a case report. Korean J Otolaryngol-Head Neck Surg. 2001;44:897Y900. 12. Kim DY, Yang SH, Song SH, et al. Dual ectopic thyroid. Korean J Otolaryngol-Head Neck Surg. 2001;44:1120Y1122. 13. Chang YW, Lee DH, Hong YH, et al. Congenital hypothyroidism: analysis of discordant US and scintigraphic findings. Radiology. 2011;258:872Y879. 14. Bayat MR, Vawda F, Campbell H. Dual ectopic thyroid. Clin Radiol. 2005; 60:821Y825.

Clinical Nuclear Medicine

& Volume 39, Number 6, June 2014

Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.

Clinical Nuclear Medicine

& Volume 39, Number 6, June 2014

Lingual and Sublingual Thyroid

FIGURE 1. An asymptomatic 47-year-old woman was referred to our outpatient clinic for further investigation after her laboratory data indicated subclinical hypothyroidism. Neck examination revealed neither palpable thyroid gland in its usual pretracheal position nor any other palpable cervical mass. Ultrasonography revealed empty thyroid bed without any ectopic thyroid tissue in the rest of the neck. However, the posterior coronal (A) and midline sagittal (B) sonograms from the submental region through the mouth floor revealed 2 spherically hypoechogenic masses. The larger one was at the base of the tongue and measured 26  25  16 mm (large arrow), and the smaller one, situated inferior and anterior, at the sublingual region measured 16  15  9 mm (small arrow). To confirm that these hypoechogenic structures belonged to the dual ectopic thyroid gland, the patient was referred to 99m TcO4 scintigraphy.

FIGURE 2. 99mTcO4 scintigraphy, anterior (A) and left lateral (B) views, showed 2 focal areas of abnormal radiotracer uptake in the lingual (large arrow) and sublingual (small arrow) regions and absence of radiotracer activity in normal cervical thyroid region. On the anterior scintigram, there is a partial overlap of activity of both ectopic glands. Thyroid scintigraphy remains the keystone diagnostic procedure for detection or confirmation of ultrasonography-detected ectopic thyroid tissue.1,2 Besides establishing the absence of the thyroid in its normal cervical position, it can detect lingual, sublingual, perihyoid, and other neck locations of ectopic thyroid glands, as well as thoracic or, in very rare cases, abdominal ectopic thyroid tissue. Scintigraphy differentiates these ectopic cervical thyroid tissues from other causes of neck swelling such as thyroglossal duct cyst, enlarged lymph node, lipoma, epidermoid cyst, malignant conditions, and vascular malformation, such as lingual thyroid from hypertrophic lingual tonsil, vallecular cyst, and mucous retention cyst. Hybrid imaging with SPECT/CT provides more convenient functional and anatomical details of ectopic thyroid.3 * 2014 Lippincott Williams & Wilkins

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FIGURE 3. Axial CT scan (A) showed 2 hyperdense soft tissue masses in the midline, larger mass posterior (large arrow), and smaller mass anterior (small arrow), with strong enhancement on contrast CT scan (B), as in normal thyroid gland. Sagittal contrast enhancement CT image (C) showed 2 strong contrast enhancement masses that corresponded to the lingual (large arrow) and sublingual (small arrow) ectopic thyroid glands. Dual ectopic thyroid gland is an exceptionally rare condition with only 49 cases reported to date.1Y14 Among them, lingual and sublingual dual ectopic thyroid was reported in only 2 studies,4,13 one with scintigraphic image only4 and the other without any images.13 Thyroxine was prescribed and at control examination; 3 months later, the serum thyroid-stimulating hormone level was normal.

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* 2014 Lippincott Williams & Wilkins

Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.

Dual ectopic thyroid gland: sonography and scintigraphy of lingual and sublingual thyroid.

Dual ectopic lingual and sublingual thyroid gland is an extraordinarily rare condition. We present 1 patient with subclinical hypothyroidism. The clin...
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