Bongiovanni, Massimo

Gourmaud, Jolanta

Pusztaszeri, Marc

Triponez, Frederic

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Ectopic Thyroid Tissue in the Adrenal GlandJolanta Gourmaud & Massimo Bongiovanni &Frederic Triponez & Marc PusztaszeriPublished online: 27 June 2013# Springer Science+Business Media New York 2013Case HistoryA 51-year-old female presented with a left adrenal mass. Thelesion was an incidental finding on an abdominal CT scanperformed for chronic megacolon. The mass was discoveredin 1996 and slowly enlarged in size during the follow-up(2.5 cm in 1996, 4.2 cm in 2009). Radiologically, the masshad tissular density with apical hypodensity and some calci-fications. The endocrine work-up did not show abnormaladrenal hormone levels. Left endoscopic adrenalectomywas performed.What is Your Diagnosis?Pathological Diagnosis: Ectopic Thyroid Tissue in the AdrenalGland with a Cystic Component and Focal Nuclear AtypiaGrossly, the lesion measured 3.3 cm in the greatest diameter. Itwas cystic and multilocular (Fig.1a), filled with yellow jellycontent. Microscopically, there were cystic spaces lined by epi-thelial cells and septa containing rare follicular structuresintermingled with the adrenal cortex. Some dystrophic calcifica-tions, cholesterol crystals, and macrophages were present. Theepithelial lining was flat or cuboidal with polymorphic andsometimes overlapping nuclei. The cystic walls were generallythin, composed of fibrous tissue, but sometimes thicker withsome smooth muscle cells. Foci of the adrenal cortex and follic-ular structures were found admixed within the fibrous tissue.Those follicles consisted of flat cells centered by eosinophilicamorphous material (colloid-like), consistent with thyroid folli-cles. Some nuclear features suspicious for papillary thyroidcarcinoma (enlarged and overlapping nuclei, clear chromatin,some nuclear grooves, and rare nuclear pseudoinclusions) wereobserved in some follicles (Fig. 1b–d) and in some of the cyst-lining cells. No psammoma bodies were found.Immunohistochemistry was performed. The cyst-liningcells and the follicular cells were positive for keratin, thyro-globulin, and thyroid transcription factor-1 (TTF-1) (Fig. 1e).Galectin-3 and HBME-1 were focally positive in the cyst-lining cells, but were negative in the follicles (Fig. 1f).CommentThe most important differential diagnosis to consider in thiscase was a cystic metastasis from a papillary thyroid carci-noma (PTC), especially in the presence of some nuclearatypia and partial expression of Galectin-3 and HBME-1,which are considered as general markers of thyroid malig-nancy [1]. For that reason, an additional clinical work-upwas performed. There were no other lesions on multipleabdominal CT scans. Thyroid ultrasonography showed afew nodules with benign characteristics, and an I-123 wholebody scan coupled with thoracic and abdominal CT scanshowed only physiological uptake in the thyroid. Therewas no clinical evidence of thyroid malignancy 2 years afterthe adrenalectomy. Moreover, the adrenal lesion was alsoknown to be present for at least 14 years. This would be veryunusual for a metastatic PTC, although extremely slowlygrowing metastases from PTC have been reported, with in-tervals between detection of the primary tumor and metasta-ses as long as 45 years [2]. Thereby, we assumed that ametastasis from a PTC or from a struma ovarii could beJ. Gourmaud :M. Pusztaszeri (*)Department of Clinical Pathology, Geneva UniversityHospital, 1 rue Michel-Servet, 1211 Genève 14, Switzerlande-mail: marc.pusztaszeri@hcuge.chM. BongiovanniInstitute of Pathology, Locarno, SwitzerlandF. TriponezDepartment of Thoracic and Endocrine Surgery,Geneva University Hospital, Geneva, SwitzerlandEndocr Pathol (2014) 25:353–355DOI 10.1007/s12022-013-9255-xreasonably excluded, and we concluded that the adrenalmass was consistent with ectopic thyroid tissue with atypia.Although the nuclear atypia was most likely reactive innature, we also assumed that a papillary carcinoma arisingfrom ectopic thyroid tissue in the adrenal could not becompletely ruled out.Ectopic thyroid tissue in the adrenal gland is an extremelyrare finding. To the best of our knowledge, only nine caseshave been reported in the English literature [3–9], mainly byJapanese authors [3–7]. Patients were almost exclusivelywomen (male/female, 1/8), and their age ranged from 38 to67 years. Most of the lesions were cystic (eight out of nine)and incidentally discovered on imaging. Their size rangedfrom 0.8 to 5 cm. Two lesions were associated with secretingadrenal adenoma [7, 8]. In one case, ectopic thyroid wasassociated with a papillary microcarcinoma of the thyroid [8].Etiology of ectopic thyroid tissue in the adrenal gland isunknown and is difficult to explain based on the embryologicaldevelopment of the thyroid gland. The thyroid develops from theendoderm and descends from the base of the tongue to theanterior trachea. Therefore, ectopic thyroid tissue is common inthe cervical and mediastinal region [9]. It is also possible to findectopic thyroid tissue below the diaphragm in several organs ofendodermic origin, including the gallbladder [10], ovary [11],small intestine, mesentery [12], pancreas [13], duodenum [14],and vagina [15]. On the other hand, the adrenal gland is com-posed of a medulla which is ectodermic in origin, and the cortexis developed from themesoderm.Other possible explanations forthis pathology would be metaplasia, teratoma, or tumor originat-ing from adrenal stem cells, akin to adrenal myelolipoma [16].The most important issue in assessing a case of thyroidtissue in an ectopic location is the exclusion of a metastasisfrom a thyroid carcinoma. The presence of atypical follicularcells should also raise the possibility of a papillary carcinomaarising in ectopic thyroid tissue. Indeed, an ectopic thyroidtissue can be subject to the same pathological processes asnormal eutopic thyroid tissue such as inflammation, hyperpla-sia, and tumorigenesis [17]. A careful history and histologicalexamination is necessary. Clinically, thyroid lesions and othermetastases have to be excluded. Thyroglobulin and TTF-1immunostaining confirm the thyroid differentiation, whileGalectin-3 and HBME-1, although not specific, may supportmalignancy. Testing for BRAF V600E mutation may also behelpful in the differential diagnosis [9, 18]. However, althoughthe mutation is highly specific for PTC, its absence does notrule out malignancy because of low sensitivity, especially insome variants of PTC such as the follicular variant. Themutation can also be screened for by immunohistochemistry,Fig. 1 a Macroscopically, theadrenal gland is centered by amultilocular cystic lesionwith jellycontent (blue square=1 cm2). bHistologically, the cystic lining isflat to cuboidal, and the septa arefibrous containing thyroid folliclesand adrenal cortical cells with clearcytoplasm (original magnification×200). c Higher magnification offollicles intermingled with theadrenal cortex (originalmagnification ×400). d Somefollicles show nuclear atypiasuspicious for papillary thyroidcarcinoma: enlarged nuclei, clearchromatin, and nuclear grooves(original magnification ×400). eTTF-1 immunostain showingnuclear positivity in follicles andcystic lining cells (originalmagnification ×400). f HBME-1immunostain is positive in thecystic epithelium but negative inthe follicles (originalmagnification×400)354 Endocr Pathol (2014) 25:353–355with a very high accuracy [19]. Other potential sources ofthyroid tissue in an ectopic location include a metastasis froma struma ovarii in females and a metastasis from the recentlydescribed and uncommon thyroid-like follicular carcinoma ofthe kidney. However, the latter is typically negative for TTF-1,HBME-1, and Galectin-3 [20].References1. Prasad ML, Pellegata NS, Huang Y, Nagaraja HN, de la ChapelleA, Kloos RT. Galectin-3, fibronectin-1, CITED-1, HBME1 andcytokeratin-19 immunohistochemistry is useful for the differentialdiagnosis of thyroid tumors. Mod Pathol 18:48–57, 20052. Kikuchi R, Isowa N, Tokuyasu H, Fukuda K, Kawasaki Y. Lungmetastases from thyroid cancer found 45 years after a righthemithyroidectomy. Chirurgia 19:439–441, 2006.3. Tsujimura A, Takaha M, Takayama H, Sugao H, Takeda M, KurataA. Ectopic thyroid tissue in a cystic adrenal mass. Br J Urol77:605–606, 1996.4. Shiraishi T, Imai H, Fukutome K, Watanabe M, Yatani R. Ectopicthyroid in the adrenal gland. Hum Pathol 30:105–108, 1999.5. Shuno Y, Kobayashi T, Morita K, Shimizu S, Nishio Y, Ito A,Kobayashi K, KawaharaM, TeruyaM. Ectopic thyroid in the adrenalgland presenting as cystic lesion. Surgery 139:580–582, 2006.6. Takao H, Doi I, Watanabe T. Ectopic thyroid in the adrenal gland:computed tomography findings. J Comput Assist Tomogr 30:221–222, 2006.7. Hagiuda J, Kuroda I, Tsukamoto T, Ueno M, Yokota C, Hirose T,Deguchi N. Ectopic thyroid in an adrenal mass: a case report. BMCUrol 6:18, 2006.8. Bohinc BN, Parker JC, Hope WW, Kotwall C, Turner J, Cheng W,Lloyd RV. Micropapillary thyroid carcinoma and concomitant ec-topic thyroid tissue in the adrenal gland: metastasis or metaplasia?Thyroid 21:1033–1038, 2011.9. Romero-Rojas A, Bella-Cueto MR, Meza-Cabrera IA, Cabezuelo-Hernández A, García-Rojo D, Vargas-Uricoechea H, Cameselle-Teijeiro J. Ectopic Thyroid Tissue in the Adrenal Gland: Report of 2Cases with Pathogenetic Implications. Thyroid 2013. doi:10.1089/thy.2013.0063.10. Cassol CA, Noria D, Asa SL. Ectopic thyroid tissue within the gallbladder: case report and brief review of the literature. Endocr Pathol21:263–265, 2010.11. Macleod DH. Struma Ovarii (Thyro-Dermoid). A note on theteratomatous origin. Proc R Soc Med 25:1386–1391, 1932.12. Gungor B, Kebat T, Ozaslan C, Akilli S. Intra-abdominal ectopicthyroid presenting with hyperthyroidism: report of a case. SurgToday 32:148–150, 2002.13. Eyuboqlu E, Kapan M, Ipek T, Ersan Y, Oz F. Ectopic thyroid in theabdomen: report of a case. Surg Today 29:472–474, 1999.14. Takahashi T, Ishikura H, Kato H, Tanabe T, Yoshiki T. Ectopicthyroid follicles in the submucosa of the duodenum. Virchows ArchA Pathol Anat Histopathol 418:547–550, 1991.15. Kurman RJ, Prabha AC. Thyroid and parathyroid glands in thevaginal wall: report of a case. Am J Clin Pathol 59:503–507, 1973.16. Bishop E, Eble JN, Cheng L,WangM, Chase DR, Orazi A, O'MalleyDP. Adrenal myelolipomas show nonrandom X-chromosome inacti-vation in hematopoietic elements and fat: support for a clonal originof myelolipomas. Am J Surg Pathol 30:838–843, 2006.17. Klubo-Gwiezdzinska J, Manes RP, Chia SH, Burman KD,Stathatos NA, Deeb ZE, Wartofsky L. Clinical review: Ectopiccervical thyroid carcinoma–review of the literature with illustrativecase series. J Clin Endocrinol Metab 96:2684–2691, 2011.18. Rossi ED, Martini M, Straccia P, Larocca LM, Fadda G. Detectionof ectopic thyroid remnants: a serious diagnostic dilemma. Whenmolecular biology and immunohistochemistry can solve the prob-lem. Pathol Res Pract 209:59–61, 2013.19. Capper D, Preusser M, Habel A, Sahm F, Ackermann U, SchindlerG, Pusch S, Mechtersheimer G, Zentgraf H, von Deimling A.Assessment of BRAF V600E mutation status by immunohisto-chemistry with a mutation-specific monoclonal antibody. ActaNeuropathol 122:11–19, 2011.20. Alessandrini L, Fassan M, Gardiman MP, Guttilla A, Zattoni F,Galletti TP, Zattoni F. Thyroid-like follicular carcinoma of thekidney: report of two cases with detailed immunohistochemicalprofile and literature review. Virchows Arch 461:345–350, 2012.Endocr Pathol (2014) 25:353–355 355

Ectopic Thyroid Tissue in the Adrenal Gland

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Ectopic Thyroid Tissue in the Adrenal Gland

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