A case of normocalcemic primary hyperparathyroidism presenting with a mass in the oral cavity and accompanying incidental papillary thyroid carcinoma

Year 2022, Volume: 47 Issue: 4, 1764 - 1767, 28.12.2022

Emek Topuz Dilek Tüzün Ümit Nur Özbay Murat Şahin İrfan Kara

https://doi.org/10.17826/cumj.1184876

Abstract

Hyperparathyroidism (HPT) is an endocrine disorder characterized by high secretion of parathyroid hormone. Brown tumor is one of the skeletal manifestations of HPT. Its overall prevalence is low (about 0.1%). Involvement of facial bones is extremely rare. The mandible is involved in 4.5% of cases. Primary HPT (PHPT)with vitamin D deficiency worsens the clinical course. Parathyroid adenoma is detected in ectopic places with a rate of 8.5%. 0.2% is intrathyroidal, 2% is located in different neck regions, 4.1% is in the upper mediastinum, and 2.2% is in the lower mediastinum. The prevalence of papillary thyroid cancer (PTC) in PHPT patients has been reported to range from 2% to 15%. In the literature, incidentally detected papillary thyroid cancer in patients with hyperparathyroidism was detected in patients who underwent total thyroidectomy associated with intrathyroidal parathyroid adenoma.
In this article, we present a case of incidental papillary thyroid carcinoma with ectopic parathyroid adenoma accompanied by severe vitamin D, and diagnosed during accompanying surgery following a brown tumor was considered after primary hyperparathyroidism was detected. However, the patient was admitted with a mass in the oral cavity, diagnosed before as a pyogenic granuloma with a delayed diagnosis without considering brown tumor because it was normocalcemic. It is the first case in the literature of papillary thyroid carcinoma detected incidentally with ectopic parathyroid adenoma in the thyrothymic region.

Keywords

brown tumor, ectopic parathyroid adenoma, normocalcemic hyperparathyroidism, papillary thyroid cancer

Oral kavitede kitle ve eşlik eden insidental papiller tiroid karsinomu ile başvuran normokalsemik primer hiperparatiroidizm olgusu

Abstract

Hiperparatiroidizm (HPT), yüksek paratiroid hormonu salgılanması ile karakterize bir endokrin bozukluktur. Brown tümör, HPT'nin iskelet belirtilerinden biridir. . Genel prevalansı düşüktür (yaklaşık %0,1) .Yüz kemikleri tutulumu son derece nadirdir. Mandibula vakaların %4,5’inde tutulur. D vitamini eksikliği ile birlikte olan Primer HPT (PHPT) klinik gidişi kötüleştirir. Paratroid adenomu %8.5 oranında ektopik yerlerde tespit edilir. %0.2 intratroidial ,%2 si boyunda farklı bölgelerde ve, %4.1 üst mediastende ve %2.2 alt mediastende bulunur. PHPT hastalarında, papiller tiroid kanseri (PTC) prevalansının %2 ila %15 arasında olduğunu bildirmiştir. . Literatürde hiperparatroidili hastalarda insidental olarak saptanan tiroid papiller kanseri intratroidial paratroid adenomu birlikteliğinde total troidektomi yapılan hastalarda saptanmıştır.
Biz bu yazıda, oral kavitede kitle ile başvurmuş, normokalsemik olduğu için önce brown tümör düşünülmeyen bu nedenle tanısı geciken piyojenik granülom tanısı alan , primer hiperparatiroidizm saptandıktan sonra brown tümör düşünülen, ciddi D vitaminin eşlik ettiği ektopik paratiroid adenomu ve buna eşlik eden cerrahi sırasında tanı konan insidental tiroid papiller karsinomu olgusunu sunuyoruz. Tirotimik bölgede ektopik paratiroid adenomu ile birlikte insidental olarak saptanan tiroid papiller karsinomu vakası olarak literatürdeki ilk vakadır.

Keywords

brown tümör, ektopik paratiroid adenomu, normokalsemik hiperparatroidizm, papiller tiroid kanseri

References

• Olvi, L.G.; Santini-Araujo, E. “Brown Tumor” of Hyperparathyroidism. In Tumors and Tumor-Like Lesions of Bone: For Surgical Pathologists, Orthopedic Surgeons and Radiologists; Santini-Araujo, E., Kalil, R.K., Bertoni, F., Park, Y.-K., Eds.; Springer: London, UK, 2015; pp. 815–825. ISBN 978-1-4471-6578-1)
• Manjunatha, B.S.; Purohit, S.; Harsh, A.; Vangala, N. A Complex Case of Brown Tumors as Initial Manifestation of Primary Hyperparathyroidism in a Young Female. J. Oral Maxillofac. Pathol. 2019, 23, 477.
• Olvi, L.G.; Santini-Araujo, E. “Brown Tumor” of Hyperparathyroidism. In Tumors and Tumor-Like Lesions of Bone: For Surgical Pathologists, Orthopedic Surgeons and Radiologists; Santini-Araujo, E., Kalil, R.K., Bertoni, F., Park, Y.-K., Eds.; Springer: London, UK, 2015; pp. 815–825. ISBN 978-1-4471-6578-1)
• Zou, H.; Song, L.; Jia, M.; Wang, L.; Sun, Y. Brown Tumor of Multiple Facial Bones Associated with Primary Hyperparathyroidism: A Clinical Case Report. Medicine 2018, 97, e11877. )
• Kar DK, Gupta SK, Agarwal A, Mishra SK. Brown tumor of the palate and mandible in association with primary hyperparathyroidism. J Oral Maxillofac Surg. 2001;59(11):1352-4.
• Cusano NE, Silverberg SJ, Bilezikian JP. Normocalcemic primary hyperparathyroidism. J Clin Densitom 2013;16:33-9.)
• Silverberg SJ. Vitamin D deficiency and primary hyperparathyroidism. J Bone Miner Res 2007; 22: V100)
• Rao DS, Hanosoge M, Divine GW, et al. Effect of vitamin D nutrition on parathyroid adenoma weight: pathogenetic and clinical implications. J Clin Endocrinol Metab 2000; 85: 1054–1058
• Silverberg SJ, Shane E, Dempster DW, et al. The effects of vitamin D insufficiency in patients with primary hyperparathyroidism. Am J Med 1999; 107: 561–567.)
• Ogburn PL, Black BM. Primary hyperparathyroidism and papillary adenocarcinoma of the thyroid: report of four cases. Proc Staff Meet Mayo Clin 1956;31:295-8).
• Yazici P, Mihmanli M, Bozdag E, Aygun N, Uludag M. Incidental finding of papillary thyroid carcinoma in the patients with primary hyperparathyroidism. Eurasian J Med 2015;47: 194-8.)
• Cinamon U, Levy D, Marom T. Is primary hyperparathyroidism a risk factor for papillary thyroid cancer? An exemplar study and literature review. Int Arch Otorhinolaryngol 2015;19:42-5.)
• Pak CY, Oata M, Lawrence EC, Snyder W. The hypercalciurias. Causes, parathyroid functions, and diagnostic criteria. J Clin Invest. 1974;54(2):387e400.
• Zou, H.; Song, L.; Jia, M.; Wang, L.; Sun, Y. Brown Tumor of Multiple Facial Bones Associated with Primary Hyperparathyroidism: A Clinical Case Report. Medicine 2018, 97, e11877.)
• Palla B, Burian E, Fliefel R, et al. Systematic review of oral manifestations related to hyperparathyroidism. Clin Oral Investig 2018;22:1–27)
• Brabyn P, Capote A, Belloti M, et al. Hyperparathyroidism diagnosed due to brown tumors of the jaw: a case report and literature review. J Oral Maxillofac Surg 2017;75:2162–9.)
• Rao DS, Hanosoge M, Divine GW, et al. Effect of vitamin D nutrition on parathyroid adenoma weight: pathogenetic and clinical implications. J Clin Endocrinol Metab 2000; 85: 1054–1058.
• Silverberg SJ, Shane E, Dempster DW, Bilezikian JP 1999 The effects of vitamin D insufficiency in patients with primary hyperparathyroidism. Am J Med 107:561–567
• Carnevale V, Manfredi G, Romagnoli E, De Geronimo S, Paglia F, Pepe J, Scillitani A, D’Erasmo E, Minisola S 2004 Vitamin D status in female patients with primary hyperparathyroidism: does it play a role in skeletal damage? Clin Endocrinol (Oxf) 0:81– 86
• Panagopoulos A, Tatani I, Kourea HP, Kokkalis ZT, Panagopoulos K, Megas P. Osteolytic lesions (brown tumors) of primary hyperparathyroidism misdiagnosed as multifocal giant cell tumor of the distal ulna and radius: a case report. J Med Case Rep. 2018 Jun;25(1):176. 12
• Azria A, Beaudreuil J, Juquel JP, Quillard A, Bardin T. Brown tumor of the spine revealing secondary hyperparathyroidism. Report of a case. Joint Bone Spine. 2000;67(3):230–3.
• Grulois V, Buysschaert I, Schoenaers J, Debruyne F, Delaere P, Vander Poorten V. Brown tumour: presenting symptom of primary hyperparathyroidism. B-ENT. 2005;1(4):191–5
• N. Mahabob, S. Kumar, and S. Raja, “Palatal pyogenic granulomaa,” Journal of Pharmacy and Bioallied Sciences, vol. 5, no. 2, pp. 179–181, 2013.
• Dojcinovic, M. Richter, and T. Lombardi, “Occurrence of a pyogenic granuloma in relation to a dental implant,” Journal of Oral and Maxillofacial Surgery, vol. 68, no. 8, pp. 1874–1876, 2010)
• S. Y. Morita, H. Somervell, C. B. Umbricht, A. P. B. Dackiw, and M. A. Zeiger, “Evaluation for concomitant thyroid nodules and primary hyperparathyroidism in patients undergoing parathyroidectomy or thyroidectomy,” Surgery, vol. 144, no. 6, pp. 862–867, 2008.
• D. J. Phillips, D. I. Kutler, and W. I. Kuhel, “Incidental thyroid nodules in patients with primary hyperparathyroidism,” Head & Neck, vol. 36, no. 12, pp. 1763–1765, 2014)
• M. Celik, S. Guldiken, S. Ayturk, et al., “Benign and malignant thyroid gland diseases in the patients with primary hyperparathyroidism,” International Journal of Applied and Basic Medical Research, vol. 7, no. 2, pp. 117–120, 2017.
• C. Preda, D. Branisteanu, I. Armasu, et al., “Coexistent papillary thyroid carcinoma diagnosed in surgically treated patients for primary versus secondary hyperparathyroidism: same incidence, different characteristics,” BMC Surgery, vol. 19, no. 1, p. 94, 2019)
• L. A. Burmeister, M. Sandberg, S. E. Carty, and C. G. Watson, “thyroid carcinoma found at parathyroidectomy: association with primary, secondary, and tertiary hyperparathyroidism,” Cancer, vol. 79, no. 8, pp. 1611–1616, 1997.)
• Y.-X. Zheng, S.-M. Xu, P. Wang, and L. Chen, “Preoperative localization and minimally invasive management of primary hyperparathyroidism concomitant with thyroid disease,” Journal of Zhejiang University Science B, vol. 8, no. 9, pp. 626–631, 2007)
• Y. Xue, Z.-Q. Ye, H.-W. Zhou, B.-M. Shi, X.-H. Yi, and K.-Q. Zhang, “Serum calcium and risk of nonmedullary thyroid cancer in patients with primary hyperparathyroidism,” Medical Science Monitor, vol. 22, pp. 4482–4489)
• Yuan Liu, Siyi Guo, Shaowei Sang, Jinbo Liu, Lin Qi, Bin Lv, and Xiaoli Zhang ‘’ Differences in Clinicopathological Characteristics of Papillary Thyroid Carcinoma between Symptomatic and Asymptomatic Patients with Primary Hyperparathyroidism ‘’ Hindawi International Journal of Endocrinology Volume 2021, Article ID 9917694, 7 pages)
• .C. Preda, D. Branisteanu, I. Armasu, et al., “Coexistent papillary thyroid carcinoma diagnosed in surgically treated patients for primary versus secondary hyperparathyroidism: same incidence, different characteristics,” BMC Surgery, vol. 19, no. 1, p. 94, 2019)
• K. Kutluturk, E. Otan, M. A. Yagci, S. Usta, C. Aydin, and B. Unal, “Thyroid pathologies accompanying primary hyperparathyroidism: a high rate of papillary thyroid microcarcinoma,” Turkish Journal of Surgery, vol. 30, no. 3, pp. 125–128, 2014)
• K. Çetin, H. E. Sıkar, S¸. Temizkan et al., “Does primary hyperparathyroidism have an association with thyroid papillary cancer? A retrospective cohort study,” World Journal of Surgery, vol. 43, no. 5, pp. 1243–1248, 2019)
• S. Vaccarella, S. Franceschi, F. Bray, C. P. Wild, M. Plummer, and L. Dal Maso, “Worldwide thyroid-cancer epidemic? +e increasing impact of overdiagnosis,” New England Journal of Medicine, vol. 375, no. 7, pp. 614–617, 2016)
• Wright MC, Jensen K, Mohamed H, Drake C, Mohsin K, Monlezun D, et al. Concomitant thyroid disease and primary hyperparathyroidism in patients undergoing parathyroidectomy or thyroidectomy. Gland Surg 2017;6:368 )
• European Thyroid Association Guidelines for Ultrasound Malignancy Risk Stratification of Thyroid Nodules in Adults: The EU-TIRADS Gilles Russa Steen J. Bonnemab Murat Faik Erdoganc Cosimo Duranted Rose Ngue Laurence Leenhardta 2017 )
• Abdul-Wahed N Meshikhes, Sohail A Butt, Basima A Al-Saihati Combined parathyroid adenoma and an occult papillary carcinoma, Saudi Med J 2004 Nov;25(11):1707-10,
• Velimezis G, Ioannidis A, Apostolakis S, et al. Concurrent intrathyroidal thymus and parathyroid in a patient with papillary thyroid carcinoma: a challenging diagnosis. Endocrinol Diabetes Metab Case Rep 2017;2017: pii: 17-0015
• Bernd HW, Horny HP. Unusually close association of ectopic intrathyroidal parathyroid gland and papillary microcarcinoma of the thyroid. Histopathology 2004;44:300–1.)
• Lappas D1, Noussios G, Anagnostis P, et al. Location, number and morphology of parathyroid glands: results from a large anatomical series. Anat Sci Int 2012;87:160–4.)