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Sarkoidozlu Bir Hastada Papiller Tiroid Kanseri ile Birlikte Seyreden Graves Hastaligi / Graves' Disease and Papillary Thyroid Carcinoma in a Patient with Active Sarcoidosis:.

Abstract

Sarcoidosis is a systemic granulomatous disease of unknown etiology. In most cases, mediastinal lymph nodes and lung parenchyma are involved. In addition, the eyes, skin, abdominal organs, central nervous system or the joints might be involved during the course of the disease. Sarcoidosis has been found to be related with other autoimmune diseases such as thyroiditis. In this report, we present the case of a patient in whom hyperthroidism was found and Graves' disease and papillary thyroid carcinoma were diagnosed while being investigated with a preliminary diagnosis of sarcoidosis. A 50-year-old male patient was admitted to our chest diseases clinic with the complaints of cough with colorless sputum, joint pain, weakness, weight loss (36 kgs), palpitations, tremor of the hands and pain in the right eye. Mediastinal lympadenopathy was detected on CT scan. Laboratory tests demonstrated hypercalcemia and low levels of parathyroid hormone (PTH). Eye examination revealed uveitis. Bronchoscopy was planned to confirm the diagnosis of sarcoidosis. Thyroid function test was performed and the patient turned out to have hyperthyroidism with high free T4 and low TSH levels. TSH receptor antibody titer was 71 U/L (0-14 U/L). Ultrasonographic examination demonstrated enlarged thyroid gland and heterogeneous parenchyma with increased blood flow pattern. 4- and 24-hour radioiodine uptakes were 30% and 60%, respectively and scintigraphy findings were compatible with diffuse toxic goiter. On Hertel exophthalmometer, the measurements of the right and left eye were 23 and 24 mm, respectively. The diagnosis of Graves' disease was made and thionamide therapy was started. Since active ophthalmopathy was present and the patient did not want to receive medical therapy for a long time, surgery was the best option for definitive therapy. The patient was prepared for the surgery with lugol solution (3x10 drops daily) and after eutyroidism was achieved, bilateral total thyroidectomy was performed. Before and after surgery, the patient was given 20 mg methylprednisolone in order to control hypercalcemia due to sarcoidosis and to control eye symptoms caused by Graves' disease. The postoperative pathology report was compatible with thyroiditis and 6 mm micropapillary carcinoma at one focal area in the right lobe. The patient received radioactive iodine therapy after thyroidectomy. Sarcoidosis occurs as a result of immune response created against environmental and genetic factors in the presence of a triggering agent. The disease can involve many organ and systems. The classical pathological finding is granulomatosis without caseification. Autoimmune thyroiditis may accompany the disease in any stage and hypo or hyperthyroidism may develop. In our case, hyperthyroidism developed during the active phase of the disease and, interestingly, papillary thyroid cancer was detected although there were no thyroid nodules found by preoperative thyroid ultrasonography. Turk Jem 2012; 16: 72-4

Key words: Sarcoidosis, tyroiditis, papillary cancer

Ozet

Sarkoidoz etiyolojisi bilinmeyen sistemik granulomatoz bir hastaliktir. Genellikle mediastinal lenf nodlari ve akciger parankimi tutulur. Bunlara ek olarak goz, cilt, abdominal organlar, santral sinir sistemi veya eklemler de etkilenebilir. Sarkoidozun bazi otoimmun hastaliklar ile iliskili oldugu bilinmektedir. Bu bildiride sarkoidoz on tanisi ile tetkik edilirken hipertroidi saptanan ve Graves hastaligi ve papiller tiroid karsinomu tanisi konulan bir olgu sunulmustur. 50 yasinda erkek hasta, bir yildir olan oksuruk, balgam, eklem agrisi, kilo kaybi, carpinti, tremor, asiri terleme ve sag gozde agri yakinmasi ile Gogus hastaliklari poliklinigine basvurdu. Hastanin akciger tomografisinde mediastinal lenfadenopati saptandi. Laboratuar incelemesinde serum Ca: 12 mg/dL, PTH: 9 pg/mL bulundu. Goz muayenesinde uveit saptanan hastaya sarkoidoz on tanisi ile bronkoskopi planlandi. Islem oncesi tetkiklerde hipertiroidi tespit edildi. TSH Reseptor Antikor duzeyi: 71 U/L(0-14 U/L) idi. Tiroid ultrasonografisinde tiroid glandi parankimi heterojendi ve her iki lob boyutu artmisti. Color-doppler incelemede tiroid glandinin kanlanmasinin artmis oldugu izlendi. Radyoaktif iyot up-take olcumu 4.saatte %30, 24.saatte %60 olup artmisti. Goz muayenesinde Hertel olcumu sag/sol: 23/24 mm idi. Hastaya fizik muayene, laboratuar ve goruntuleme tetkikleri sonucu Graves Hastaligi tanisi konuldu. Aktif goz tutulumu olan hasta potasyum iyodur (1 hafta sure ile 3x10 damla) ile operasyona hazirlandi ve otiroidi saglaninca bilateral total tiroidektomi uygulandi. Operasyon oncesi ve sonrasinda sarkoidoza bagli hiperkalsemisi olan ve PTH degeri baskili olan hastaya 1 hafta sure ile gunluk 20 mg metilprednizolon ve hidrasyon tedavisi verildi. Operasyon sonrasi histopatolojik incelemede tiroidit zemininde bir odakta papiller ca saptandi. Sarkoidoz cevresel ve genetik faktorlerin bir araya gelmesi sonucu tetikleyici bir ajanin varliginda immun cevabin olusmasi sonucu ortaya cikar. Hastalik bircok organ ve sistemi tutabilir. Klasik patolojik tutulum kazeifikasyonsuz granulomlardir. Sarkoidoza hastaligin herhangi bir evresinde otoimmun tiroidit eslik edebilir ve hipotiroidi veya hipertirodi gelisebilir. Bizim olgumuzda hipertiroidi ve sarkoidoz tanilari es zamanli olarak konulmustur. Ilginc olarak, tiroid ultrasonografisinde nodul olmadigi halde oftalmopati nedeniyle hipertiroidinin kalici tedavisine yonelik yapilan tiroidektomi sonrasi parankim icinde bir odakta insidental olarak tiroid papiller karsinomuna rastlanmistir. Turk Jem 2012; 16: 72-4

Anahtar kelimeler: Sarkoidoz, tiroidit, papiller kanser

Introduction

Sarcoidosis is a systemic granulomatous disorder of unknown etiology. In most cases, mediastinal lymph nodes and lung parenchyma are involved. In addition, the eye, skin, abdominal organs, central nervous system and the joints might be involved during the course of the disease (1), (2). Sarcoidosis is known to be related with other autoimmune conditions such as thyroiditis (3), (4). In this report we present a case in whom hyperthroidism was found and Graves disease and papillary thyroid carcinoma were diagnosed while being investigated with a preliminary diagnosis of sarcoidosis.

Case

A 50-year-old male patient, who was using oral antidiabetic agents for type 2 diabetes, was admitted to our chest diseases clinic with complaints of cough with colorless sputum, joint pain, weakness, weight loss (36 kgs), palpitations, tremor of the hands and pain in the right eye. Mediastinal lymphadenopathy was observed on CT scan. Hypercalcemia (12 mg/dl) was also detected in routine biochemistry tests whereas parathormone level was low (9 pg/ml). The patient had uveitis on the ophtalmological examination. Bronchoscopy was planned to confirm the diagnosis of sarcoidosis. Thyroid function tests were performed; thyrotoxicosis was detected with high free T4 and low TSH levels [TSH: 0.005 uIU/mL (0.27-4.2 uIU/mL), ST3: 26 pg/mL (1.8-4.6 pg/mL), St4: 7 ng/dL (0.9-1.7 ng/dL), anti TG: 12 IU/ml(0-115 IU/ml), anti TPO: 5 IU/ml (0-34 IU/ml)]. Consultation was made and the patient was transferred to the endocrinology and metabolism clinic for the differential diagnosis and the treatment of thyrotoxicosis. TSH receptor antibody titer was 71 U/L (0-14 U/L).On the ultrasonographic examination, thyroid gland size was found to be enlarged and parenchyma was heterogeneous with increased blood flow pattern. 4- and 24- hour radioiodine uptake were high and scintigraphy findings were compatible with diffuse toxic goiter. Soft tissue involvement and edema were noticed on the physical examination of the eyes (picture 1). Hertel measurements of the right and left eye were 23 and 24 mm, respectively. We made the diagnosis of Graves' disease and started antithyroid thionamide therapy. Since there was active ophthalmopathy and the patient did not want to receive propylthiouracil for a long time, we have decided that total thyroidectomy was the best option for definitive therapy. The patient was prepared for the surgery with lugol solution (3x10 drops daily) and, after eutyroidism was achieved, bilateral total thyroidectomy was performed. Before and after surgery, the patient was given 20mg methylprednisolone in order to control hypercalcemia due to sarcoidosis and to control eye symptoms of Graves' disease. The postoperative pathology report was compatible with thyroiditis and 6mm micropapillary carcinoma at one focal area in the right lobe. After thyroidectomy, bronchoscopy was made and the diagnosis of sarcoidosis was confirmed. Steroid therapy was discontinued and the patient received radioactive iodine therapy after surgery.

Discussion

Sarcoidosis occurs as a result of immune response created against environmental and genetic factors in the presence of a triggering agent. The disease can involve many organ and systems. The classical pathological finding is granulomatosis without caseification (4).In the granulomatous tissue, active form of vitamin D is produced and as a result of that hypercalcemia and hypercalciuria develop. The endocrine organs can also be involved. When the hypothalamus and hypophysis are affected, polydipsia, diabetes insipidus, hypogonadism, hypoadrenalism, hypothyroidism and, less frequently, hyperprolactinemia might be seen. Rarely, adrenal glands and thyroid gland involvement and hypofunction can develop. In addition to that, autoantibodies produced against self-antigens play a major role in the pathogenesis of the disease (5), and this phenomenon explains the co-occurrence of other autoimmune diseases (6), (7).

Autoimmune thyroiditis might accompany sarcoidosis at any stage and hypo or hypertyrodism might develop. Although the incidence of hyperthyroidism is considered rare (11), one study (12) showed a significantly higher prevalence of Graves' disease in sarcoidosis patients than in matched controls. In a study (8), 10 autoimmune thyroiditis cases were reported among 348 cases of sarcoidosis. Six of ten patients developed Hashimoto's thyroiditis, 3 Graves' disease and one had postpartum thyroiditis. In one patient, tyroditis had developed ten years before the diagnosis of sarcoidosis was made. In the other 5 patients, thyroiditis occurred 4-17 months after the diagnosis of sarcoidosis. In another study reported by Papadopoulos et al., 13(%16) thyroiditis was detected among 78 sarcoidosis patients and the incidence of thyroid dysfunction was significantly higher compared to controls (9). Nakamura et al. (10) have reported that 17 of 62 patients with sarcoidosis had high levels of serum thyroid autoantibodies. However, only 7 of all autoantibody positive patients had Hashimato's thyroiditis.

Concomitant occurrence of papillary cancer, Graves' disease and thyroid sarcoidosis had been reported before by Zimmermann-Belsing et al. (13). Their patient, known to have sarcoidosis, underwent total thyroidectomy because of inadequate control of hyperthyroidism with thiamazole treatment in the course of Graves' disease (the goiter gradually enlarged). Thyroid sarcoidosis and papillary cancer of the gland with metastases to the lymph nodes were incidental findings in histological examination. The difference between our case and that patient is the absence of sarcoid reaction within the gland. Therefore, we should keep in mind that sarcoidosis may coexist with thyroid cancer and sometimes papillary cancer might mimic sarcoid infiltration and make it difficult to interpret fine needle aspiration cytology (14).

Usually autoimmune thyroid disease does not develop during the period of activity of sarcoidosis (8). In our case, the diagnosis of sarcoidosis and Graves' disease were synchronous. Graves' disease occurred during the active phase of the disease, unlike the previous reports. TSH receptor antibody titers were high in our patient and there was eye involvement (Graves' ophthalmopathy). In most of the other cases, sarcoidosis was with the symptoms of Lofgren syndrome (arthritis, erythema nodosum, bilateal hilar lymphadenopathy) whereas there was more systemic involvement in our case. Our patient had no family history of thyroid disease.

In conclusion, the presence of other autoimmune diseases should be considered in patients with sarcoidosis. In addition, a careful physical examination of the thyroid gland as well as thyroid function tests and thyroid antibody testing should be performed. We should be careful before starting potassium iodide therapy for erythema nodosum because it can trigger hyperthyroidism in patients with thyroiditis, especially in iodine deficient cases.

DOI: 10.4274/Tjem.2042

Address for Correspondence/Yazisma Adresi: Sefika Burcak Polat MD, Ataturk Hospital, Endocrinology, Ankara, Turkey

Phone: +90 312 291 25 25 E-mail: burcakugurlu@gmail.com Recevied/Gelis Tarihi: 27.05.2012 Accepted/Kabul Tarihi: 28.09.2012

Turkish Journal of Endocrinology and Metabolism, published by Galenos Publishing.

References

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(3.) Hancock BW, Millard LG. Sarcoidosis and thyrotoxicosis.A study of five patients. Br J Dis Chest 1976;70:129-33.

(4.) Bell NH. Endocrine complications of sarcoidosis. Endocrinol Metab Clin North Am 1991;20:645-54.

(5.) Chida K, Sato A, Yasuda K, et al. Clinical aspects of sarcoidosis with autoantibodies. Nihon Kyobu Shikkan Gakkai Zasshi 1989;27:194-9.

(6.) Hunninghake GW, Crystal RG. Mechanisms of hypergammaglobulinemia in pulmonary sarcoidosis. Site of increased antibody production and role of T lymphocytes. J Clin Invest 1981;67:86-92.

(7.) Wiesenhutter CW, Sharma OP. Is sarcoidosis an autoimmune disorder?: Report of four cases and review of literature. Semin Arthritis Rheum 1979;9:124-44.

(8.) Isern V, Lora-Tamayo J, Capdevila O, Villabona C, Mana J. Sarcoidosis and autoimmune thyroid disease. A case series of ten patients. Sarcoidosis Vasc Diffuse Lung Dis 2007;24:148-52.

(9.) Papadopoulos KI, Hornblad Y, Lilijebladh H, Hallengren B. High frequency of endocrine autoimmunity in patients with sarcoidosis. Eur J Endocrinol 1996;134: 331-6.

(10.) Nakamura H, Genma R, Mikami T, et al. High incidence of positive autoantibodies against thyroid peroxidase and thyroglobulin in patients with sarcoidosis. Clinl Endocrinol (Oxf) 1997;46:467-72.

(11.) Antonelli A, Fazzi P, Fallahi P, Ferrari SM, Ferrannini E. Prevalence of hypothyroidism and Graves disease in sarcoidosis. Chest 2006;130:526-32.

(12.) Yarman S, Kahraman H, Tanakol R, Kapran Y. Concomitant association of thyroid sarcoidosis and Graves' disease. Horm Res 2003;59:43-6.

(13.) Zimmermann-Belsing T, Christensen L, Hansen HS, et al. A case of sarcoidosis and sarcoid granuloma, papillary carcinoma, and Graves' disease in the thyroid gland. Thyroid 1997;7:901-3.

(14.) Kmiec P, Lewandowska M, Dubaniewicz A, et al. Two cases of thyroid sarcoidosis presentation as painful, recurrent goiter in patients with Graves' disease. Arq Bras Endocrinol Metabol 2012;56:209-14.

Sefika Burcak Polat, Mukremin Er *, Eda Demir Onal, Reyhan Ersoy, Bekir Cakir

Ataturk Hospital, Endocrinology, Ankara, Turkey

* Ataturk Hospital, Chest Diseases, Ankara, Turkey
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Title Annotation:Case Report Olgu Sunumu
Author:Polat, Sefika Burcak; Er, Mukremin; Onal, Eda Demir; Ersoy, Reyhan; Cakir, Bekir
Publication:Turkish Journal of Endocrinology and Metabolism
Date:Sep 1, 2012
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