DOI: 10.14744/ejmo.2017.98608 EJMO 2017;1(1):49–52 Case Report Synchronous Parathyroid Carcinoma and Multifocal Papillary Thyroid Carcinoma: A Case Report Fatih Kuzu,1 Ahmet Cinkaya,2 Mehmet Fatih Ekici,3 Hilmi Kodaz,4 Aysenur Deger5 1Department of Endocrinology, Evliya Celebi Training and Research Hospital, Kutahya, Turkey 2Department of Radiation Oncology, Dumlupinar University Faculty of Medicine, Kutahya, Turkey 3Department of General Surgery, Evliya Celebi Training and Research Hospital, Kutahya, Turkey 4Department of Medical Oncology, Acıbadem Eskisehir Hospital, Eskisehir, Turkey 5Department of Pathology, Dumlupinar University Faculty of Medicine, Kutahya, Turkey Abstract The coexistence of parathyroid carcinoma and thyroid carcinoma is extremely rare. Presently described is a case with multinodular goiter and parathyroid adenoma pre-diagnosis for which operation was planned that was postopera- tively diagnosed with multifocal papillary thyroid carcinoma accompanying parathyroid carcinoma. A review of the current literature is also provided. Keywords: Multinodular goiter, papillary thyroid carcinoma, parathyroid adenoma, parathyroid carcinoma Cite This Article: Kuzu F, Cinkaya A, Ekici M, Kodaz H, Deger A. Synchronous Parathyroid Carcinoma and Multifocal Papillary Thyroid Carcinoma: A Case Report. EJMO. 2017; 1(1): 49-52 arathyroid carcinoma (PC) is a rare endocrine tumor neoplasia type 2 (MEN2, Sipple syndrome). In contrast, the Pand constitutes less than 1% of all cases of primary hy- coexistence of PHPT and non-medullary thyroid carcinoma perparathyroidism (PHPT).[1] It is difficult to diagnose PC is very rare (1–2%). The coexistence of PC and thyroid car- preoperatively, as its clinical features are similar to PHPT. cinoma is extremely rare. Only 12 cases of coexistence of Severe end-organ damage, including symptoms related to PC and non-medullary thyroid carcinoma were returned in hypercalcemia, renal insufficiency, bone diseases, cardiac PubMed and Medline searches. arrhythmia, and neurocognitive dysfunction may be ob- served. PC has a more aggressive progress compared with Case Report clinical PHPT, and may emerge with a mass in the neck. A 52-year-old female patient following-up diagnosis of os- On the other hand, some patients show no symptoms. Al- teoporosis in the physical therapy and rehabilitation clinic though it is very rarely observed, patients with nonfunc- was referred to the endocrinology clinic due to hypercal- tional PC may also have normal calcium and parathormone cemia found in routine examinations. The patient had re- [2] (PTH) levels. ceived nonsteroidal anti-inflammatory and muscle relaxant The coexistence of PHPT with thyroid disease is not com- treatment for muscle aches and cramps during follow-up mon. Thyroid pathology has been reported in 15% to 70% for osteoporosis. Examination of the patient's history re- of patients with PHPT. The coexistence of PHPT with med- vealed diabetes mellitus type 2, hypertension, multinodu- ullary thyroid cancer is common in multiple endocrine lar goiter, peptic ulcer, and depression, and she was receiv- Address for correspondence: Fatih Kuzu, MD. Dumlupinar Universitesi, Evliya Celebi Egitim ve Arastirma Hastanesi, Endokrinoloji Klinigi, Kutahya, Turkey Phone: +90 530 826 62 04 E-mail: [email protected] Submitted Date: May 22, 2017 Accepted Date: July 03, 2017 Available Online Date: August 03, 2017 ©Copyright 2017 by Eurasian Journal of Medicine and Oncology - Available online at www.ejmo.org Kuzu et al., Parathyroid Papillary Thyroid Cancer / doi: 10.14744/ejmo.2017.98608 50 ing medical treatment in line with these diagnoses. There was no known thyroid or parathyroid cancer in her family history. Physical examination results were weight of 76 kg, height of 163 cm, blood pressure of 135/85 mmHg, thyroid stage 1b, and with palpation it was found that she had nod- ules of moderate hardness, approximately 1-1.5 cm in size in both lobes. Serum biochemical evaluation values were alkaline phos- phatase: 74 U/L, calcium: 11.4 mg/dL, phosphorus: 2.6 mg/dL, and 25-hydroxy vitamin D: 11.8 ng/mL. Full blood count was normal. Hormonal evaluation values were thy- roid-stimulating hormone: 0.69 uIU/mL, free thyroxine: 0.69 ng/dL and PTH: 208 pg/mL. The patient's bone scin- tigraphy was compatible with osteoporosis, and urinary sonographic imaging was assessed as normal. Follow-up sonographic imaging based on diagnosis of Figure 1. Thyroid papillary carcinoma. Hematoxylin and eosin x100. multinodular goiter and hypothyroidism revealed hetero- geneous, hypoechoic, and isoechoic nodules, the largest of which in the right lobe was 15 × 9 × 8 mm and 17 × 10 × 6 mm in the left lobe. Some nodules had faint contour and some included coarse calcification. In addition, a hy- poechoic lesion, thought to be parathyroid adenoma, 18 × 10 × 5 mm in size was detected adjacent to the right lower lobe of the thyroid and extending to the posterior carot- id artery. Activity involvement compatible with parathy- roid adenoma in the thyroid area and mediastinum were not detected in the scintigraphic evaluation of the lesion. Result of fine needle aspiration (FNA) biopsy of thyroid nodules was benign. PTH level was 3482 pg/mL after PTH washout of the suspected parathyroid adenoma lesion. The patient was referred to the general surgery clinic with a recommendation of total thyroidectomy and parathyroid Figure 2. Parathyroid carcinoma and common area of necrosis with surgery, since thyroid cancers are typically asymptomatic eosinophilic features. Hematoxylin and eosin x100. and can easily be overlooked in patients with hyperpara- thyroidism. PC was considered, since the parathyroid-orig- inated lesion demonstrated invasion into the common ca- rotid artery during surgery accompanied by intraoperative gama probe application. The operation concluded with en bloc resection and right lobectomy because the PC was suspicious in frozen sections. Pathology report of the histo- morphological and histochemical findings was PC. The nodule on the right thyroid lobe was reported as a classic variant papillary microcarcinoma. Nodular hyperplasia and lymphocytic infiltration were detected in the thyroid tissue outside the tumor (Figures 1–3). PTH, phosphorus, and cal- cium levels were found to be 41 pg/mL, 3 mg/dL, and 8.2 mg/dL, respectively, after the surgery. Oral calcium and vi- tamin D3 treatment was planned. Thoracic and abdominal tomographic images and bone scintigraphy of the patient Figure 3. Papillary carcinoma and tumor cells with a frosted glass ap- were normal. pearance. Hematoxylin and eosin x400. 51 EJMO Complementary thyroidectomy was performed 6 weeks mL. FNA biopsy process performed on patients who are later. The pathology was reported as papillary thyroid can- thought to have PC carries the risk of tumor seeding. The cer. The tumor was 10 × 8 × 5 mm in size. PTH washout procedure can be used to distinguish sus- The patient was treated with 100 mL of radioactive iodine picious parathyroid lesions that have negative sestamibi for papillary thyroid carcinoma. As the patient had a nor- scan but detected on sonographic and CT scans from neg- mocalcemic process, follow-up of 1-thyroxine treatment in ative thyroid lesions and metastatic/non-metastatic lymph suppressive dose was initiated. nodes.[9] Although a relationship between hyperparathyroidism and Discussion well differentiated thyroid carcinoma is rare, it has been re- PC pathogenesis is not fully known. Although it is gener- ported in numerous studies. A total of 824 primary, second- ally seen as a sporadic disease, it can also emerge as part ary, and tertiary hyperparathyroidism patients who under- of a genetic syndrome. Hyperparathyroidism jaw tumor went minimal lobectomy were evaluated in a retrospective syndrome (HPT-JT), MEN1, MEN2 and isolated familial hy- study conducted by Burmeister et al. Thyroid carcinoma perparathyroidism are genetic syndromes reported to be was detected in 18 (2.6%) of 700 patients with PHPT, and associated with PC. The mutations of the MEN1 gene seen 22 patients were found to have thyroid carcinoma among in MEN1 syndrome may be responsible. As a result of the total of 824 patients. One of the 22 patients was diagnosed MEN1 gene mutation, the inhibitory effect of transforming with follicular carcinoma, while 21 patients were diagnosed growth factor beta/SMAD3 on parathyroid cell prolifera- with papillary carcinoma. Thyroid carcinoma was not found tion is eliminated.[3, 4] Mutations in RET proto oncogenes in any of the 9 patients with PC in this retrospective study. [10, 11] are found in MEN2 syndrome. HPT-JT is associated with an Nodular thyroid disease was detected in 102 of the onco suppressor parafibromin (HRPT2/CDC73) gene muta- 200 patients for whom surgery was planned with diagnosis tion. Abnormal expression of microRNAs (miRNAs) also has of hyperparathyroidism in the study reported by Morita et an important role in the development and progression of al. Thyroid cancer was identified in 12 patients (6%) after PC.[5, 6] Genetic syndromes are the only precisely defined surgery. All cases had thyroid papillary cancer, with 1 case risk factor. PC has been reported previously in cases with of follicular variant. The incidence of thyroid cancer in hy- radiation exposure to the head and neck region, and in pa- perparathyroidism patients who underwent surgery in sev- tients with secondary or tertiary hyperparathyroidism due eral series varied between 2.2% and 17.6%.[12] to chronic renal insufficiency.[7,