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Synchronous intrathyroidal parathyroid carcinoma and thyroid carcinoma: case report and review of the literature

BMC Endocrine Disorders volume 21, Article number: 60 (2021) Cite this article

Abstract

Background

Parathyroid carcinoma is a rare endocrine malignancy, rarer when synchronous with a non medullary well differentiated thyroid carcinoma. Parathyroid carcinoma accounts of 0.005% of all malignant tumors and it is responsible for less than 1% of primary hyperparathyroidism. The intrathyroidal localization of a parathyroid gland is not frequent with a reported prevalence of 0.2%. Carcinoma of parathyroids with intrathyroidal localization represents an even rarer finding, reported in only 16 cases described in literature. The rare constellation of synchronous parathyroid and thyroid carcinomas has prompted us to report our experience and perform literature review.

Case presentation

We herein report a case of a 63-years-old man with multinodular goiter and biochemical diagnosis of hyperparathyroidism. Total thyroidectomy with radio-guide technique using gamma probe after intraoperative sesta-MIBI administration and intraoperative PTH level was performed. The high radiation levels in the posterior thyroid lobe discovered an intrathyroidal parathyroid. Microscopic examination revealed a parathyroid main cell carcinoma at the posterior thyroidal left basal lobe, a classic papillary carcinoma at the same lobe and follicular variant of papillary carcinoma at the thyroidal right lobe. To the best of our knowledge, this is the first case documenting a synchronous multicentric non medullary thyroid carcinomas and intrathyroidal parathyroid carcinoma.

Conclusions

Our experience was reported and literature review underlining challenging difficulties in diagnostic workup and surgical management was carried out.

Peer Review reports

Background

The intrathyroidal localization of a parathyroid gland is not frequent, with a reported prevalence of 0.2% [1]. Intrathyroidal parathyroids (IP) could also become pathological and primary hyperparathyroidism supported by an IP has an incidence of 1% [2].

Parathyroid carcinoma (PC) accounts of 0.005% of all malignant tumors and it is responsible for less than 1% of primary hyperparathyroidism [3, 4]. Carcinoma of parathyroids with intrathyroidal localization represents an even rarer finding, reported in only 16 cases described in literature [5,6,7,8,9,10,11,12,13,14,15,16,17,18,19,20].

The association between hyperparathyroidism and non-medullary well differentiated thyroid carcinoma is found in 2.4–3.7% of hyperparathyroidism cases [21], but hyperparathyroidism is mainly observed in benign parathyroid diseases [21,22,23].

At the best of our knowledge, only 11 cases of synchronous extrathyroidal parathyroid carcinomas and non-medullary well differentiated thyroid carcinoma are reported up to date in literature [24,25,26,27,28,29,30,31,32,33,34]. Indeed, our patient is the first documented case of synchronous non-medullary well differentiated thyroid carcinoma and parathyroid carcinoma with an intrathyroidal localization.

The rare constellation of synchronous thyroid carcinoma and intrathyroidal parathyroid carcinoma has prompted us to report this case and perform literature review to stress the challenging difficulties in diagnostic workup and surgical management.

Case presentation

A 63 years-old Caucasian male patient, with a negative family history of thyroid diseases and without a personal anamnesis of neck irradiation, was referred to general surgery unit for multinodular goiter, bigger on the right side, complicated by a tracheal deviation and symptoms of compression.

Routine preoperative tests, unexpectedly, showed a mild serum hypercalcemia (13.3 mg / dL) [normal range (n.r.): 8.6–10.2)], hypophosphatemia [2.2 mg / dL (n.r. 2.7–4.5)], with no symptomatology of urolithiasis, myasthenia, osteoarthralgia, dyspepsia, constipation or psychotic depression. Serum parathyroid hormone (PTH) value was increased 159 pg / mL (n.r. 4.6–58.1), the urine calcium was 896 mg / 24 h (n.r. 100–400) and urine phosphorus was 1000 mg / 24 h (n.r. 400–1300). No kidney disorders were found thus a diagnosis of primary hyperparathyroidism was made.

A neck ultrasound (US) exam confirmed a multinodular goiter without extra-thyroid lump: in particular, a 14 mm-diameter nodule with intravascular spot signal at Doppler exam was observed at the left posterior base of the thyroid. The parathyroid subtraction scintigraphy with 99mTechnetium and Sesta-Meta-iodo-Benzylguanidine showed a strong posterior signal at the left thyroid base in correspondence of the nodule (Fig. 1). No pathological signal was observed in other extrathyroidal sites.

Fig. 1
figure 1

Scintigraphy with 99mtechnetium: Late hypercaptation at lower left pole of the thyroid, no mediastinal uptake

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Therefore, these observations suggested the suspect of an IP adenoma: the decision was made not to proceed with a needle aspiration, due to the risk of severe fibrosis that could complicate the final histological diagnosis [35].

Otherwise fine-needle cytology (FNC) on the major nodule of the thyroidal right lobe diagnosed Thy 2 according to Bethesda System.

The patient received an intravenous injection of 12.3 mCi of Tc99m-sestamibi approximately 1 hour prior to surgery. A total thyroidectomy with radio-guide technique using gamma probe after intraoperative sesta-MIBI administration and intraoperative PTH level evaluation was performed.

The intraoperative PTH level evaluation, performed with a chemiluminescence immunoassay, allows the determination of the serum bio-intact PTH value. The basal serum PTH level before thyroidectomy was 164 pg/mL. After 20 min from the surgery, the value decreased at 38 pg/mL and 30 min later at 19 pg/mL.

The use of the gamma probe after surgery was essential to identify high radiation levels in the posterior left thyroid lobe, where a yellow-brown nodule was found (Fig. 2).

Fig. 2
figure 2

Posterior view of the operative specimen, node of the lower left pole of the thyroid subcapsular seat, reddish-yellow in color

Full size image

Conversely, the postoperative thyroid lodge did not show any radiation activity.

The patient was discharged on the third post-operative day without complications and with normal range serum values of PTH, calcemia and phosphoremia.

Histopathological examination revealed a classic papillary carcinoma of 0.8 cm of diameter at the thyroidal left lobe (Fig. 3a), a follicular variant of a papillary carcinoma of 0.6 cm of diameter at the thyroidal right lobe (Fig. 3b) and a parathyroidal main cell carcinoma of 1.2 × 1.0 cm of diameter at the posterior thyroidal left basal lobe (Fig. 3c, d, Fig. 4). Photographs of histological slides were obtained using a digital acquisition system (Olympus DP2-SAL4).

Fig. 3
figure 3

Histopathological finding. a Conventional papillary carcinoma. Histological evaluation showing a neoplasm with a papillary architectural pattern (H&E, original magnification 200x). b Follicular variant of a papillary carcinoma. Histological evaluation showing a neoplastic proliferation with a follicular architectural pattern and cytological atypia including ground-glass nuclei and nuclear grooves (H&E, original magnification 200x). c-d Parathyroid carcinoma. Histological evaluation showing neoplastic islets separated by thick bands of connective tissue (yellow star). The neoplasm shows an infiltrative growth in the adjacent soft tissue (blue arrows). Thyroid tissue is present in the lower right (C, H&E, original magnification 40x). The neoplasm is constituted by main cells (D, H&E, original magnification 200x). Abbreviation: H&E: hematoxylin and eosin

Full size image
Fig. 4
figure 4

Nests of main cells (yellow star) associated with thyroid follicles (blue star) (H&E, original magnification 200x). Abbreviation: H&E: hematoxylin and eosin

Full size image

Postoperatively, 131I ablation therapy was administered at a dose of 50 mCi.

The patient was followed up closely for both malignancies. US examination was performed every 6 months in the first year and every year later. TSH, FT3, FT4, Thyreoglobulin (Tg), Thyreoglobulin Antibodies, PTH, calcemia and phosphoremia was scheduled every 3 months.

Tg value was 6 ng/ml before I-131 therapy and remained < 0.2 ng/ml after treatment and for the 7 years follow-up; serum calcium, phosphorus and PTH levels remained in the respective normal ranges, with multiple negative neck US.

Discussion and conclusions

In literature only 11 cases of simultaneous occurrence of extrathyroidal parathyroid carcinoma and thyroid tumor has been reported. To the best of our knowledge, the association of intrathyroidal parathyroid carcinoma and thyroid tumor has never been described.

We have illustrated this unique association and performed a review of the last 40 years’ literature on the association of synchronous parathyroid carcinomas and non-medullary well differentiated thyroid carcinomas.

Parathyroid carcinoma is usually sporadic or related to other diseases [(e.g., multiple Endocrine Neoplasia (MEN) 1 and MEN 2, single familial hyperparathyroidism (FIHP), hyperparathyroidism “jaw tumor” (HPTJT)].

Studies of hereditary and syndromic forms of PC have revealed some genetic mechanisms underlying PC. Somatic mutations of CDC73 gene, involved in the Jaw tumor syndrome, would be the cause of hypercalcemic disorders and can be identified in up to 70% of patients with PC. In one-third of cases the mutations are germline [4, 36,37,38].

Nearly 90% of parathyroid carcinomas are hyperfunctioning versus 7–10% of non-functioning forms [4]; this percentage is confirmed in our literature review of synchronous parathyroid carcinoma and well differentiated thyroid tumor with a value of 9.09%. In non-functioning forms the patient typically presents late because of “mass effect” of a palpable tumor or lymphadenopathies (15–30%), whereas the lung, liver or bone metastases are found in one third of the cases [3].

Instead, hyperfunctioning forms usually present with nephrolithiasis, myasthenia, psychotic depression, osteoarthralgia; 7–12% of the cases have a parathyroid crisis, usually with serum PTH value > 16 mg/dl, characterized by cardiac arrhythmias and altered consciousness that can lead to coma [4, 39].

Currently, clinical, biochemical or imaging criteria are not able to distinguish benignant from malignant diseases. A malignant diagnosis should be suspected in the following conditions: a) fast onset of acute symptoms; b) serum calcemia > 14 mg/dL, usually associated to parathyroid carcinomas in 65–75% of the cases; c) serum PTH level beyond 10 times the normal value (81% predictive positive value); d) metastasis detection on the radiological investigations [4, 39, 40].

The routine use of FNC is unnecessary and should be avoided because of fibrosis and related reactive changes that can make histologic interpretation of benign versus malignant disease difficult [35]. Moreover, the risk of lump rupture with malignant cell spread should be considered. Therefore, FNC is indicated only in case of recurrence suspicion.

In order to obtain a definitive diagnosis, histopathological examination is necessary using Shantz and Castleman criteria, such as trabecular pattern, mitotic figures detection, thick fibrous band and capsular and blood vessel invasion [41]. The immunohistochemical exam (PTH, GATA3, TTF-1, PAX8 and thyroglobulin) is useful to identify the uncertain forms [20].

According to our literature review summarized in Table 1, in 12 reports with synchronous parathyroid malignancy and thyroid disease, 11 patients were hypercalcemic, whereas the one reported by Savli et al. [28] was normocalcemic; in all of the 11 extrathyroidal cases reported there were no preoperative suspect of malignancy.

Table 1 Clinical features of 12 patients with synchronous parathyroid and thyroid carcinoma
Full size table

Preoperative diagnostic management is paramount to lead to the correct surgical approach of thyroidal and parathyroidal synchronous carcinomas.

In five out of 11 reported cases, a preoperative thyroidal FNC has been performed and was suggestive of malignancy; in the other six cases the diagnosis of malignancy was rendered at the histological examination.

In patients in whom the preoperative findings supported the diagnosis of thyroidal carcinoma, the planned surgical technique was to perform an en-bloc resection of parathyroids with total thyroidectomy, while ipsilateral and/or contralateral hemithyroidectomy was reserved to patients with the suspect of malignancy or with follicular thyroid neoplasm (Table 1).

Our experience and literature review assess that nowadays the treatment of this synchronous oncologic disease is incredibly challenging and specific guidelines are still missing.

In our case the preoperative clinical findings supported the diagnosis of a multinodular goiter (FNC on the major nodule Thy 2 according to Bethesda System), complicated by a tracheal deviation and symptoms of compression with a suspect of a parathyroidal adenoma.

Therefore, our patient underwent a total thyroidectomy because of his diagnosis and according to the gold standard surgical procedure. Moreover, this approach allowed the incidental discovery of the multicentric thyroid malignancy and the accidental excision of intracapsular parathyroid carcinoma.

In our experience, radioguided technique (gamma-probe) and the intraoperative PTH dosage turned out indispensable, in order to assess the completeness of the excision.

For the parathyroid carcinoma, indeed, bloc excision of the mass and any involved surrounding structures, preserving the integrity of the capsule, is the only curative approach for long term survival.

Ipsilateral lymphadenectomy of the central and/or laterocervical compartment is recommended in case of lymph node involvement evidence [3].

Recurrence of parathyroid cancer occurs in a range between 49 and 60% of the cases, with a mean time of 2.5–4.8 years after surgery [3]. Negative prognostic factors include young age and lymphadenopathies or metastases present at the onset. The most common recurrences are locoregional or distant metastasis involving lung, liver and bones [3, 39, 40, 42, 43].

Because of the exceptional rare condition for parathyroid cancer, there is no general consensus on treatment and follow-up [44]. According to the literature, patients should be followed up life-long measuring calcium and PTH and performing regular US surveillance [38, 44]. Moreover, our treatment strategy was based also on the evidence of thyroid malignancy; even if histopathological examinations revealed a multifocal thyroid microcarcinoma without signs of aggressive histology, the specific features of the patient, such as age > 60, sex, Tg value and patient preference have prompted us to recommend RAI remnant ablation, also according to nuclear counselling.

At 7 years-follow up, there is no evidence of both thyroidal and parathyroidal malignant recurrences or parathyroid hyperfunction.

According to our experience and recent studies, radical surgical approach is currently the goal standard of treatment for recurrences [3, 39, 40, 42, 45,46,47].

In fact, according to literature, only a few authors [48,49,50,51,52,53,54] attest the usefulness of radiotherapy and chemotherapy in improving rate of recurrence and survivor but the efficacy of adjuvant therapies has recently been disappointed in the management of recurrent parathyroid carcinoma, even if multicentric studies for improved long-term outcomes are still lacking [46, 47].

Nevertheless it is paramount to consider that the causes of death in patient with lymphatic recurrence or secondary malignance are more closely related to chronic and refractory hypercalcemia than the presence of metastatic mass [54].

In conclusion, our experience emphasizes that in case of intraoperative suspect of parathyroid carcinoma, an en-bloc resection of the parathyroid tumor and the ipsilateral thyroid lobe is mandatory, even if there is no evidence of thyroidal involvement.

This approach avoids a successive neck surgical procedure that could be justified by the high local and vascular aggressiveness of the carcinoma and could be related to serious and frequent complications as recurrent laryngeal nerve injury [3, 4, 45].

Availability of data and materials

The datasets used and/or analysed during the current study are available from the corresponding author on reasonable request.

Abbreviations

IP:

Intradthyroidal parathyroids

FNC:

Fine-needle cytology

n.r:

Normal range

US:

Ultrasound

Tg:

Thyreoglobulin

PTH:

Parathyroid hormone

PC:

Parathyroid carcinoma

MEN:

Multiple endocrine Neoplasia

FIHP:

Single familial hyperparathyroidism

HPTJT:

Hyperparathyroidism “jaw tumor”

References

  1. Lappas D, Noussios G, Anagnostis P, Adamidou F, Chatzigeorgiou A, Skandalakis P. Location, number and morphology of parathyroid glands: results from a large anatomical series. Anat Sci Int. 2012;87(3):160–4. https://doi.org/10.1007/s12565-012-0142-1.

    Article  PubMed  Google Scholar 

  2. Mazeh H, Kouniavsky G, Schneider DF, Makris KI, Sippel RS, Dackiw AP, et al. Intrathyroidal parathyroid glands: small, but mighty (a Napoleon phenomenon). Surgery. 2012;152(6):1193–200. https://doi.org/10.1016/j.surg.2012.08.026.

    Article  PubMed  Google Scholar 

  3. Wei CH, Harari A. Parathyroid carcinoma: update and guidelines for management. Curr Treat Options Oncol. 2012;13(1):11–23. https://doi.org/10.1007/s11864-011-0171-3.

    Article  PubMed  Google Scholar 

  4. Al-Kurd A, Mekel M, Mazeh H. Parathyroid carcinoma. Surg Oncol. 2014;23(2):107–14. https://doi.org/10.1016/j.suronc.2014.03.005.

    Article  PubMed  Google Scholar 

  5. Vila Duckworth L, Winter WE, Vaysberg M, Moran CA, Al-Quran SZ. Intrathyroidal parathyroid carcinoma: report of an unusual case and review of the literature. Case Rep Pathol. 2013;2013:198643.

    PubMed  PubMed Central  Google Scholar 

  6. Cook MI, Qureshi YA, Todd CE, Cummins RS. An unusual ectopic location of a parathyroid carcinoma arising within the thyroid gland. J Clin Endocrinol Metab. 2012;97(6):1829–33. https://doi.org/10.1210/jc.2011-3115.

    Article  CAS  PubMed  Google Scholar 

  7. Temmim L, Sinowatz F, Hussein WI, Al-Sanea O, El-Khodary H. Intrathyroidal parathyroid carcinoma: a case report with clinical and histological findings. Diagn Pathol. 2008;3(1):46. https://doi.org/10.1186/1746-1596-3-46.

    Article  PubMed  PubMed Central  Google Scholar 

  8. Herrera-Hernández AA, Aranda-Valderrama P, Díaz-Pérez JA, Herrera LP. Intrathyroidal parathyroid carcinoma in a pediatric patient. Pediatr Surg Int. 2011;27(12):1361–5. https://doi.org/10.1007/s00383-011-2904-6.

    Article  PubMed  Google Scholar 

  9. Tejera Hernández AA, Gutiérrez Giner MI, Vega Benítez V, Fernández San Millan D, Hernández JR. Intrathyroidal parathyroid carcinoma. A case report and review of literature. Endocrinol Nutr. 2016;63(1):46–8. https://doi.org/10.1016/j.endonu.2015.09.004.

    Article  PubMed  Google Scholar 

  10. Foppiani L, Del Monte P, Sartini G, Arlandini A, Quilici P, Bandelloni R, et al. Intrathyroidal parathyroid carcinoma as cause of hypercalcemia and pitfall of localization techniques: clinical and biologic features. Endocr Pract. 2007;13(2):176–81. https://doi.org/10.4158/EP.13.2.176.

    Article  PubMed  Google Scholar 

  11. Hussein WI, El-Maghraby TA, Al-Sanea O. Hyperfunctioning intrathyroidal parathyroid carcinoma. Saudi Med J. 2006;27(8):1226–9.

    PubMed  Google Scholar 

  12. Crescenzo DG, Shabahang M, Garvin D, Evans SR. Intrathyroidal parathyroid cancer presenting as a left neck mass. Thyroid. 1998;8(7):597–9. https://doi.org/10.1089/thy.1998.8.597.

    Article  CAS  PubMed  Google Scholar 

  13. Schmidt JL, Perry RC, Philippsen LP, Wu HH. Intrathyroidal parathyroid carcinoma presenting with only hypercalcemia. Otolaryngol Head Neck Surg. 2002;127(4):352–3. https://doi.org/10.1067/mhn.2002.128553.

    Article  PubMed  Google Scholar 

  14. Ernst M, Lippmann M, Fleige B. Primary hyperparathyroidism in intrathyroid parathyroid cancer. Zentralbl Chir. 1993;118(11):682–5 discussion 685-6.

    CAS  PubMed  Google Scholar 

  15. Kirstein LJ, Ghosh BC. Intrathyroid parathyroid carcinoma. J Surg Oncol. 2001;77(2):136–8. https://doi.org/10.1002/jso.1084.

    Article  CAS  PubMed  Google Scholar 

  16. Lee KM, Kim EJ, Choi WS, Park WS, Kim SW. Intrathyroidal parathyroid carcinoma mimicking a thyroid nodule in a MEN type 1 patient. J Clin Ultrasound. 2014;42(4):212–4. https://doi.org/10.1002/jcu.22090.

    Article  PubMed  Google Scholar 

  17. Balakrishnan M, George SA, Rajab SH, Francis IM, Kapila K. Cytological challenges in the diagnosis of intrathyroidal parathyroid carcinoma: a case report and review of literature. Diagn Cytopathol. 2018;46(1):47–52. https://doi.org/10.1002/dc.23847.

    Article  PubMed  Google Scholar 

  18. Alharbi N, Asa SL, Szybowska M, Kim RH, Ezzat S. Intrathyroidal parathyroid carcinoma: an atypical thyroid lesion. Front Endocrinol. 2018;9:641. https://doi.org/10.3389/fendo.2018.00641.

    Article  Google Scholar 

  19. Quartey B, Shriver C, Russell D. Intrathyroidal parathyroid carcinoma presenting as asymptomatic high Normal serum calcium and slightly elevated intact parathyroid hormone: a case report and review of literature. World J Oncol. 2011;2(3):138–42. https://doi.org/10.4021/wjon311w.

    Article  PubMed  PubMed Central  Google Scholar 

  20. Wong YP, Sharifah NA, Taan GC, Gill AJ, Ali SZ. Intrathyroidal oxyphilic parathyroid carcinoma: a potential diagnostic caveat in cytology? Diagn Cytopathol. 2016;44(8):688–92. https://doi.org/10.1002/dc.23493.

    Article  PubMed  Google Scholar 

  21. Leitha T, Staudenherz A. Concomitant hyperparathyroidism and nonmedullary thyroid cancer, with a review of the literature. Clin Nucl Med. 2003;28(2):113–7. https://doi.org/10.1097/01.RLU.0000048680.30820.52.

    Article  PubMed  Google Scholar 

  22. Ghorra C, Rizk H, Abi Hachem R, Tannoury J, Abboud B. Association of pathyroid pathology with well- differentiated thyroid carcinoma. Presse Med. 2012;41(6):e265–71. https://doi.org/10.1016/j.lpm.2011.12.016.

    Article  PubMed  Google Scholar 

  23. Campennì A, Giovinazo S, Pignata SA, Di Mauro F, Santoro D, Curtò L, et al. Association of parathyroid carcinoma and thyroid disorders: a clinical review. Endrocine. 2017;56:19–26.

    Google Scholar 

  24. Lin SD, Tu ST, Hsu SR, Chang JH, Yang KT, Yang LH. Synchronous parathyroid and papillary thyroid carcinoma. J Chin Med Assoc. 2005;68(2):87–91. https://doi.org/10.1016/S1726-4901(09)70141-8.

    Article  PubMed  Google Scholar 

  25. Goldfarb M, O'Neal P, Shih JL, Hartzband P, Connolly J, Hasselgren PO. Synchronous parathyroid carcinoma, parathyroid adenoma, and papillary thyroid carcinoma in a patient with severe and long-standing hyperparathyroidism. Endocr Pract. 2009;15(5):463–8. https://doi.org/10.4158/EP09075.CRR.

    Article  PubMed  PubMed Central  Google Scholar 

  26. Chaychi L, Belbruno K, Golding A, Memoli V. Unusual manifestation of parathyroid carcinoma in the setting of papillary thyroid cancer. Endocr Pract. 2010;16(4):664–8. https://doi.org/10.4158/EP10061.CR.

    Article  PubMed  Google Scholar 

  27. Kurita S, Mihashi S, Hirano M, Nakashima T, Tanimura A. Hyperfunctioning parathyroid carcinoma combined with papillary carcinoma of the thyroid gland--report of a case (author's transl). Nihon Gan Chiryo Gakkai Shi. 1979;14(7):1127–35.

    CAS  PubMed  Google Scholar 

  28. Savli H, Sevinc A, Sari R, Ozen S, Buyukberber S, Ertas E. Occult parathyroid carcinoma in a patient with papillary thyroid carcinoma and Hashimoto's thyroiditis. J Endocrinol Investig. 2001;24(1):42–4. https://doi.org/10.1007/BF03343807.

    Article  CAS  Google Scholar 

  29. Christmas TJ, Chapple CR, Noble JG, Milroy EJ, Cowie AG. Hyperparathyroidism after neck irradiation. Br J Surg. 1988;75:873–4.

    Article  CAS  Google Scholar 

  30. Schoretsanitis G, Melissas J, Kafousi M. Synchronous parathyroid and papillary thyroid carcinoma: a case report. Am J Otolaryngol. 2002;23(6):382–5. https://doi.org/10.1053/ajot.2002.126317.

    Article  PubMed  Google Scholar 

  31. Marcy PY, Thariat J, Sudaka A, Poissonnet G. Synchronous parathyroid and papillary thyroid carcinomas. Thyroid. 2009;19(10):1131–3. https://doi.org/10.1089/thy.2009.0016.

    Article  PubMed  Google Scholar 

  32. Zakerkish M, Rajaei E, Dargahi M, Bahadoram M. A Rare Constellation of Hürthle cell Thyroid Carcinoma and Parathyroid Carcinoma. J Clin Diagn Res. 2015;9(12):OD08–10.

    PubMed  PubMed Central  Google Scholar 

  33. Dikmen K, Bostanci H, Gobut H, Yildiz A, Ertunc O, Celik A, et al. Nonfunctional double parathyroid carcinoma with incidental thyroid micropapillary carcinoma : a rare case. Pan Afr Med J. 2017;27:241.

    Article  Google Scholar 

  34. Baek C-O, Kim KH, Song SK. Synchronous parathyroid carcinoma and papillary thyroid carcinoma in a patient with long-standing schizophrenia. Korean J Intern Med. 2017;32(6):1104–7. https://doi.org/10.3904/kjim.2015.072.

    Article  PubMed  PubMed Central  Google Scholar 

  35. Norman J, Politz D, Browarsky I. Diagnostic aspiration of parathyroid adenomas causes severe fibrosis complicating surgery and final histologic diagnosis. Thyroid. 2007;17(12):1251–5. https://doi.org/10.1089/thy.2007.0081.

    Article  PubMed  Google Scholar 

  36. Sharretts JM, Simonds WF. Clinical and molecular genetics of parathyroid neoplasms. Best Pract Res Clin Endocrinol Metab. 2010;24(3):491–502. https://doi.org/10.1016/j.beem.2010.01.003.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  37. Zhao L, Sun LH, Liu DM, He XY, Tao B, Ning G, et al. Copy number variation in CCND1 gene is implicated in the pathogenesis of sporadic parathyroid carcinoma. World J Surg. 2014;38(7):1730–7. https://doi.org/10.1007/s00268-014-2455-9.

    Article  PubMed  Google Scholar 

  38. Salcuni AS, Cetani F, Guarnieri V, Nicastro V, Romagnoli E, De Martino D, et al. Parathyroid carcinoma. Best Pract Res Clin Endocrinol Metab. 2018;32(6):877–89. https://doi.org/10.1016/j.beem.2018.11.002.

    Article  CAS  PubMed  Google Scholar 

  39. Harari A, Waring A, Fernandez-Ranvier G, Hwang J, Suh I, Mitmaker E, et al. Parathyroid carcinoma: a 43-year outcome and survival analysis. J Clin Endocrinol Metab. 2011;96(12):3679–86. https://doi.org/10.1210/jc.2011-1571.

    Article  CAS  PubMed  Google Scholar 

  40. Busaidy NL, Jimenez C, Habra MA, Schultz PN, El-Naggar AK, Clayman GL, et al. Parathyroid carcinoma: a 22-year experience. Head Neck. 2004;26(8):716–26. https://doi.org/10.1002/hed.20049.

    Article  PubMed  Google Scholar 

  41. Shantz A, Castleman B. Parathyroid carcinoma: a study of 70 cases. Cancer. 1973;31(3):600–5. https://doi.org/10.1002/1097-0142(197303)31:3<600::AID-CNCR2820310316>3.0.CO;2-0.

    Article  Google Scholar 

  42. Wynne AG, van Heerden J, Carney JA, Fitzpatrick LA. Parathyroid carcinoma: clinical and pathologic features in 43 patients. Medicine. 1992;71(4):197–205. https://doi.org/10.1097/00005792-199207000-00002.

    Article  CAS  PubMed  Google Scholar 

  43. Talat N, Schulte KM. Clinical presentation, staging and long-term evolution of parathyroid cancer. Ann Surg Oncol. 2010;17(8):2156–74. https://doi.org/10.1245/s10434-010-1003-6.

    Article  PubMed  Google Scholar 

  44. Ferraro V, Sgaramella LI, Di Meo G, Prete FP, Logoluso F, Minerva F, et al. Current concepts in parathyroid carcinoma: a single Centre experience. BMC Endocr Disord. 2019;19(Suppl 1):46. https://doi.org/10.1186/s12902-019-0368-1.

    Article  PubMed  PubMed Central  Google Scholar 

  45. Kebebew E, Arici C, Duh QY, Clark OH. Localization and reoperation results for persistent and recurrent parathyroid carcinoma. Arch Surg. 2001;136(8):878–85. https://doi.org/10.1001/archsurg.136.8.878.

    Article  CAS  PubMed  Google Scholar 

  46. Goswamy J, Lei M, Simo R. Parathyroid carcinoma. Curro Opin Otolaryngol Head Neck Surg. 2016;24(2):155–62. https://doi.org/10.1097/MOO.0000000000000234.

    Article  Google Scholar 

  47. Limberg J, Stefanova D, Ullman TM, Thiesmeyer JW, Bains S, Beninato T, et al. The use and benefit of adjuvant radiotherapy in parathyroid carcinoma: a National Cancer Database Analysis. Ann Surg Oncol. 2021;28(1):502–11. https://doi.org/10.1245/s10434-020-08825-8.

    Article  PubMed  Google Scholar 

  48. Calandra DB, Chejfec G, Foy BK, Lawrence AM, Paloyan E. Parathyroid carcinoma: biochemical and pathologic response to DTIC. Surgery. 1984;96(6):1132–7.

    CAS  PubMed  Google Scholar 

  49. Munson ND, Foote RL, Northcutt RC, Tiegs RD, Fitzpatrick LA, Grant CS, et al. Parathyroid carcinoma: is there a role for adjuvant radiation therapy? Cancer. 2003;98(11):2378–84. https://doi.org/10.1002/cncr.11819.

    Article  PubMed  Google Scholar 

  50. Tochio M, Takaki H, Yamakado K, Uraki J, Kashima M, Nakatsuka A, et al. A case report of 20 lung radiofrequency ablation sessions for 50 lung metastases from parathyroid carcinoma causing hyperparathyroidism. Cardiovasc Intervent Radiol. 2010;33(3):657–9. https://doi.org/10.1007/s00270-009-9730-4.

    Article  PubMed  Google Scholar 

  51. Denney AM, Watts NB. The effect of octreotide on parathyroid carcinoma. J Clin Endocrinol Metab. 2004;89(2):1016. https://doi.org/10.1210/jc.2003-031825.

    Article  CAS  PubMed  Google Scholar 

  52. Bukowski RM, Sheeler L, Cunningham J, Esselstyn C. Successful combination chemotherapy for metastatic parathyroid carcinoma. Arch Intern Med. 1984;144(2):399–400. https://doi.org/10.1001/archinte.1984.00350140229032.

    Article  CAS  PubMed  Google Scholar 

  53. Warrell RP Jr, Issacs M, Alcock NW, Bockman RS. Gallium nitrate for treatment of refractory hypercalcemia from parathyroid carcinoma. Ann Intern Med. 1987;107(5):683–6. https://doi.org/10.7326/0003-4819-107-5-683.

    Article  PubMed  Google Scholar 

  54. Silverberg SJ, Rubin MR, Faiman C, Peacock M, Shoback DM, Smallridge RC, et al. Cinacalcet hydrochloride reduces the serum calcium concentration in inoperable parathyroid carcinoma. J Clin Endocrinol Metab. 2007;92(10):3803–8. https://doi.org/10.1210/jc.2007-0585.

    Article  CAS  PubMed  Google Scholar 

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This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

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Authors and Affiliations

  1. General Surgery Unit, Department of Advanced Medical and Surgical Sciences, University of Campania “Luigi Vanvitelli”, Piazza Miraglia, 80138, Naples, Italy

    Nadia De Falco, Giuseppe Santangelo, Angelo Cangiano, Maria Giulia Sommella, Angelo Cosenza, Gianluca Pellino, Domenico Parmeggiani, Silvestro Canonico & Massimo De Falco

  2. Maxillofacial Surgery Unit, Federico II University, Naples, Italy

    Fabrizio Chirico

  3. Division of Morphopathology, Department of Advanced Medical and Surgical Sciences, University of Campania “Luigi Vanvitelli”, Naples, Italy

    Andrea Ronchi & Marina Accardo

Authors
  1. Nadia De Falco
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  2. Giuseppe Santangelo
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  12. Massimo De Falco
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Contributions

All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by MDF, GS, MA, AR, GP, NDF and FC. AC, MGS, DP e SC participated substantially in the analysis and interpretation of the data. All authors read and approved the final manuscript.

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Correspondence to Massimo De Falco.

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De Falco, N., Santangelo, G., Chirico, F. et al. Synchronous intrathyroidal parathyroid carcinoma and thyroid carcinoma: case report and review of the literature. BMC Endocr Disord 21, 60 (2021). https://doi.org/10.1186/s12902-021-00724-7

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Keywords

BMC Endocrine Disorders

ISSN: 1472-6823

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