Endocr Pathol (2015) 26:33–36 DOI 10.1007/s12022-014-9351-6
An Extraordinary Case of Mesenchymal Chondrosarcoma Metastasis in the Thyroid Santiago Ortiz & Francisco Tortosa & Manuel Sobrinho Simões
Published online: 16 December 2014 # Springer Science+Business Media New York 2014
Abstract Secondary tumours of the thyroid gland account for 1.25 to 3 % in clinical series and reach 24 % in autopsy series. Chondrosarcoma is a rare malignant mesenchymal tumour of chondrogenic nature; the mesenchymal variant represents less than 3 % of all chondrosarcomas, being therefore extremely rare. A mesenchymal chondrosarcoma metastasis in the thyroid is exceptional; to our knowledge, only three previous cases of chondrosarcoma metastasis in the thyroid have been reported to date but none of such cases corresponded to a mesenchymal chondrosarcoma. We present the first of such a case in a 27-year-old woman with a 4-year history of mesenchymal chondrosarcoma of the sacrum that was treated by surgery and chemotherapy. At the present admission, head and neck computed tomography revealed a well-defined nodule in the thyroid gland. The diagnosis of metastasis from the mesenchymal chondrosarcoma was made in the right lobectomy specimen. Keywords Mesenchymal chondrosarcoma . Metastasis . Thyroid . Tumour . Sarcoma
Introduction Secondary tumours of the thyroid gland account for 1.25 to 3 % in clinical series and reach 24 % in autopsy series [1]. Nakhjavani et al. reported that in clinical series, the most common primary sites of tumours metastasizing to the thyroid were the kidney, colon, lung, breast and skin (melanoma) [2]. S. Ortiz (*) : F. Tortosa Centro Hospitalar Lisboa Norte, EPE—Hospital de Santa Maria, Lisbon, Portugal e-mail: [email protected] M. Sobrinho Simões Ipatimup, Porto, Portugal
Only 1 to 12 % of all chondrosarcomas occur in the head and neck region representing 0.1 % of the neoplasms of this region [3]. Thyroid metastasis arising from sarcomas is extremely unusual [4]. To our knowledge, only three previous cases of chondrosarcoma metastasis in the thyroid have been reported in the world medical literature [5–7]. We report the first case of a metastasis in the thyroid from a mesenchymal chondrosarcoma.
Materials and Methods A 27-year-old woman was admitted in 2013 for an enlargement of the thyroid gland, without pain or compression signs. A computed tomography (CT) scan showed a well-defined, heterogeneous and bulky tumour of the right thyroid lobe, with a diameter of about 4 cm. Fine needle aspiration cytology revealed atypical cells and a subsequent core biopsy disclosed a lesion compatible with a metastasis from the mesenchymal chondrosarcoma. A right lobectomy was performed. In 2009, when the patient was 23, she had been admitted for lumbosacral pain, radiating to the left lower limb. Magnetic resonance revealed an expansive lesion of the sacrum to the S1 and S2 level with cortical destruction and extra-osseous penetration into the soft tissue. A complete CT scan of the all body did not disclose any distant metastasis. A CT-guided biopsy showed a mesenchymal chondrosarcoma. In 2010, the patient was submitted to total resection of S1 to S4 with reconstruction of the pelvis and the diagnosis of mesenchymal chondrosarcoma was confirmed. After surgery, she underwent chemotherapy.
Results The surgical specimen consisted of a right lobectomy weighing 42.6 g and 6.4×3.4×4.5 cm. At the cut surface, it exhibited a 4×3.3 cm, apparently encapsulated yellowish-
34
Endocr Pathol (2015) 26:33–36
grey nodule, displaying a firm consistency (Fig. 1). Microscopically, the sections revealed a neoplastic proliferation composed by round cells. Amongst the cellular proliferation, areas of mature hyaline cartilage are recognized (Fig. 2). We made the diagnosis of a metastasis in the thyroid of the mesenchymal chondrosarcoma diagnosed 4 years before. At present, the patient has evidence of another metastasis in the right humerus.
Discussion Metastasis in the thyroid gland account from 1.25 to 3 % in clinical series and reach 24 % in autopsy series [1]. Based on recent literature, the most frequent primary sites of tumours metastasizing to the thyroid are kidney (48.1 %), colorectal (10.4 %), lung (8.3 %), breast (7.8 %), skin (5 %) and sarcoma (4 %) [8]. Metastasis to the thyroid are more common in women than men (female to male ratio=1.4 to 1). A palpable thyroid nodule is discovered in 72 % of patients with metastasis in the gland [9]. The principal symptom is pain. Although the thyroid is richly vascularised and the primary pathway of sarcoma metastasis is hematogenous, the frequency of intrathyroid metastasis from sarcomas is very low [5]. In Table 1, we have listed the cases of sarcomas metastasising in the thyroid reported in the English literature. We found a total of 33 cases: leiomyosarcoma (n=12), malignant fibrous histiocytoma (n=6), liposarcoma (n=4), chondrosarcoma (n=3), endometrial sarcoma (n=2), angiosarcoma (n=2), artery intimal sarcoma (n=1), dermatofibrosarcoma protuberans (n=1), rhabdomyosarcoma (n=1) and osteosarcoma (n=1). Other characteristics of these cases are summarized in Table 1. Chondrosarcoma is a rare malignant mesenchymal tumour of chondrogenic nature; the mesenchymal variant represents
Fig. 1 The nodule is well circumscribed and shows a focal cartilage appearance (arrow)
Fig. 2 Histological examination. There is a clear separation between the normal thyroid tissue and the metastasis from the mesenchymal chondrosarcoma (a H&E ×100). The typical biphasic pattern is composed of undifferentiated small round cells admixed with islands of well-differentiated hyaline cartilage (b H&E ×200)
less than 3 % of all chondrosarcomas, being therefore an exceedingly rare soft tissue sarcoma [10]. Mesenchymal chondrosarcoma tends to develop mostly in the skeleton. Extraskeletal mesenchymal chondrosarcoma usually occurs in younger individuals, especially during the second and third decade of life, and has a female predominance [11]. Primary chondrosarcomas of the head and neck may involve the sinonasal region, jaws, larynx or skull base [12, 13]. Sarcoma as a primary cancer of the thyroid gland is extremely rare [14]. Histologically, there are conventional chondrosarcoma (90 % of all cases), and other variants which include dedifferentiated, clear cell, myxoid and mesenchymal chondrosarcomas [5]. Mesenchymal chondrosarcoma, like in our case, is characterized by a biphasic pattern that is composed by undifferentiated, small, monomorphic, basophilic, atypical mesenchymal cells and nodules or islands of welldifferentiated hyaline cartilaginous tissue. Recently, a HEY1NCOA2 fusion was identified in all mesenchymal chondrosarcoma, being absent in other subtypes [8]. It is a fusion of exon 4 to exon 13 on chromosome 8 [15]. In this case, we did not perform cytogenetic analysis because the patient already had a diagnosis of mesenchymal chondrosarcoma of the sacrum and the histopathological characteristics of the thyroid lesion were fairly typical. Mesenchymal chondrosarcoma is a highly aggressive sarcoma. The optimal treatment for chondrosarcoma is complete surgical resection with free margins. Incomplete resections are complicated by high rates of local recurrence and an increased risk of metastatic disease [16]. Radiation therapy and chemotherapy do not appear to have a significant effect on survival and they should be used for palliative purposes only [17].
Endocr Pathol (2015) 26:33–36 Table 1 Sarcomas that have metastasized to the in thyroid [1, 6, 7, 18–43]
35
Sex
Age (years)
Diagnosis
Primary tumour
Male Female Not known Not known Not known
68 30 Not known Not known Not known
Malignant fibrous histiocytoma Malignant fibrous histiocytoma Malignant fibrous histiocytoma Malignant fibrous histiocytoma Malignant fibrous histiocytoma
Arm Ankle Thigh Not known Not known
Not known Female Female Female Female Female Female Female Female Female Female Not known Not known Male Female Female Female Female
Not known 52 59 54 54 59 50 Not known Not known 63 55 Not known Not known 66 69 Not known 62 63
Malignant fibrous histiocytoma Dermatofibrosarcoma protuberans Endometrial sarcoma Endometrial carcinosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Pulmonary artery leiomyosarcoma Pulmonary artery intimal sarcoma Epithelioid angiosarcoma
Not known Abdominal wall Uterus Uterus Uterus Uterus Uterus Uterus Uterus Leg Lung Not known Not known Leg Duodenum Lung Lung Scalp
Male Female Male Female Female Not known Female Not known Not known Not known
Not known 22 51 86 30 Not known 51 66 Not known Not known
Angiosarcoma Rhabdomyosarcoma Myxoid liposarcoma Myxoid liposarcoma Pleomorphic liposarcoma Pleomorphic liposarcoma Chondrosarcoma Chondrosarcoma Osteosarcoma Chondrosarcoma
Pyriform fossa Leg Thigh Thigh Not known Not known Tibia Femur Not known Not known
The treatment of metastasis in the thyroid gland is controversial because the prognosis is poor. Thyroidectomy may not be effective in prolonging survival time; however, it may help preserve the quality of life in case of extension to surrounding tissues [6, 14]. There is no clear consensus for therapy. Five and 10-year survival rates of patients are approximately 50 and 25 %, respectively [9]. There is no correlation of prognosis with the histological features [8]. Presently, the patient has another metastasis of the mesenchymal chondrosarcoma in the right humerus. Summing up, this case report is interesting because it is the first case of metastatic mesenchymal chondrosarcoma on record. It is important to keep in mind that the thyroid in not unfrequently a site of metastatic deposits. In a patient with a
previous history of chondrosarcoma, metastasis should be considered in the differential diagnosis of the thyroid tumour.
References 1. Gerges AS, Shehata SR, Gouda IA (2006) Metastasis to the thyroid gland: unusual site of metastasis. J Egypt Natl Canc Inst 18:67–72. 2. DeLellis R (2004) Secondary tumours of the thyroid. In: DeLellis R, Lloyd R, Heitz P, Eng C (eds) Pathology and Genetics of Tumours of Endocrine Organs. IARC, pp 122–123. 3. Coca-Pelaz A, Rodrigo JP, Triantafyllou A, Hunt JL, FernándezMiranda JC, Strojan P, de Bree R, Rinaldo A, Takes RP, Ferlito A
36
4.
5.
6.
7.
8.
9. 10.
11.
12. 13.
14.
15.
16.
17.
18. 19.
20.
21.
22.
Endocr Pathol (2015) 26:33–36 (2013) Chondrosarcomas of the head and neck. Eur Arch Otorhinolaryngol 271:2601–2609. Giorgadze T, Ward RM, Baloch ZW, LiVolsi VA (2002) Phyllodes tumor metastatic to thyroid Hürthle cell adenoma. Arch Pathol Lab Med 126:1233–1236. Youssef D, Mohamed MT, Mehdi C, Karim N, Mostapha B, Haddou A, Brahim B (2014) Chondrosarcoma metastasis in the thyroid gland: a case report. Journal of Medical Case Reports 8:157. El Otmany A, Hafid H, Hamada H, El Hafed A, Souadka A, Amrani L, Jahid M, Belabbas M (2001) Métastase intrathyroïdienne d’un chondrosarcome. Médecine Du Maghreb 87:53–54. Bakx PA, van den Ingh HF, Baggen RG, Veen HF, Oosterhuis WJ (1993) An unusual metastasis of a chondrosarcoma in the thyroid gland. Eur J Surg 159:643–644. Chung AY, Tran TB, Brumund KT, Weisman RA, Bouvet M (2012) Metastases to the thyroid: a review of the literature from the last decade. Thyroid 22:258–268. Ishikawa M, Hirano S, Tsuji T, Ito J (2011) Management of metastasis to the thyroid gland. Auris Nasus Larynx 38:426–430. Nakashima Y, de Pinieux G, Ladanyi M (2013) Mesenchymal chondrosarcoma. In: Fletcher CDM, Bridge JA, Hogendoorn PCW, Mertens F (eds) WHO Classification of Tumours of Soft Tissue and Bone. IARC, pp 271–272. Oliveira AM (2013) Cartilaginous and Osseous Soft Tissue Tumors. In: Hornick JL (ed) Practical Soft Tissue Pathology A Diagnostic Approach. Elsevier, pp 380–381. Gupta P, Bhalla AS, Karthikeyan V, Bhutia O (2012) Two rare cases of craniofacial chondrosarcoma. World J Radiol 4:283–285. Prado FO, Nishimoto IN, Perez DE, Kowalski LP, Lopes MA (2009) Head and neck chondrosarcoma: analysis of 16 cases. Br J Oral Maxillofac Surg 47:555–557. Haeri H, Asadi Amoli F, Shariat Torbaghan S (1999) A case report of well differentiated chondrosarcoma of thyroid gland. Acta Medica Iranica 37:249–251. Wang L, Motoi T, Khanin R, et al. (2012) Identification of a novel, recurrent HEY1-NCOA2 fusion in mesenchymal chondrosarcoma based on a genome-wide screen of exon-level expression data. Genes Chromosomes Cancer 51:127–139. Gholamreza M, Ali H, Hossein A, Farzane H, Reza Basirat N, Nushin A, Mehdi E, Saleh J (2013) A rare case of laryngotracheal chondrosarcoma in a patient with past history of radioiodine therapy for thyroid cancer. Adv Biomed Res 2:74. Pontes HA, Pontes FS, de Abreu MC, de Carvalho PL, de Brito Kato AM, Fonseca FP, de Freitas Silva BS, Neto NC (2012) Clinicopathological analysis of head and neck chondrosarcoma: three case reports and literature review. Int J Oral Maxillofac Surg 41:203–210. Vankalakunti M, Kaur J, Srinivasan R (2008) Cytology of sarcoma metastasizing to the thyroid: a case report. Acta Cytol 52:729–32. Gattuso P, Castelli MJ, Reyes CV (1989) Fine needle aspiration cytology of metastatic sarcoma involving the thyroid. Southern Medical Journal 82:1158–60. Haugen BR, Nawaz S, Cohn A, Shroyer K, Bunn PA Jr, Liechty DR, Ridgway EC (1994) Secondary malignancy of the thyroid gland: a case report and review of the literature. Thyroid 4:297–300. Ackerman L, Romyn A, Khedkar N, Kaplan E (1987) Malignant fibrous histiocytoma metastatic to the thyroid gland. Clin Nucl Med 12:648–9. Akimov OV (1994) Malignant fibrous histiocytoma of the heart with metastases to the brain and thyroid gland. Arkh Patol 56:70–2.
23. Kreze A Jr, Zápotocká A, Urbanec T, Koskuba J, Pura M, Vítek P, Praženica P, Traboulsi E (2012) Metastasis of dermatofibrosarcoma from the abdominal wall to the thyroid gland: case report. Case Rep Med 2012:654–9. 24. Socrier Y, D’Aure D, Lacoste-Collin L, Escourrou G, SouleTholy M, Delisle MB, Courtade-Saïdi M (2013) Cytology of a thyroid metastasis from an endometrial sarcoma: a case report. Cytopathology 24:408–9. 25. Mete O, Ozbey N, Erbil Y, Salmasloglu A, Aral F, Temel B, Dizdaroglu F (2007) Thyroid metastasis of endometrial carcinosarcoma associated with Graves’ disease. Gynecol Endocrinol 23:562–6. 26. Giannikaki E, Mantadakis E, Mamalaki E, Delides G, Samonis G (2006) Metastatic uterine leiomyosarcoma coexisting with papillary carcinoma of the thyroid gland. Int J Gynecol Cancer 16:442–5. 27. Michelow PM, Leiman G (1995) Metastases to the thyroid gland: diagnosis by aspiration cytology. Diagn Cytopathol 13:209–13. 28. Leath CA 3rd, Huh WK, Straughn JM Jr, Conner MG (2002) Uterine leiomyosarcoma metastatic to the thyroid. Obstet Gynecol 100:1122–4. 29. Nemenqani D, Yaqoob N, Khoja H (2010) Leiomyosarcoma metastatic to the thyroid diagnosed by fine needle aspiration cytology. Journal of the Pakistan Medical Association 60:307–9. 30. Wang TY, Lee CH, Yang AH, P’eng FK, Lui WY (1998) Metastatic leiomyosarcoma of the thyroid: a case report. Zhonghua Yi Xue Za Zhi 61:353–7. 31. Woo Young K, Young Ran K, Sang Uk W, Jae Bok L (2011) Pulmonary leiomyosarcoma metastatic to the thyroid gland: case report and review of the literature. Ann Endocrinol 72:314–6. 32. Deng XR, Wang G, Kuang CJ, Peng GZ, Chen RS (2005) Metastasis of leiomyosarcoma to the thyroid. Chinese Medical Journal 118:174–6. 33. Bode-Lesniewska B, Schroder S, Gemsenjager E, Staubli M, Pfaltz M (1994) Leiomyosarcoma in the thyroid gland—primary or metastatic tumour?. Pathologe 15:303–7. 34. D’Andrea N, Romano V, Mattioli F, Candia S, Montanari A, Guidi L, Carvalho M, Sanguinetti CM (2003) Pulmonary artery leiomyosarcoma with thyroid metastases. Monaldi Arch Chest Dis 59:304–7. 35. Choi YM Jang EK, Ahn SH, Jeon MJ, Han JM, Kim SC, Han DJ, Gong G, Kim TY, Shong YK, Kim WB (2013) Long-term survival of a patient with pulmonary artery intimal sarcoma after sequential metastasectomies of the thyroid and adrenal glands. Endocrinol Metab 28:46–9. 36. Fulciniti F, Di Mattia D, Bove P, Mastro AA, De Chiara A, Botti G, Petrillo A, Apice G (2008) Fine needle aspiration of metastatic epithelioid angiosarcoma: a report of 2 cases. Acta Cytol 52:612–8. 37. Eng SP, Goh CH, Khoo JB, Yang TL, Lim LH (2005) Metastatic angiosarcoma to the thyroid. Rev Laryngol Otol Rhinol 126:111–4. 38. Hafez MT, Hegazy MA, Abd Elwahab K, Arafa M, Abdou I, Refky B (2012) Metastatic rhabdomyosarcoma of the thyroid gland, a case report. Head Neck Oncol 4:27. 39. Tysome JR, Sandison A, Clarke PM. (2006) Myxoid liposarcoma metastatic to the thyroid gland: a case report and literature review. J Laringol Otol 120:511–3. 40. Brandwein-Gensler M, Urken M, Wang B (2004) Collision tumor of the thyroid: a case report of metastatic liposarcoma plus papillary thyroid carcinoma. Head Neck 26:637–41. 41. Azar AR, Weynand B, Daumerie C, Coche E (2003) Metastatic liposarcoma of the thyroid gland. Br J Radiol 76:750–2. 42. Bashir H, Nawaz MK, Shah MA, Ahmad E (2002) Pleomorphic liposarcoma metastatic to the thyroid gland. Clin Nucl Med 27:9–10. 43. Lam KY, Lo CY (1998) Metastatic tumors of the thyroid gland: a study of 79 cases in Chinese patients. Arch Pathol Lab Med 122:37–41.
An Extraordinary Case of Mesenchymal Chondrosarcoma Metastasis in the Thyroid Santiago Ortiz & Francisco Tortosa & Manuel Sobrinho Simões
Published online: 16 December 2014 # Springer Science+Business Media New York 2014
Abstract Secondary tumours of the thyroid gland account for 1.25 to 3 % in clinical series and reach 24 % in autopsy series. Chondrosarcoma is a rare malignant mesenchymal tumour of chondrogenic nature; the mesenchymal variant represents less than 3 % of all chondrosarcomas, being therefore extremely rare. A mesenchymal chondrosarcoma metastasis in the thyroid is exceptional; to our knowledge, only three previous cases of chondrosarcoma metastasis in the thyroid have been reported to date but none of such cases corresponded to a mesenchymal chondrosarcoma. We present the first of such a case in a 27-year-old woman with a 4-year history of mesenchymal chondrosarcoma of the sacrum that was treated by surgery and chemotherapy. At the present admission, head and neck computed tomography revealed a well-defined nodule in the thyroid gland. The diagnosis of metastasis from the mesenchymal chondrosarcoma was made in the right lobectomy specimen. Keywords Mesenchymal chondrosarcoma . Metastasis . Thyroid . Tumour . Sarcoma
Introduction Secondary tumours of the thyroid gland account for 1.25 to 3 % in clinical series and reach 24 % in autopsy series [1]. Nakhjavani et al. reported that in clinical series, the most common primary sites of tumours metastasizing to the thyroid were the kidney, colon, lung, breast and skin (melanoma) [2]. S. Ortiz (*) : F. Tortosa Centro Hospitalar Lisboa Norte, EPE—Hospital de Santa Maria, Lisbon, Portugal e-mail: [email protected] M. Sobrinho Simões Ipatimup, Porto, Portugal
Only 1 to 12 % of all chondrosarcomas occur in the head and neck region representing 0.1 % of the neoplasms of this region [3]. Thyroid metastasis arising from sarcomas is extremely unusual [4]. To our knowledge, only three previous cases of chondrosarcoma metastasis in the thyroid have been reported in the world medical literature [5–7]. We report the first case of a metastasis in the thyroid from a mesenchymal chondrosarcoma.
Materials and Methods A 27-year-old woman was admitted in 2013 for an enlargement of the thyroid gland, without pain or compression signs. A computed tomography (CT) scan showed a well-defined, heterogeneous and bulky tumour of the right thyroid lobe, with a diameter of about 4 cm. Fine needle aspiration cytology revealed atypical cells and a subsequent core biopsy disclosed a lesion compatible with a metastasis from the mesenchymal chondrosarcoma. A right lobectomy was performed. In 2009, when the patient was 23, she had been admitted for lumbosacral pain, radiating to the left lower limb. Magnetic resonance revealed an expansive lesion of the sacrum to the S1 and S2 level with cortical destruction and extra-osseous penetration into the soft tissue. A complete CT scan of the all body did not disclose any distant metastasis. A CT-guided biopsy showed a mesenchymal chondrosarcoma. In 2010, the patient was submitted to total resection of S1 to S4 with reconstruction of the pelvis and the diagnosis of mesenchymal chondrosarcoma was confirmed. After surgery, she underwent chemotherapy.
Results The surgical specimen consisted of a right lobectomy weighing 42.6 g and 6.4×3.4×4.5 cm. At the cut surface, it exhibited a 4×3.3 cm, apparently encapsulated yellowish-
34
Endocr Pathol (2015) 26:33–36
grey nodule, displaying a firm consistency (Fig. 1). Microscopically, the sections revealed a neoplastic proliferation composed by round cells. Amongst the cellular proliferation, areas of mature hyaline cartilage are recognized (Fig. 2). We made the diagnosis of a metastasis in the thyroid of the mesenchymal chondrosarcoma diagnosed 4 years before. At present, the patient has evidence of another metastasis in the right humerus.
Discussion Metastasis in the thyroid gland account from 1.25 to 3 % in clinical series and reach 24 % in autopsy series [1]. Based on recent literature, the most frequent primary sites of tumours metastasizing to the thyroid are kidney (48.1 %), colorectal (10.4 %), lung (8.3 %), breast (7.8 %), skin (5 %) and sarcoma (4 %) [8]. Metastasis to the thyroid are more common in women than men (female to male ratio=1.4 to 1). A palpable thyroid nodule is discovered in 72 % of patients with metastasis in the gland [9]. The principal symptom is pain. Although the thyroid is richly vascularised and the primary pathway of sarcoma metastasis is hematogenous, the frequency of intrathyroid metastasis from sarcomas is very low [5]. In Table 1, we have listed the cases of sarcomas metastasising in the thyroid reported in the English literature. We found a total of 33 cases: leiomyosarcoma (n=12), malignant fibrous histiocytoma (n=6), liposarcoma (n=4), chondrosarcoma (n=3), endometrial sarcoma (n=2), angiosarcoma (n=2), artery intimal sarcoma (n=1), dermatofibrosarcoma protuberans (n=1), rhabdomyosarcoma (n=1) and osteosarcoma (n=1). Other characteristics of these cases are summarized in Table 1. Chondrosarcoma is a rare malignant mesenchymal tumour of chondrogenic nature; the mesenchymal variant represents
Fig. 1 The nodule is well circumscribed and shows a focal cartilage appearance (arrow)
Fig. 2 Histological examination. There is a clear separation between the normal thyroid tissue and the metastasis from the mesenchymal chondrosarcoma (a H&E ×100). The typical biphasic pattern is composed of undifferentiated small round cells admixed with islands of well-differentiated hyaline cartilage (b H&E ×200)
less than 3 % of all chondrosarcomas, being therefore an exceedingly rare soft tissue sarcoma [10]. Mesenchymal chondrosarcoma tends to develop mostly in the skeleton. Extraskeletal mesenchymal chondrosarcoma usually occurs in younger individuals, especially during the second and third decade of life, and has a female predominance [11]. Primary chondrosarcomas of the head and neck may involve the sinonasal region, jaws, larynx or skull base [12, 13]. Sarcoma as a primary cancer of the thyroid gland is extremely rare [14]. Histologically, there are conventional chondrosarcoma (90 % of all cases), and other variants which include dedifferentiated, clear cell, myxoid and mesenchymal chondrosarcomas [5]. Mesenchymal chondrosarcoma, like in our case, is characterized by a biphasic pattern that is composed by undifferentiated, small, monomorphic, basophilic, atypical mesenchymal cells and nodules or islands of welldifferentiated hyaline cartilaginous tissue. Recently, a HEY1NCOA2 fusion was identified in all mesenchymal chondrosarcoma, being absent in other subtypes [8]. It is a fusion of exon 4 to exon 13 on chromosome 8 [15]. In this case, we did not perform cytogenetic analysis because the patient already had a diagnosis of mesenchymal chondrosarcoma of the sacrum and the histopathological characteristics of the thyroid lesion were fairly typical. Mesenchymal chondrosarcoma is a highly aggressive sarcoma. The optimal treatment for chondrosarcoma is complete surgical resection with free margins. Incomplete resections are complicated by high rates of local recurrence and an increased risk of metastatic disease [16]. Radiation therapy and chemotherapy do not appear to have a significant effect on survival and they should be used for palliative purposes only [17].
Endocr Pathol (2015) 26:33–36 Table 1 Sarcomas that have metastasized to the in thyroid [1, 6, 7, 18–43]
35
Sex
Age (years)
Diagnosis
Primary tumour
Male Female Not known Not known Not known
68 30 Not known Not known Not known
Malignant fibrous histiocytoma Malignant fibrous histiocytoma Malignant fibrous histiocytoma Malignant fibrous histiocytoma Malignant fibrous histiocytoma
Arm Ankle Thigh Not known Not known
Not known Female Female Female Female Female Female Female Female Female Female Not known Not known Male Female Female Female Female
Not known 52 59 54 54 59 50 Not known Not known 63 55 Not known Not known 66 69 Not known 62 63
Malignant fibrous histiocytoma Dermatofibrosarcoma protuberans Endometrial sarcoma Endometrial carcinosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Leiomyosarcoma Pulmonary artery leiomyosarcoma Pulmonary artery intimal sarcoma Epithelioid angiosarcoma
Not known Abdominal wall Uterus Uterus Uterus Uterus Uterus Uterus Uterus Leg Lung Not known Not known Leg Duodenum Lung Lung Scalp
Male Female Male Female Female Not known Female Not known Not known Not known
Not known 22 51 86 30 Not known 51 66 Not known Not known
Angiosarcoma Rhabdomyosarcoma Myxoid liposarcoma Myxoid liposarcoma Pleomorphic liposarcoma Pleomorphic liposarcoma Chondrosarcoma Chondrosarcoma Osteosarcoma Chondrosarcoma
Pyriform fossa Leg Thigh Thigh Not known Not known Tibia Femur Not known Not known
The treatment of metastasis in the thyroid gland is controversial because the prognosis is poor. Thyroidectomy may not be effective in prolonging survival time; however, it may help preserve the quality of life in case of extension to surrounding tissues [6, 14]. There is no clear consensus for therapy. Five and 10-year survival rates of patients are approximately 50 and 25 %, respectively [9]. There is no correlation of prognosis with the histological features [8]. Presently, the patient has another metastasis of the mesenchymal chondrosarcoma in the right humerus. Summing up, this case report is interesting because it is the first case of metastatic mesenchymal chondrosarcoma on record. It is important to keep in mind that the thyroid in not unfrequently a site of metastatic deposits. In a patient with a
previous history of chondrosarcoma, metastasis should be considered in the differential diagnosis of the thyroid tumour.
References 1. Gerges AS, Shehata SR, Gouda IA (2006) Metastasis to the thyroid gland: unusual site of metastasis. J Egypt Natl Canc Inst 18:67–72. 2. DeLellis R (2004) Secondary tumours of the thyroid. In: DeLellis R, Lloyd R, Heitz P, Eng C (eds) Pathology and Genetics of Tumours of Endocrine Organs. IARC, pp 122–123. 3. Coca-Pelaz A, Rodrigo JP, Triantafyllou A, Hunt JL, FernándezMiranda JC, Strojan P, de Bree R, Rinaldo A, Takes RP, Ferlito A
36
4.
5.
6.
7.
8.
9. 10.
11.
12. 13.
14.
15.
16.
17.
18. 19.
20.
21.
22.
Endocr Pathol (2015) 26:33–36 (2013) Chondrosarcomas of the head and neck. Eur Arch Otorhinolaryngol 271:2601–2609. Giorgadze T, Ward RM, Baloch ZW, LiVolsi VA (2002) Phyllodes tumor metastatic to thyroid Hürthle cell adenoma. Arch Pathol Lab Med 126:1233–1236. Youssef D, Mohamed MT, Mehdi C, Karim N, Mostapha B, Haddou A, Brahim B (2014) Chondrosarcoma metastasis in the thyroid gland: a case report. Journal of Medical Case Reports 8:157. El Otmany A, Hafid H, Hamada H, El Hafed A, Souadka A, Amrani L, Jahid M, Belabbas M (2001) Métastase intrathyroïdienne d’un chondrosarcome. Médecine Du Maghreb 87:53–54. Bakx PA, van den Ingh HF, Baggen RG, Veen HF, Oosterhuis WJ (1993) An unusual metastasis of a chondrosarcoma in the thyroid gland. Eur J Surg 159:643–644. Chung AY, Tran TB, Brumund KT, Weisman RA, Bouvet M (2012) Metastases to the thyroid: a review of the literature from the last decade. Thyroid 22:258–268. Ishikawa M, Hirano S, Tsuji T, Ito J (2011) Management of metastasis to the thyroid gland. Auris Nasus Larynx 38:426–430. Nakashima Y, de Pinieux G, Ladanyi M (2013) Mesenchymal chondrosarcoma. In: Fletcher CDM, Bridge JA, Hogendoorn PCW, Mertens F (eds) WHO Classification of Tumours of Soft Tissue and Bone. IARC, pp 271–272. Oliveira AM (2013) Cartilaginous and Osseous Soft Tissue Tumors. In: Hornick JL (ed) Practical Soft Tissue Pathology A Diagnostic Approach. Elsevier, pp 380–381. Gupta P, Bhalla AS, Karthikeyan V, Bhutia O (2012) Two rare cases of craniofacial chondrosarcoma. World J Radiol 4:283–285. Prado FO, Nishimoto IN, Perez DE, Kowalski LP, Lopes MA (2009) Head and neck chondrosarcoma: analysis of 16 cases. Br J Oral Maxillofac Surg 47:555–557. Haeri H, Asadi Amoli F, Shariat Torbaghan S (1999) A case report of well differentiated chondrosarcoma of thyroid gland. Acta Medica Iranica 37:249–251. Wang L, Motoi T, Khanin R, et al. (2012) Identification of a novel, recurrent HEY1-NCOA2 fusion in mesenchymal chondrosarcoma based on a genome-wide screen of exon-level expression data. Genes Chromosomes Cancer 51:127–139. Gholamreza M, Ali H, Hossein A, Farzane H, Reza Basirat N, Nushin A, Mehdi E, Saleh J (2013) A rare case of laryngotracheal chondrosarcoma in a patient with past history of radioiodine therapy for thyroid cancer. Adv Biomed Res 2:74. Pontes HA, Pontes FS, de Abreu MC, de Carvalho PL, de Brito Kato AM, Fonseca FP, de Freitas Silva BS, Neto NC (2012) Clinicopathological analysis of head and neck chondrosarcoma: three case reports and literature review. Int J Oral Maxillofac Surg 41:203–210. Vankalakunti M, Kaur J, Srinivasan R (2008) Cytology of sarcoma metastasizing to the thyroid: a case report. Acta Cytol 52:729–32. Gattuso P, Castelli MJ, Reyes CV (1989) Fine needle aspiration cytology of metastatic sarcoma involving the thyroid. Southern Medical Journal 82:1158–60. Haugen BR, Nawaz S, Cohn A, Shroyer K, Bunn PA Jr, Liechty DR, Ridgway EC (1994) Secondary malignancy of the thyroid gland: a case report and review of the literature. Thyroid 4:297–300. Ackerman L, Romyn A, Khedkar N, Kaplan E (1987) Malignant fibrous histiocytoma metastatic to the thyroid gland. Clin Nucl Med 12:648–9. Akimov OV (1994) Malignant fibrous histiocytoma of the heart with metastases to the brain and thyroid gland. Arkh Patol 56:70–2.
23. Kreze A Jr, Zápotocká A, Urbanec T, Koskuba J, Pura M, Vítek P, Praženica P, Traboulsi E (2012) Metastasis of dermatofibrosarcoma from the abdominal wall to the thyroid gland: case report. Case Rep Med 2012:654–9. 24. Socrier Y, D’Aure D, Lacoste-Collin L, Escourrou G, SouleTholy M, Delisle MB, Courtade-Saïdi M (2013) Cytology of a thyroid metastasis from an endometrial sarcoma: a case report. Cytopathology 24:408–9. 25. Mete O, Ozbey N, Erbil Y, Salmasloglu A, Aral F, Temel B, Dizdaroglu F (2007) Thyroid metastasis of endometrial carcinosarcoma associated with Graves’ disease. Gynecol Endocrinol 23:562–6. 26. Giannikaki E, Mantadakis E, Mamalaki E, Delides G, Samonis G (2006) Metastatic uterine leiomyosarcoma coexisting with papillary carcinoma of the thyroid gland. Int J Gynecol Cancer 16:442–5. 27. Michelow PM, Leiman G (1995) Metastases to the thyroid gland: diagnosis by aspiration cytology. Diagn Cytopathol 13:209–13. 28. Leath CA 3rd, Huh WK, Straughn JM Jr, Conner MG (2002) Uterine leiomyosarcoma metastatic to the thyroid. Obstet Gynecol 100:1122–4. 29. Nemenqani D, Yaqoob N, Khoja H (2010) Leiomyosarcoma metastatic to the thyroid diagnosed by fine needle aspiration cytology. Journal of the Pakistan Medical Association 60:307–9. 30. Wang TY, Lee CH, Yang AH, P’eng FK, Lui WY (1998) Metastatic leiomyosarcoma of the thyroid: a case report. Zhonghua Yi Xue Za Zhi 61:353–7. 31. Woo Young K, Young Ran K, Sang Uk W, Jae Bok L (2011) Pulmonary leiomyosarcoma metastatic to the thyroid gland: case report and review of the literature. Ann Endocrinol 72:314–6. 32. Deng XR, Wang G, Kuang CJ, Peng GZ, Chen RS (2005) Metastasis of leiomyosarcoma to the thyroid. Chinese Medical Journal 118:174–6. 33. Bode-Lesniewska B, Schroder S, Gemsenjager E, Staubli M, Pfaltz M (1994) Leiomyosarcoma in the thyroid gland—primary or metastatic tumour?. Pathologe 15:303–7. 34. D’Andrea N, Romano V, Mattioli F, Candia S, Montanari A, Guidi L, Carvalho M, Sanguinetti CM (2003) Pulmonary artery leiomyosarcoma with thyroid metastases. Monaldi Arch Chest Dis 59:304–7. 35. Choi YM Jang EK, Ahn SH, Jeon MJ, Han JM, Kim SC, Han DJ, Gong G, Kim TY, Shong YK, Kim WB (2013) Long-term survival of a patient with pulmonary artery intimal sarcoma after sequential metastasectomies of the thyroid and adrenal glands. Endocrinol Metab 28:46–9. 36. Fulciniti F, Di Mattia D, Bove P, Mastro AA, De Chiara A, Botti G, Petrillo A, Apice G (2008) Fine needle aspiration of metastatic epithelioid angiosarcoma: a report of 2 cases. Acta Cytol 52:612–8. 37. Eng SP, Goh CH, Khoo JB, Yang TL, Lim LH (2005) Metastatic angiosarcoma to the thyroid. Rev Laryngol Otol Rhinol 126:111–4. 38. Hafez MT, Hegazy MA, Abd Elwahab K, Arafa M, Abdou I, Refky B (2012) Metastatic rhabdomyosarcoma of the thyroid gland, a case report. Head Neck Oncol 4:27. 39. Tysome JR, Sandison A, Clarke PM. (2006) Myxoid liposarcoma metastatic to the thyroid gland: a case report and literature review. J Laringol Otol 120:511–3. 40. Brandwein-Gensler M, Urken M, Wang B (2004) Collision tumor of the thyroid: a case report of metastatic liposarcoma plus papillary thyroid carcinoma. Head Neck 26:637–41. 41. Azar AR, Weynand B, Daumerie C, Coche E (2003) Metastatic liposarcoma of the thyroid gland. Br J Radiol 76:750–2. 42. Bashir H, Nawaz MK, Shah MA, Ahmad E (2002) Pleomorphic liposarcoma metastatic to the thyroid gland. Clin Nucl Med 27:9–10. 43. Lam KY, Lo CY (1998) Metastatic tumors of the thyroid gland: a study of 79 cases in Chinese patients. Arch Pathol Lab Med 122:37–41.
