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Ben Kridis W, Sakka D, Saguem I, Charfeddine I, Daoud J, Khanfir A. Primary Leiomyosarcoma of the Thyroid. Head Neck Pathol 2025; 19:11. [PMID: 39883292 PMCID: PMC11782753 DOI: 10.1007/s12105-024-01729-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/03/2024] [Accepted: 11/01/2024] [Indexed: 01/31/2025]
Abstract
Leiomyosarcoma (LMS) is a malignant mesenchymal neoplasm showing smooth muscle differentiation. Primary LMS of the thyroid gland is quite rare, accounting for only 0.014% of cases. We report a case of a leiomyosarcoma arising in the right thyroid lobe of a 43-year-old man with complete remission following surgery, radiation therapy and chemotherapy.
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Affiliation(s)
- Wala Ben Kridis
- Department of medical oncology, Habib Bourguiba Hospital, University of Sfax, Sfax, 3029, Tunisia.
| | - Dhouha Sakka
- Department of medical oncology, Habib Bourguiba Hospital, University of Sfax, Sfax, 3029, Tunisia
| | | | - Ilhem Charfeddine
- Ear nose and throat department, Habib Bourguiba Hospital, University of Sfax, Sfax, Tunisia
| | - Jamel Daoud
- Department of radiotherapy, Habib Bourguiba Hospital, University of Sfax, Sfax, Tunisia
| | - Afef Khanfir
- Department of medical oncology, Habib Bourguiba Hospital, University of Sfax, Sfax, 3029, Tunisia
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2
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Huang Z, Yu X, Yan J, Yin Y, Li J, Zheng Y. Primary leiomyosarcoma of the thyroid gland: A rare case report and literature review. Int J Surg Case Rep 2024; 119:109773. [PMID: 38772244 PMCID: PMC11128507 DOI: 10.1016/j.ijscr.2024.109773] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/23/2024] [Accepted: 05/15/2024] [Indexed: 05/23/2024] Open
Abstract
INTRODUCTION Primary thyroid leiomyosarcoma is an extremely rare soft tissue sarcoma, characterized by high malignancy and poor prognosis. Currently, only 13 cases of primary thyroid leiomyosarcoma have been described in the medical literature (limited to English). CASE PRESENTATION A 76-year-old female presented with a giant neck mass. Physical examination revealed a large, firm mass in the left thyroid gland. No symptoms such as hoarseness or dysphagia were noted at the time of presentation. The patient underwent unilateral thyroidectomy and cervical lymph node dissection. CLINICAL DISCUSSION Pathologic findings revealed a low-grade sarcoma with spindle-shaped tumor cells in an interwoven, sheet-like distribution. Immunohistochemistry showed positivity for desmin, SMMHC, STAT6, CK19, and Galectin3, but negativity for S-100, MyoD1, CD34, CK (AE1/AE3), CD117, and CD56. The findings were consistent with thyroid leiomyosarcoma. CONCLUSION The treatment of primary thyroid LMS presents challenges due to its atypical symptoms and high malignance, highlighting the imperative for further exploration of therapeutic strategies to improve the outcomes.
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Affiliation(s)
- Ziwei Huang
- Department of General Surgery, School of Medicine, The Fourth Affiliated Hospital, Zhejiang University, Yiwu 322000, China
| | - Xiping Yu
- Department of Pathology, School of Medicine, The Fourth Affiliated Hospital, Zhejiang University, Yiwu 322000, China
| | - Jialang Yan
- Department of General Surgery, School of Medicine, The Fourth Affiliated Hospital, Zhejiang University, Yiwu 322000, China
| | - Yuanxiao Yin
- Department of General Surgery, School of Medicine, The Fourth Affiliated Hospital, Zhejiang University, Yiwu 322000, China
| | - Junhao Li
- Department of General Surgery, School of Medicine, The Fourth Affiliated Hospital, Zhejiang University, Yiwu 322000, China
| | - Yixiong Zheng
- Department of General Surgery, School of Medicine, The Fourth Affiliated Hospital, Zhejiang University, Yiwu 322000, China.
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3
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Asiri M, Alsarrani F, Altasan A, Alqahtani F, Ali LA, Pharaon M, Alshehri S, Alshahrani A. Primary leiomyosarcoma of the thyroid with concurrent papillary thyroid cancer: a rare case report and a review of literature. Thyroid Res 2023; 16:16. [PMID: 37271804 DOI: 10.1186/s13044-023-00157-5] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/23/2023] [Accepted: 04/14/2023] [Indexed: 06/06/2023] Open
Abstract
BACKGROUND Leiomyosarcoma (LMS) is a soft tissue malignant tumor that has a predilection to the abdominopelvic and limb smooth muscles. LMS of the thyroid is exceptionally rare. Papillary thyroid cancer (PTC) is the most common thyroid malignancy and originates from the thyroid epithelial layer. To our knowledge, the presence of both tumors in the same patient has not been reported previously. CASE PRESENTATION & LITERATURE REVIEW A 42-year-old woman presented with a progressively enlarging neck mass for a few months. She underwent left thyroid lobectomy, and the histology showed high-grade primary LMS of the thyroid. She subsequently underwent a complete thyroidectomy, which identified a classical PTC on her right lobe. Our comprehensive literature review identified 39 published cases of primary LMS of the thyroid. The average tumor size was 5.88 cm and occurred more in women. The most common presentation was neck mass, followed by compressive symptoms. Recurrence and metastasis were uncommon at 15% and 10-25%, respectively. CONCLUSION Thyroid LMS is a rare malignancy with a worse prognosis than PTC. A thorough workup must be done to rule out metastasis before labeling it as primary thyroid cancer.
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Affiliation(s)
- Mohamed Asiri
- College of Medicine, King Saud Bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia.
- King Abdullah International Medical Research Center, Riyadh, Saudi Arabia.
| | - Faisal Alsarrani
- King Abdullah International Medical Research Center, Riyadh, Saudi Arabia
- Department of General Surgery, Ministry of National Guard - Health Affairs, Riyadh, Saudi Arabia
| | - Abdullah Altasan
- College of Medicine, King Saud Bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia
- King Abdullah International Medical Research Center, Riyadh, Saudi Arabia
| | - Faisal Alqahtani
- College of Medicine, King Saud Bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia
- King Abdullah International Medical Research Center, Riyadh, Saudi Arabia
| | - Lujain Akram Ali
- College of Medicine, Umm Al-Qura University, Makkah, Saudi Arabia
| | - Majed Pharaon
- King Abdullah International Medical Research Center, Riyadh, Saudi Arabia
- Department of Pathology and Laboratory Medicine, Ministry of National Guard - Health Affairs, Riyadh, Saudi Arabia
| | - Saad Alshehri
- King Abdullah International Medical Research Center, Riyadh, Saudi Arabia
- Department of General Surgery, Ministry of National Guard - Health Affairs, Riyadh, Saudi Arabia
| | - Awad Alshahrani
- King Abdullah International Medical Research Center, Riyadh, Saudi Arabia
- Department of Medicine, Ministry of National Guard - Health Affairs, Riyadh, Saudi Arabia
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4
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Bashir MT, Bradish T, Rasul U, Shakeel M. Primary thyroid leiomyosarcoma: a diagnostic and therapeutic challenge. BMJ Case Rep 2021; 14:14/4/e236399. [PMID: 33910786 PMCID: PMC8094372 DOI: 10.1136/bcr-2020-236399] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/09/2022] Open
Abstract
Leiomyosarcoma is a malignant mesenchymal tumour of smooth muscle origin. It is extremely rare as a primary thyroid cancer with only 33 cases previously described in the literature. We present the case of a 69-year-old Caucasian man who presented with a 5-month history of left cervical lymphadenopathy and a suspicious mass in the left thyroid lobe on ultrasound scan. Left hemithyroidectomy confirmed the diagnosis of leiomyosarcoma. A review of current understanding and approaches to management of this rare condition are discussed.
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Affiliation(s)
| | - Tom Bradish
- Department of Otorhinolaryngology, Head and Neck surgery, Aberdeen Royal Infirmary, Aberdeen, UK
| | - Usman Rasul
- School of Medicine, University of Aberdeen, Aberdeen, UK
| | - Muhammad Shakeel
- Department of Otorhinolaryngology, Head and Neck surgery, Aberdeen Royal Infirmary, Aberdeen, UK
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5
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Rossi ED, Pantanowitz L, Hornick JL. Cytologic and histological features of rare nonepithelial and nonlymphoid tumors of the thyroid. Cancer Cytopathol 2021; 129:583-602. [PMID: 33493367 DOI: 10.1002/cncy.22404] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/28/2020] [Revised: 12/02/2020] [Accepted: 12/03/2020] [Indexed: 11/09/2022]
Abstract
Thyroid tumors can be classified into epithelial, nonepithelial, and nonprimary lesions. Nonepithelial thyroid tumors are rare. They can be of primary origin within the thyroid gland, arise secondary to contiguous growth from adjacent tissues, or represent metastatic disease. The incidence of nonepithelial tumors of the thyroid is only 1% to 2%, most of which are lymphomas; the remainder includes mesenchymal and histiocytic tumors. This review examines the cytohistological features of various nonepithelial and nonlymphoid tumors of the thyroid, including vascular lesions, neural tumors (including granular cell tumor and paraganglioma), smooth muscle tumors, solitary fibrous tumor, histiocytic neoplasms (eg, Langerhans cell histiocytosis and Rosai-Dorfman disease), and follicular dendritic cell sarcoma. Their differential diagnosis is discussed, including recommendations to prevent the pitfall of mistaking these rare tumors for more common epithelial thyroid neoplasms.
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Affiliation(s)
- Esther Diana Rossi
- Division of Anatomic Pathology and Histology, Catholic University of Sacred Heart, Rome, Italy
| | - Liron Pantanowitz
- Department of Pathology, University of Michigan, Ann Arbor, Michigan
| | - Jason L Hornick
- Department of Pathology, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
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6
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Reddy B, Aggarwal V, Ajmani AK, Sachan S, Khandelwal D. Primary Leiomyosarcoma of the Thyroid Gland - A Rare Malignancy. EUROPEAN ENDOCRINOLOGY 2019; 15:44-46. [PMID: 31244910 PMCID: PMC6587898 DOI: 10.17925/ee.2019.15.1.44] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 08/27/2018] [Accepted: 01/03/2019] [Indexed: 12/16/2022]
Abstract
Primary leiomyosarcoma (LMS) of thyroid is rare malignant neoplasm usually seen in elderly patients. These tumours are characterised by a rapidly growing mass in the neck, difficult to diagnose preoperatively and are highly aggressive with high recurrence rates in spite of radical surgery, chemotherapy and radiotherapy. We herein report a case of 50-year-old female with a rapidly growing painless mass in the neck. Total thyroidectomy with bilateral neck node clearance was done. Histology and Immunohistochemistry revealed primary LMS of thyroid gland. Within 1 month of radical surgery she developed local recurrence. Diagnosis of primary LMS is difficult and immunohistochemistry is necessary to differentiate it from other malignancies of the thyroid. Primary LMS remains a life-threatening tumour and more effective treatment are needed to improve outcome.
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Affiliation(s)
| | | | | | - Seema Sachan
- Department of Pathology, B L Kapoor Superspeciality Hospital, Pusa Road, New Delhi, India
| | - Deepak Khandelwal
- Department of Endocrinology, Maharaja Agrasen Hospital, Punjabi Bagh, New Delhi, India
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7
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Vujosevic S, Krnjevic D, Bogojevic M, Vuckovic L, Filipovic A, Dunđerović D, Sopta J. Primary leiomyosarcoma of the thyroid gland with prior malignancy and radiotherapy: A case report and review of literature. World J Clin Cases 2019; 7:473-481. [PMID: 30842958 PMCID: PMC6397817 DOI: 10.12998/wjcc.v7.i4.473] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/26/2018] [Revised: 01/04/2019] [Accepted: 01/26/2019] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Leiomyosarcoma (LMS) of the thyroid gland is a rarely presented tumor that offers poor prognosis. To the best of the authors’ knowledge, there currently exist only 28 known cases described in the literature (limited to English).
CASE SUMMARY Herein a case is reported of a 60-year-old female patient who had an LMS of the thyroid, which was accompanied by periodic dysphonia and breathing disorder as well as the feeling of pressure in the chest and neck. At the time the disease was diagnosed, no metastases were detected. Prior to the diagnosis, the patient experienced a uterine adenocarcinoma that had been treated by surgical procedure and radiotherapy. For the LMS, a total thyroidectomy was performed, followed by radiotherapy. Since metastases were also discovered in the lungs, sternum, and femur, chemotherapy was administered as well. Immunohistochemically, the tumor cells in the thyroid indicated positively for alpha smooth muscle actin, calponin, and H-caldesmon, but were negative for CD34, p63, estrogen receptor, progesterone receptor, and Epstein-Barr virus.
CONCLUSION Although the etiology of the LMS is as of yet unknown, prior malignancy and radiation should be considered as risk factors.
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Affiliation(s)
- Snezana Vujosevic
- Endocrinology, Clinical Center of Montenegro, Podgorica 81000, Montenegro
| | - Djordjije Krnjevic
- Endocrinology, Clinical Center of Montenegro, Podgorica 81000, Montenegro
| | - Milan Bogojevic
- Internal Medicine Clinic, Clinical Center of Montenegro, Podgorica 81000, Crna Gora, Montenegro
| | - Ljiljana Vuckovic
- Institute of Pathology, Clinical Center of Montenegro, Podgorica 81000, Montenegro
| | | | - Duško Dunđerović
- Institute of Pathology, University of Belgrade, Belgrade 11000, Serbia
| | - Jelena Sopta
- Institute of Pathology, University of Belgrade, Belgrade 11000, Serbia
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8
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Zou ZY, Ning N, Li SY, Li J, DU XH, Li R. Primary thyroid leiomyosarcoma: A case report and literature review. Oncol Lett 2016; 11:3982-3986. [PMID: 27313727 PMCID: PMC4888224 DOI: 10.3892/ol.2016.4496] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/15/2015] [Accepted: 02/11/2016] [Indexed: 12/18/2022] Open
Abstract
Primary thyroid leiomyosarcoma (LMS) is an extremely rare soft tissue cancer; only 22 cases have been reported in the literature to date. In the current study, the case of an 83-year-old male patient who presented with a neck mass that had grown rapidly over the previous 3 months is reported. The patient underwent thyroid lobectomy twice and two cycles of immunotherapy for the treatment of primary thyroid LMS; however, he succumbed to the disease 5 months after the second surgery. An accurate diagnosis of primary thyroid LMS is difficult, as the disease is often misdiagnosed as anaplastic carcinoma, and requires the combined assessment of clinical, imaging and pathological data. Diagnosis of the current patient with primary thyroid LMS and a comprehensive review of the relevant literature are presented herein.
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Affiliation(s)
- Zhen-Yu Zou
- Department of General Surgery, Chinese People's Liberation Army General Hospital, Beijing 100853, P.R. China
| | - Ning Ning
- Department of Gastrointestinal Surgery, Peking University International Hospital, Beijing 102206, P.R. China
| | - Song-Yan Li
- Department of General Surgery, Chinese People's Liberation Army General Hospital, Beijing 100853, P.R. China
| | - Jie Li
- Department of Pathology, Chinese People's Liberation Army General Hospital, Beijing 100853, P.R. China
| | - Xiao-Hui DU
- Department of General Surgery, Chinese People's Liberation Army General Hospital, Beijing 100853, P.R. China
| | - Rong Li
- Department of General Surgery, Chinese People's Liberation Army General Hospital, Beijing 100853, P.R. China
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9
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Şahin Mİ, Vural A, Yüce İ, Çağlı S, Deniz K, Güney E. Thyroid leiomyosarcoma: presentation of two cases and review of the literature. Braz J Otorhinolaryngol 2016; 82:715-721. [PMID: 27080750 PMCID: PMC9444791 DOI: 10.1016/j.bjorl.2015.11.020] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/15/2015] [Accepted: 11/13/2015] [Indexed: 12/11/2022] Open
Abstract
Introduction Leiomyosarcoma is a tumor which is rarely seen in the thyroid gland. The diagnosis may be difficult and the treatment is controversial. Objective The objective of the study is to review the literature about a rare malignant disease of the thyroid gland which has high mortality. Methods Two cases of thyroid leiomyosarcoma are presented and the previous 23 cases in the current literature are reviewed. Results A total of 25 cases of thyroid leiomyosarcoma are reviewed; the most common complaint was rapidly growing anterior neck mass, and ten of the 25 patients had distant metastasis at the initial admission. Fifteen of the 25 patients died with the disease in the first 12 months after the diagnosis. Conclusion The differential diagnosis of thyroid leiomyosarcoma is important and should be performed with other malignancies of the gland, especially with anaplastic carcinoma. The prognosis is poor and there is no consensus regarding the treatment.
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Affiliation(s)
- Mehmet İlhan Şahin
- Erciyes University KBB Klinigi, Department of Otorhinolaryngology, Kayseri, Turkey
| | - Alperen Vural
- Erciyes University KBB Klinigi, Department of Otorhinolaryngology, Kayseri, Turkey.
| | - İmdat Yüce
- Erciyes University KBB Klinigi, Department of Otorhinolaryngology, Kayseri, Turkey
| | - Sedat Çağlı
- Erciyes University KBB Klinigi, Department of Otorhinolaryngology, Kayseri, Turkey
| | - Kemal Deniz
- Erciyes University KBB Klinigi, Department of Pathology, Kayseri, Turkey
| | - Ercihan Güney
- Erciyes University KBB Klinigi, Department of Otorhinolaryngology, Kayseri, Turkey
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10
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Conzo G, Candela G, Tartaglia E, Gambardella C, Mauriello C, Pettinato G, Bellastella G, Esposito K, Santini L. Leiomyosarcoma of the thyroid gland: A case report and literature review. Oncol Lett 2014; 7:1011-1014. [PMID: 24944660 PMCID: PMC3961299 DOI: 10.3892/ol.2014.1853] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/17/2013] [Accepted: 11/22/2013] [Indexed: 11/17/2022] Open
Abstract
Primary smooth muscle tumors of the thyroid gland are extremely rare neoplasms. Due to their rarity, clinical case studies concerning management are lacking. According to a literature review, only 19 cases of primary thyroid leiomyosarcomas (TLs) have been reported. In the majority of patients, the prognosis is poor since adjuvant radiochemotherapy is ineffective on local recurrence and on long-term survival. In this study, we report the case of a 77-year-old male affected by a rapidly enlarging mass of the anterior neck, associated with bilateral lung metastases, and increasing dysphagia and dyspnea during the previous 6 months. A Tir4 neoplasm fine needle cytological diagnosis of the right thyroid lobe was reached and the patient underwent total thyroidectomy (TT). Definitive histological examination identified a TL. The patient succumbed 40 days later due to respiratory distress. A literature review was performed and TL differential diagnoses, management, including alternative treatment strategies, and adjuvant therapy were analyzed. TL is an aggressive rare mesenchymal malignant tumor. Although an improved multimodal approach is often necessary, TT and neck dissection represent the treatment of choice and are often the only possible therapy. Adjuvant radiochemotherapy appears to be ineffective and a high mortality rate is observed. TL remains a fatal tumor, and innovative and more effective therapeutic strategies to improve management and outcomes are required.
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Affiliation(s)
- Giovanni Conzo
- Department of Anaesthesiology, Surgical and Emergency Science VII Division of General Surgery, Second University of Naples, Naples I-80131, Italy
| | - Giancarlo Candela
- Department of Anaesthesiology, Surgical and Emergency Science VII Division of General Surgery, Second University of Naples, Naples I-80131, Italy
| | - Ernesto Tartaglia
- Department of Anaesthesiology, Surgical and Emergency Science VII Division of General Surgery, Second University of Naples, Naples I-80131, Italy
| | - Claudio Gambardella
- Department of Anaesthesiology, Surgical and Emergency Science VII Division of General Surgery, Second University of Naples, Naples I-80131, Italy
| | - Claudio Mauriello
- Department of Anaesthesiology, Surgical and Emergency Science VII Division of General Surgery, Second University of Naples, Naples I-80131, Italy
| | - Guido Pettinato
- Thoracic Surgery Unit, Second University of Naples, Naples I-80138, Italy
| | - Giuseppe Bellastella
- Department of Cardio-Thoracic and Respiratory Sciences, Unit of Endocrinology and Diabetes Clinic, Second University of Naples, Naples I-80131, Italy
| | - Kathrine Esposito
- Department of Cardio-Thoracic and Respiratory Sciences, Unit of Endocrinology and Diabetes Clinic, Second University of Naples, Naples I-80131, Italy
| | - Luigi Santini
- Department of Anaesthesiology, Surgical and Emergency Science VII Division of General Surgery, Second University of Naples, Naples I-80131, Italy
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11
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Abstract
Primary leiomyosarcoma of the thyroid gland is uncommon. To date, 20 cases have been reported in English in the literature. The tumors usually present in elderly patients with female predilection and are associated with poor clinical outcome. Herein, we report an additional case of primary thyroid leiomyosarcoma in a 64-year-old woman. She underwent total thyroidectomy and later was discovered to have multiple lung and liver metastases. The patient died 3 months after surgery. The major differential diagnoses including undifferentiated (anaplastic) carcinoma of the thyroid, spindle cell variant of medullary thyroid carcinoma, spindle cell tumor with thymus-like differentiation, uncommon primary tumor of the thyroid and metastatic tumors with predominant spindle cells are discussed.
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Affiliation(s)
- Jantima Tanboon
- Department of Pathology, Faculty of Medicine, Siriraj Hospital, Mahidol University, Bangkok Noi, Bangkok, 10700, Thailand.
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12
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Ege B, Leventoğlu S. Primary leiomyosarcoma of the thyroid. JOURNAL OF THE KOREAN SURGICAL SOCIETY 2013; 85:43-6. [PMID: 23833760 PMCID: PMC3699687 DOI: 10.4174/jkss.2013.85.1.43] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 05/15/2012] [Revised: 01/21/2013] [Accepted: 02/07/2013] [Indexed: 12/21/2022]
Abstract
A 56-year-old male with primary leiomyosarcoma of the thyroid is presented. The paucity of diagnostic maneuvers, including tumor markers, fine needle aspiration, and frozen section biopsy, are stressed, in addition to the fulminate course of the disease.
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Affiliation(s)
- Bahadır Ege
- Clinic of General Surgery, Private Koru Hospital, Ankara, Turkey
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13
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[Thyroid gland primary leiomyosarcoma]. ACTA ACUST UNITED AC 2010; 54:326-30. [PMID: 20520964 DOI: 10.1590/s0004-27302010000300012] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/11/2009] [Accepted: 11/02/2009] [Indexed: 01/28/2023]
Abstract
Despite the fact that 15% to 20% of sarcomas occur in the head and neck and 80% in adults, only 0.014% are primary thyroid leiomyosarcomas. To the best of our knowledge, only 16 cases have been reported around the world, none in South America. Cytologic diagnosis is challenging and these tumors may be mistaken by more common ones such as anaplastic or medullary carcinomas. The treatment of choice for thyroid leiomyosarcomas is not well established yet because of its poor prognosis. Radical surgery associated with chemoradiotherapy has not been effective and did not improve survival rates. The authors report a case of primary thyroid leiomyosarcoma in a young male, who has been submitted to total thyroidectomy and selective neck dissection. Extensive literature review was performed by the authors. The patient received adjuvant radiotherapy, presenting good postoperative course. After four years evolution, there was no local recurrence or distant metastasis.
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14
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Abstract
Thyroid neoplasms are classified into three major categories: epithelial, nonepithelial, and secondary. Most primary epithelial tumors of thyroid are derived from follicular cells. These include follicular adenoma and carcinoma (Hürthle and non-Hürthle), and papillary carcinoma and its variants. Other primary epithelial tumors include medullary carcinoma, mixed medullary and follicular carcinomas, insular and poorly differentiated carcinoma, anaplastic carcinoma, and the least common squamous carcinoma and related tumors. The nonepithelial tumors are rare; the most common include malignant lymphoma and tumors arising from the mesenchymal elements. The secondary tumors represent metastatic tumors to the thyroid usually originating in lung, kidney, and breast. In this article, the authors review the unusual tumors of the thyroid, their morphologic features, and clinical and prognostic implications.
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Affiliation(s)
- Zubair W Baloch
- Department of Pathology and Laboratory Medicine, University of Pennsylvania Medical Center, 3400 Spruce Street, 6 Founders Pavilion, Philadelphia, PA 19104, USA
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15
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Abstract
Primary leiomyosarcomas of the thyroid gland are rare. We present the case of a 65-year-old woman with a rapidly enlarging neck mass for 2 months. The preoperative differential diagnosis included medullary thyroid cancer, anaplastic thyroid cancer, and primary versus metastatic sarcoma. The patient underwent total thyroidectomy, bilateral central neck dissections, and cervical thymectomy; she is currently being treated with ifosfamide and adriamycin. We review the literature on leiomyosarcoma of the thyroid, including the differential diagnoses, pathology, and alternative treatment strategies, including surgery and adjuvant therapy.
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Affiliation(s)
- Tracy S Wang
- Section of Endocrine Surgery, Department of Surgery, Yale University School of Medicine, New Haven, Connecticut 06520-8062, USA
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16
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Mansouri H, Gaye M, Errihani H, Kettani F, El Gueddari B. Leiomyosarcoma of the thyroid gland. Acta Otolaryngol 2008; 128:335-6. [PMID: 18274920 DOI: 10.1080/00016480500527193] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
The existence of primary thyroid sarcomas represents a contentious issue. Some authors believe that thyroid sarcomas are, in fact, anaplastic carcinomas with sarcomatous (spindle cell) features. From the standpoint of treatment and prognosis, the histogenesis of thyroid 'sarcomas' is academic, because these tumors, irrespective of the therapeutic intervention, are lethal 1. Primary thyroid leiomyosarcomas are extremely rare. Only nine isolated cases have been reported in the literature and such tumors carry a very poor prognosis [1-7] .
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17
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Just PA, Guillevin R, Capron F, Le Charpentier M, Le Naour G, Menegaux F, Leenhardt L, Simon JM, Hoang C. An unusual clinical presentation of a rare tumor of the thyroid gland: report on one case of leiomyosarcoma and review of literature. Ann Diagn Pathol 2007; 12:50-6. [PMID: 18164417 DOI: 10.1016/j.anndiagpath.2006.06.006] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/21/2022]
Abstract
Primary muscle tumors of the thyroid gland are exceedingly rare. We report on the case of a patient with primary leiomyosarcoma of the thyroid gland and review the literature. An 83-year-old woman complaining of neuropathic pain in her left arm and enlargement of her anterior neck underwent multiple surgical biopsies of the thyroid gland. The tumor was composed of interlacing fascicles of spindle-shaped cells that expressed smooth muscle actin and vimentin but were negative for cytokeratins and thyroglobulin. Ultrastructurally, bundles of myofilaments were present. Magnetic resonance imaging showed a thyroid tumor that directly extended to the adjacent vertebra with an associated pachymeningitis. The patient died 2 months after surgery. The diagnosis of primary leiomyosarcoma of the thyroid gland is difficult and requires numerous clinical, radiologic, and pathologic data. To our knowledge, this case is the first one with such a locoregional extension.
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Affiliation(s)
- Pierre-Alexandre Just
- Service d'Anatomie et de Cytologie Pathologiques, Groupe Hospitalier Pitié-Salpêtrière, 75651 Paris Cedex 13, France
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Day AS, Lou PJ, Lin WC, Chou CC. Over-expression of c-kit in a primary leiomyosarcoma of the thyroid gland. Eur Arch Otorhinolaryngol 2007; 264:705-8. [PMID: 17256123 DOI: 10.1007/s00405-007-0242-z] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/09/2006] [Accepted: 12/09/2006] [Indexed: 11/30/2022]
Abstract
Primary leiomyosarcoma of the thyroid gland is rare. In this paper, we report a case of high-grade leiomyosarcoma of the thyroid gland in a 43-year-old man. Lung metastasis was also noted in this patient. Despite of aggressive surgical treatment, the patient died of uncontrolled local recurrent disease 6 months after the initial operation. Immunohistochemical studies showed the tumor cells were positive for c-kit proto-oncogene product. Imatinib mesylate was used as a post-operative adjuvant treatment but the response was poor. The role of tyrosine kinase inhibitors on the treatment of thyroid leiomyosarcomas is still unclear because this is the first report of c-kit over-expression in such tumors. Nevertheless, our results show that c-kit over-expression might not be an indicator of good response to imatinib mesylate treatment in thyroid leiomyosarcomas.
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Affiliation(s)
- An-Shiou Day
- Department of Otolaryngology, National Taiwan University Hospital and National Taiwan University College of Medicine, Taipei, Taiwan, ROC
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19
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Carda C, Ferrer J, Vilanova M, Peydró A, Llombart-Bosch A. Anaplastic carcinoma of the thyroid with rhabdomyosarcomatous differentiation: a report of two cases. Virchows Arch 2004; 446:46-51. [PMID: 15517365 DOI: 10.1007/s00428-004-1123-0] [Citation(s) in RCA: 22] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/10/2004] [Accepted: 08/19/2004] [Indexed: 11/28/2022]
Abstract
Anaplastic carcinoma of the thyroid gland (ACT) is a highly malignant tumor that is almost invariably associated with a fatal outcome. It demonstrates a variety of peculiar histological features, with squamoid, giant cell and spindle cell growth patterns. The spindle cell variant of ACT is usually indistinguishable from a true sarcoma and it can simulate fibrosarcoma, malignant fibrous histiocytoma (MFH), hemangiopericytoma and angiosarcoma or rhabdomyosarcoma. Although a rhabdomyosarcomatous appearance has sometimes been mentioned in the literature, true skeletal muscle differentiation has never been consistently proved. We report two cases of ACT with rhabdomyosarcomatous differentiation, as demonstrated by means of immunohistochemistry and electron microscopy. Both cases disclosed a very similar histological appearance, with a main population of small, pleomorphic, round-to-oval cells arranged in a storiform pattern, admixed with scattered pleomorphic giant cells, an image similar to that of the usual type of MFH. Stains for epithelial markers showed only few, scattered, weakly positive cells. Thyroglobulin and calcitonin were negative in tumor cells in both cases. On the contrary, positivity to vimentin was strong and generalized. Immunomarkers of muscular differentiation showed a consistent positivity. At the ultrastructural level, the cells disclosed the same spindle and pleomorphic morphology, with large, bizarre nuclei and cytoplasm with abundant mitochondria, rough endoplasmic reticulum, secretory granules and lipid droplets. There were also cells with wide cytoplasm filled with filamentous material, either of actin or myosin, as well as Z-band material. In conclusion, the cases reported here show a clear-cut rhabdomyosarcomatous differentiation of ACT, confirmed both immunohistochemically and ultrastructurally, a feature not previously reported in the literature. These findings may contribute to the broadening of the differentiation spectrum of this unusual neoplasm.
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Affiliation(s)
- Carmen Carda
- Department Pathology, Faculty of Medicine, University of Valencia, Av. Blasco Ibañez 17, 46010 Valencia, Spain.
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20
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Takayama F, Takashima S, Matsuba H, Kobayashi S, Ito N, Sone S. MR imaging of primary leiomyosarcoma of the thyroid gland. Eur J Radiol 2001; 37:36-41. [PMID: 11274837 DOI: 10.1016/s0720-048x(00)00217-5] [Citation(s) in RCA: 20] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/21/2022]
Abstract
Primary leiomyosarcoma of the thyroid gland is extremely rare and radiological information on this tumor is scant. We presented radiological findings on primary thyroid leiomyosarcoma in a 66-year-old woman in which anaplastic carcinoma was suspected based on clinical and cytological features and discussed the radiological clues to distinguish between the two diseases. Ultrasonography showed an ill-defined hypoechoic mass without halo in the left lobe and the isthmus of the thyroid gland. Computed tomography depicted a low-density mass with calcification and necrosis, which invaded the thyroid cartilage. No lymphadenopathy was seen. The tumor was demonstrated as an isointense mass on T1-weighted MR images and a mass of intermediate signal on T2-weighted images. The tumor showed a fair enhancement on gadolinium-enhanced T1-weighted images. Although the radiological picture was nonspecific, primary thyroid leiomyosarcoma appeared less invasive and far less frequent in developing nodal metastasis than anaplastic carcinoma in light of the literature.
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Affiliation(s)
- F Takayama
- Department of Radiology, Shinshu University School of Medicine, Asahi, Matsumoto, Japan
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21
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22
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Tulbah A, Al-Dayel F, Fawaz I, Rosai J. Epstein-Barr virus-associated leiomyosarcoma of the thyroid in a child with congenital immunodeficiency: a case report. Am J Surg Pathol 1999; 23:473-6. [PMID: 10199478 DOI: 10.1097/00000478-199904000-00013] [Citation(s) in RCA: 63] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/21/2022]
Abstract
We report an unusual case of multifocal leiomyosarcoma involving the thyroid gland, liver, and right lung in a child with congenital immunodeficiency disease. The smooth muscle nature of these neoplasms was confirmed by immunohistochemistry and electron microscopic studies. In situ hybridization showed large amounts of Epstein-Barr virus messenger RNA within the tumor cells. Although Epstein-Barr virus-associated smooth muscle tumors have been reported in children with AIDS and after organ transplantation, we are unaware of any case report in congenital immunodeficiency disease.
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Affiliation(s)
- A Tulbah
- Department of Pathology and Laboratory Medicine, King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia
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23
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Abstract
BACKGROUND Primary smooth muscle tumors of the thyroid gland are rare. To date, there are few cases reported of primary thyroid leiomyomas and leiomyosarcomas. METHODS One leiomyoma and four leiomyosarcomas arising within the thyroid gland were identified in the files of the Endocrine Tumor Registry of the Armed Forces Institute of Pathology. Histologic and immunohistochemical features were reviewed and follow-up obtained. RESULTS The patients included 2 females, ages 56 and 64 years, and 3 males, ages 45, 68, and 83 years. The patients presented with a mass in the thyroid gland that had increased in size over a number of months. All the tumors originated within a single lobe of the thyroid gland and measured from 1.1 to 9 cm in greatest dimension. Histologically, there was a fascicular pattern of growth comprised of spindle-shaped cells with blunt-ended nuclei. The leiomyoma was encapsulated, cytologically bland, and amitotic; the leiomyosarcomas were invasive with increased cellularity, pleomorphism, a high mitotic rate, necrosis, and hemorrhage. Immunohistochemical staining showed reactivity with vimentin, smooth muscle actin, muscle specific actin, and desmin. The patient with the leiomyoma was alive without evidence of disease 11 years after the initial presentation, with surgical resection as the only treatment. Three of the patients with leiomyosarcomas were dead within 2 years of diagnosis, in spite of aggressive therapeutic intervention. The remaining patient was still alive 10 months after initial presentation with multiple lung metastases. CONCLUSIONS Smooth muscle tumors of the thyroid gland are distinctive tumors. Leiomyosarcomas can be distinguished from anaplastic carcinoma, although patient outcome is uniformly unfavorable.
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Affiliation(s)
- L D Thompson
- Department of Endocrine and Otorhinolaryngic-Head & Neck Pathology, Armed Forces Institute of Pathology, Washington, DC 20306-6000, USA
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Ozaki O, Sugino K, Mimura T, Ito K, Tamai S, Hosoda Y. Primary leiomyosarcoma of the thyroid gland. Surg Today 1997; 27:177-80. [PMID: 9018000 DOI: 10.1007/bf02385912] [Citation(s) in RCA: 21] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
Primary leiomyosarcoma of the thyroid gland is extremely rare, and to the best of our knowledge only five well-documented cases have been reported in the world literature. We herein report a 58-year-old female patient with primary leiomyosarcoma of the thyroid who was successfully treated by total thyroidectomy with a modified neck dissection. Immunohistochemically, the tumor cells showed positive reactivity to alpha-smooth muscle actin and vimentin. Radical surgery was thus considered to be essential in the treatment of this rare but rather aggressive malignancy.
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Affiliation(s)
- O Ozaki
- Department of Surgery, Ito Hospital, Shibuya-ku, Tokyo, Japan
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25
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Thompson LDR, Wenig BM, Adair CF, Heffess CS. Peripheral Nerve Sheath Tumors of the Thyroid Gland: A Series of Four Cases and a Review of the Literature. Endocr Pathol 1996; 7:309-318. [PMID: 12114802 DOI: 10.1007/bf02739838] [Citation(s) in RCA: 21] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/16/2022]
Abstract
Primary peripheral nerve sheath tumors (PNSTs) of the thyroid gland are exceptionally rare. Two schwannomas and two malignant PNSTs (MPNSTs), arising primarily within the thyroid gland, were identified in the files of the Endocrine Tumor Registry at the Armed Forces Institute of Pathology. The patients included two females, age 69 and 80 yr, and two males, age 18 and 33 yr. The patients presented with a mass in the thyroid gland confined to a single lobe of the thyroid without involvement of the cervical neck region. None of the patients had a history of neurofibromatosis. The benign tumors were encapsulated, one of them cystic, with the characteristic cellular and nuclear features of schwannomas. The MPNSTs were invasive tumors, effacing the thyroid parenchyma, with a fascicular pattern of growth composed of neural appearing cells with increased cellularity, increased mitotic activity, and with focal necrosis. Immunoreactivity for 5100 protein and vimentin was seen in all tumors. The patients with schwannomas, treated only by surgical resection, were alive without evidence of disease, over a period of 5-33 yr. Both patients with MPNSTs died of the disease 8 mo and 42 mo, respectively, with widely disseminated disease. Primary thyroid PNSTs are exceptionally rare tumors. MPNSTs, in this limited experience, have a fatal outcome irrespective of aggressive adjuvant therapy.
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