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1
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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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Ullah S, Fazal H, Hassan S, Saqib M, Khan AW, Nashwan AJ, Ullah I. Primary leiomyosarcoma of thyroid with pulmonary metastasis: A diagnostic odyssey. Clin Case Rep 2024; 12:e8875. [PMID: 38736569 PMCID: PMC11087220 DOI: 10.1002/ccr3.8875] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/24/2023] [Revised: 04/18/2024] [Accepted: 04/21/2024] [Indexed: 05/14/2024] Open
Abstract
The presented primary thyroid leiomyosarcoma (TL) case report underscores the importance of recognizing and addressing the diagnostic challenges and management complexities associated with this exceedingly rare malignancy. Given the limited effective therapeutic strategies available, timely intervention, thorough diagnostics, and vigilant follow-up are paramount in managing such intricate tumors. Further research focusing on molecular-based treatment modalities is imperative to improve patient outcomes in cases of primary TL.
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Affiliation(s)
- Safi Ullah
- Kuwait Teaching HospitalPeshawarPakistan
| | | | - Subtain Hassan
- Department of Internal MedicineKhyber Teaching HospitalPeshawarPakistan
| | - Muhammad Saqib
- Department of Internal MedicineKhyber Teaching HospitalPeshawarPakistan
| | - Abdul Wali Khan
- University of Missouri Kansas City‐School of MedicineKansasMontanaUSA
| | | | - Irfan Ullah
- Department of Internal MedicineKhyber Teaching HospitalPeshawarPakistan
- Kabir Medical CollegeGandhara UniversityPeshawarPakistan
- Institute of Public Health and Social Sciences (IPH&SS)Khyber Medical UniversityPeshawarPakistan
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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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López F, Al Ghuzlan A, Zafereo M, Vander Poorten V, Robbins KT, Hamoir M, Nixon IJ, Tufano RP, Randolph G, Pace-Asciak P, Angelos P, Coca-Pelaz A, Khafif A, Ronen O, Rodrigo JP, Sanabria Á, Palme CE, Mäkitie AA, Kowalski LP, Rinaldo A, Ferlito A. Neck Surgery for Non-Well Differentiated Thyroid Malignancies: Variations in Strategy According to Histopathology. Cancers (Basel) 2023; 15:cancers15041255. [PMID: 36831604 PMCID: PMC9954150 DOI: 10.3390/cancers15041255] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/11/2023] [Revised: 02/04/2023] [Accepted: 02/13/2023] [Indexed: 02/18/2023] Open
Abstract
Lymph node metastases in non-well differentiated thyroid cancer (non-WDTC) are common, both in the central compartment (levels VI and VII) and in the lateral neck (Levels II to V). Nodal metastases negatively affect prognosis and should be treated to maximize locoregional control while minimizing morbidity. In non-WDTC, the rate of nodal involvement is variable and depends on the histology of the tumor. For medullary thyroid carcinomas, poorly differentiated thyroid carcinomas, and anaplastic thyroid carcinomas, the high frequency of lymph node metastases makes central compartment dissection generally necessary. In mucoepidermoid carcinomas, malignant peripheral nerve sheath tumors, sarcomas, and malignant thyroid teratomas or thyroblastomas, central compartment dissection is less often necessary, as clinical lymphnode involvement is less common. We aim to summarize the medical literature and the opinions of several experts from different parts of the world on the current philosophy for managing the neck in less common types of thyroid cancer.
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Affiliation(s)
- Fernando López
- Head and Neck Surgery, Hospital Universitario Central de Asturias (HUCA), 33011 Oviedo, Spain
- Instituto Universitario de Oncología del Principado de Asturias, Instituto de Investigación Sanitaria del Principado de Asturias, University of Oviedo, CIBERONC, 330011 Oviedo, Spain
- Correspondence: ; Tel.: +34-985108000
| | - Abir Al Ghuzlan
- Department of Biology and Pathology, Gustave Roussy Cancer Campus, University Paris-Saclay, 91190 Villejuif, France
| | - Mark Zafereo
- Department of Head and Neck Surgery, The University of Texas M. D. Anderson Cancer Center, Houston, TX 77030, USA
| | - Vincent Vander Poorten
- Otorhinolaryngology Head and Neck Surgery and Department of Oncology, Section Head and Neck Oncology, University Hospitals Leuven, 3000 Leuven, Belgium
- European Reference Network for Rare Adult Solid Cancers (EURACAN), 69008 Lyon, France
| | - K. Thomas Robbins
- Department of Otolaryngology-Head and Neck Surgery, Southern Illinois University School of Medicine, Springfield, IL 62702, USA
| | - Marc Hamoir
- Department of Head and Neck Surgery, UC Louvain, St Luc University Hospital and King Albert II Cancer Institute, 1200 Brussels, Belgium
| | - Iain J. Nixon
- Department of Otorhinolaryngology Head and Neck Surgery, NHS Lothian, Edinburgh EH1 3EG, UK
| | - Ralph P. Tufano
- FPG Thyroid and Parathyroid Center, Division of Head and Neck Endocrine Surgery, The Sarasota Memorial Health Care System, Sarasota, FL 34239, USA
| | - Gregory Randolph
- Division of Otolaryngology-Endocrine Head and Neck Surgery, Massachusetts Eye and Ear Infirmary, Harvard University, Boston, MA 02138, USA
| | - Pia Pace-Asciak
- Department of Otolaryngology-Head and Neck Surgery, University of Toronto, Toronto, ON M5S, Canada
| | - Peter Angelos
- Department of Surgery and MacLean Center for Clinical Medical Ethics, The University of Chicago, Chicago, IL 60637, USA
| | - Andrés Coca-Pelaz
- Head and Neck Surgery, Hospital Universitario Central de Asturias (HUCA), 33011 Oviedo, Spain
- Instituto Universitario de Oncología del Principado de Asturias, Instituto de Investigación Sanitaria del Principado de Asturias, University of Oviedo, CIBERONC, 330011 Oviedo, Spain
| | - Avi Khafif
- A.R.M. Center of Otolaryngology-Head and Neck Surgery, Assuta Medical Center, Affiliated with Ben-Gurion University of the Negev, Tel Aviv 8410501, Israel
| | - Ohad Ronen
- Department of Otolaryngology—Head and Neck Surgery, Galilee Medical Center, Affiliated with Azrieli Faculty of Medicine, Bar Ilan University, Safed 5290002, Israel
| | - Juan Pablo Rodrigo
- Head and Neck Surgery, Hospital Universitario Central de Asturias (HUCA), 33011 Oviedo, Spain
- Instituto Universitario de Oncología del Principado de Asturias, Instituto de Investigación Sanitaria del Principado de Asturias, University of Oviedo, CIBERONC, 330011 Oviedo, Spain
| | - Álvaro Sanabria
- Department of Surgery, Universidad de Antioquia, CEXCA Centro de Excelencia en Enfermedades de Cabeza y Cuello, Medellín 050021, Colombia
| | - Carsten E. Palme
- Department of Head and Neck Surgery, Sydney Head and Neck Cancer Institute, Chris O’Brien Lifehouse, Sydney, NSW 2050, Australia
- Faculty of Medicine and Health Sciences, Sydney Medical School, The University of Sydney, Sydney, NSW 2006, Australia
- Royal Prince Alfred Institute of Academic Surgery, Sydney Local Health District, Sydney, NSW 2050, Australia
| | - Antti A. Mäkitie
- Department of Otorhinolaryngology-Head and Neck Surgery, University of Helsinki and Helsinki University Hospital, FI-00029 Helsinki, Finland
| | - Luiz P. Kowalski
- Department of Head and Neck Surgery, University of Sao Paulo Medical School, Sao Paulo 01246-903, Brazil
- Department of Head and Neck Surgery and Otorhinolaryngology, AC Camargo Cancer Center, Sao Paulo 01509-001, Brazil
| | | | - Alfio Ferlito
- Coordinator of International Head and Neck Scientific Group, 35125 Padua, Italy
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5
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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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6
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Canu GL, Bulla JS, Lai ML, Medas F, Baghino G, Erdas E, Mariotti S, Calò PG. Primary thyroid leiomyosarcoma: a case report and review of the literature. G Chir 2019; 39:51-56. [PMID: 29549682 DOI: 10.11138/gchir/2018.39.1.051] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Abstract
Primary thyroid leiomyosarcoma (LMS) is an extremely rare tumor. We report a case of a 47-year-old male with a rapidly growing neck mass and disfagia. Preoperative investigations were diagnostic of anaplastic carcinoma. Total thyroidectomy with partial esophagectomy and dissection of right infrahyoid muscles was performed. Through histolological and immunohistochemical evaluations a primary thyroid high-grade LMS was diagnosed. At 2 months of follow-up a local recurrence was detected and consequently the patient was submitted to chemotherapy with partial response. He is still alive 9 months after surgery. Diagnosis of primary thyroid LMS is difficult due to its similarity to other more common thyroid tumors. To date, there is no standard therapy and prognosis is poor.
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7
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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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8
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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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9
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Wei J, Yang J, Liang W, Xu C, Wen Y. Clinicopathological features of primary thyroid leiomyosarcoma without Epstein-Barr virus infection: A case report. Oncol Lett 2019; 17:281-287. [PMID: 30655765 PMCID: PMC6313169 DOI: 10.3892/ol.2018.9609] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/19/2017] [Accepted: 09/07/2018] [Indexed: 12/12/2022] Open
Abstract
Primary thyroid leiomyosarcoma (LMS) is a rare tumor type with an unusual location, the diagnosis is based entirely on histological and immunohistochemical evaluations. In the present study, a rare case of a 74-year-old female patient who exhibited a right anterior neck mass for 12 months, which rapidly enlarged for the last 3 months. Ultrasound of the thyroid revealed a 55×42 mm hypoechoic mass with clear margins in the right lobe. Histological examination of the tumor demonstrated malignant spindle cells in interlacing fascicles and whorls. Additionally, nuclear pleomorphism, tumor giant cells, necrosis and abnormal mitotic figures were observed. The immunohistochemistry indicated that the tumor cells were strongly positive for smooth muscle actin, desmin, p53 and vimentin expression, but negative for cytokeratin, epithelial membrane antigen, thyroid transcription factor-1, paired box-8, 34βE12, cytokeratin 5/6, cluster of differentiation (CD)117, myoglobin, S100, p16. The final histopathological diagnosis was primary thyroid LMS.
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Affiliation(s)
- Jianguo Wei
- Department of Pathology, Shaoxing People's Hospital (Shaoxing Hospital, Zhejiang University School of Medicine), Shaoxing, Zhejiang 312000, P.R. China
| | - Jianfeng Yang
- Department of Radiology, Shaoxing People's Hospital (Shaoxing Hospital, Zhejiang University School of Medicine), Shaoxing, Zhejiang 312000, P.R. China
| | - Wenqing Liang
- Department of Pathology, Shaoxing People's Hospital (Shaoxing Hospital, Zhejiang University School of Medicine), Shaoxing, Zhejiang 312000, P.R. China
| | - Chunwei Xu
- Department of Pathology, Fujian Provincial Cancer Hospital, Fujian Medical University Cancer Hospital, Fuzhou, Fujian 350014, P.R. China
| | - Yuanyuan Wen
- Department of Pathology, Zhoushan Hospital of Zhejiang Province, Zhoushan, Zhejiang 316021, P.R. China
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10
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Ayadi M, Gabsi A, Meddeb K, Mokrani A, Yahiaoui Y, Letaief F, Chraiet N, Rais H, Mezlini A. Primary leiomyosarcoma of thyroid gland: the youngest case. Pan Afr Med J 2017; 26:113. [PMID: 28533836 PMCID: PMC5429415 DOI: 10.11604/pamj.2017.26.113.11472] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/22/2016] [Accepted: 02/02/2017] [Indexed: 12/21/2022] Open
Abstract
Primary leiomyosarcomas of the thyroid gland are extremely rare. We report a case of a 32 year-old women with a multinodular goiter. She underwent total thyroidectomy. The tumor histology showed spindle-shaped cells that expressed desmine, caldesmone and smooth muscle actine but were negative cytokeratins.
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Affiliation(s)
- Mouna Ayadi
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Azza Gabsi
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Khdija Meddeb
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Amina Mokrani
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Yosra Yahiaoui
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Feryel Letaief
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Nesrine Chraiet
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Henda Rais
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
| | - Amel Mezlini
- Service d'Oncologie Médicale, Institut Salah Azaiez Tunis, Faculté de Médecine de Tunis, Université el Manar, Tunisie
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11
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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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12
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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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13
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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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14
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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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15
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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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16
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Mouaqit O, Belkacem Z, Ifrine L, Mohsine R, Belkouchi A. A rare tumor of the thyroid gland: report on one case of leiomyosarcoma and review of literature. Updates Surg 2013; 66:165-7. [PMID: 23335096 DOI: 10.1007/s13304-013-0196-1] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/05/2012] [Accepted: 01/09/2013] [Indexed: 12/21/2022]
Affiliation(s)
- Ouadii Mouaqit
- Surgery Department «A», Ibn Sina University Hospital, Rabat, Morocco,
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17
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Piana S, Valli R, Foscolo AM. Thyroid leiomyosarcoma: primary or metastasis? That's the question! Endocr Pathol 2011; 22:226-8. [PMID: 21935659 DOI: 10.1007/s12022-011-9181-8] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/17/2022]
Affiliation(s)
- Simonetta Piana
- Department of Pathology, IRCCS-Arcispedale Santa Maria Nuova, Viale Risorgimento 80, 42100, Reggio Emilia, Italy.
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18
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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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19
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Postovsky S, Vlodavsky E, Kuten A, Shendler Y, Doweck I, Ben Arush MW. Undifferentiated sarcoma of the thyroid in a child. Pediatr Blood Cancer 2010; 54:1038-40. [PMID: 20127848 DOI: 10.1002/pbc.22435] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/24/2022]
Abstract
The most common malignant tumor of the thyroid is papillary carcinoma. Sarcoma of the thyroid is encountered very rarely; its therapy is complex and poses significant problems due to the problematic location of the tumor. A 14-year-old female was diagnosed with undifferentiated sarcoma of the thyroid and received combined therapy comprising surgery, chemo- and radiotherapy without significant side effects. This case underlines the fact that undifferentiated thyroid sarcoma may be a diagnostic possibility in children with malignant masses of the neck and may be successfully treated with modern therapeutic strategies.
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Affiliation(s)
- Sergey Postovsky
- Department of Pediatric Hematology-Oncology, Meyer Children's Hospital, Rambam Health Care Campus, Haifa, Israel.
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20
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Shi H, Wang C, Wei L, Lu S, Cao D. Malignant mesenchymoma of the thyroid: case report and literature review. TUMORI JOURNAL 2010; 96:345-348. [PMID: 20572598 DOI: 10.1177/030089161009600227] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/03/2025]
Abstract
Malignant mesenchymoma of the thyroid is extremely rare. We report such a tumor involving the bilateral lobes of the thyroid which showed simultaneous chondrosarcomatous, osteosarcomatous, fibrosarcomatous and rhabdomyosarcomatous differentiation. The patient was a 52-year-old woman admitted with a history of facial swelling, neck thickness and swallowing discomfort of one month's duration. Sonographic examination indicated a thyroid mass involving the bilateral lobes. Macroscopically, the tumors of both lobes were well demarcated, solid, greyish-white, and multinodular on the cut surface. Some nodules were translucent in appearance and hard in texture. Microscopically, the tumor was composed of small primitive mesenchymal cells with osteoid formation resembling the small cell variant of osteosarcoma interspersed with multiple cartilaginous nodules that indicated chondrosarcomatous differentiation. Some tumor cells showed prominent rhabdomyoblastic differentiation with eosinophilic cytoplasm and eccentric nuclei. Fibrosarcomatous areas were also observed. Immunohistochemically, the small primitive mesenchymal cells were positive for vimentin and CD99 and negative for CD56, Syn, CgA, CK, TG, TTF-1, calcitonin, and S-100. The tumor cells in the rhabdomyosarcomatous area were MyoD1 and muscle-specific actin positive. Molecular analysis for BRAFand RAS gene alterations showed no point mutation. The tumor recurred four months after surgery and tumor thrombi were suspected in the bilateral internal carotid arteries on ultrasonography. Primary malignant mesenchymoma of the thyroid is a high-grade malignant tumor with a poor prognosis. Its differerential diagnosis includes anaplastic carcinoma and other rare sarcomas with chondroid, osteoid, and other mesenchymal metaplasia.
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Affiliation(s)
- Huaiyin Shi
- Department of Pathology Chinese PLA General Hospital, Beijing, China.
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