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Xu H, Qian T, He X, Huang C, Fei Z, Xu S, Ma B, Wang Y, Wang J. Carcinoma Showing Thymus-Like Differentiation (CASTLE): A Case Report and Review of the Literature. EAR, NOSE & THROAT JOURNAL 2025:1455613251326051. [PMID: 40114079 DOI: 10.1177/01455613251326051] [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: 03/22/2025] Open
Abstract
Carcinoma showing thymus-like differentiation (CASTLE) is a rare tumor occurring in the thyroid and soft tissues of the neck. Treatments and prognosis for CASTLE significantly differ from those associated with squamous cell carcinoma and anaplastic thyroid carcinoma. To date, fewer than 100 cases have been reported. Given the lack of a standardized treatment protocol and the diagnostic complexity, documenting these cases is essential. A 74-year-old male patient was admitted to the hospital due to hoarseness lasting for 1 month and thyroid nodules for over 2 weeks. The ultrasound revealed a grade 4 nodule in the left thyroid, and the needle biopsy reported the presence of glandular epithelial cells of varying sizes, some arranged in a papillary pattern, and was negative for BRAF mutation. Thyroid surgeons performed a left partial thyroidectomy and a left cervical lymph node biopsy, and the immunohistochemical staining led to a diagnosis of intrathyroid thymus carcinoma. Follow-up on his postoperative recovery shown no signs of local recurrence or distant metastasis during this period. This paper conducts a systematic review of the literature to enhance understanding, diagnosis, treatment, and prognosis of this condition. The goal was to raise clinical awareness and guide appropriate treatment strategies.
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Affiliation(s)
- Hongyan Xu
- Department of Thyroid and Breast Surgery, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
| | - Tao Qian
- Department of Thyroid and Breast Surgery, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
| | - Xinyi He
- Department of Thyroid and Breast Surgery, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
| | - Chi Huang
- Department of Thyroid and Breast Surgery, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
| | - Zongqi Fei
- Department of Thyroid and Breast Surgery, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
| | - Shuhang Xu
- Department of Endocrinology, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
| | - Bing'e Ma
- Department of Thyroid and Breast Surgery, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
| | - Yihua Wang
- Biological Sciences, Faculty of Environmental and Life Sciences, University of Southampton, UK
- Institute for Life Sciences, University of Southampton, UK
- NIHR Southampton Biomedical Research Centre, University Hospital Southampton, UK
| | - Jianhua Wang
- Department of Thyroid and Breast Surgery, Affiliated Hospital of Integrated Traditional Chinese and Western Medicine, Nanjing University of Chinese Medicine, Jiangsu, People's Republic of China
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Balaji AN, Balasubramanian B, Hazeem JA. Intrathyroidal Thymoma: A Diagnostic Challenge. Cureus 2024; 16:e68987. [PMID: 39385927 PMCID: PMC11463876 DOI: 10.7759/cureus.68987] [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] [Accepted: 09/09/2024] [Indexed: 10/12/2024] Open
Abstract
Intrathyroidal thymoma is a rare tumor that can be challenging to diagnose due to its unusual location and resemblance to more common thyroid conditions. We present the case of a 58-year-old woman with an incidentally discovered thyroid nodule during evaluation for an upper respiratory infection. Ultrasonography revealed an exophytic nodule in the left thyroid lobe, categorized as TR 3. Fine-needle aspiration cytology suggested a neoplastic process, leading to a left hemithyroidectomy. Histopathology confirmed a diagnosis of intrathyroidal thymoma, Type B2, with extensive necrosis, and immunohistochemistry validated the findings. This case underscores the diagnostic challenges of intrathyroidal thymoma, emphasizing its consideration in the differential diagnosis of atypical thyroid nodules. Despite the difficulties in preoperative identification, surgical resection and subsequent histopathological examination remain essential for a definitive diagnosis. The patient is currently under surveillance, and there is no evidence of residual thymic tissue or abnormalities in the remaining thyroid tissue.
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Hsu YC, Hsueh C, Lin WN, Tsai TY, Hung SY, Lu YA. Carcinoma Showing Thymus-like Differentiation (CASTLE) with Synchronous Papillary Thyroid Carcinoma: A Case Report and Review. EAR, NOSE & THROAT JOURNAL 2024; 103:NP411-NP421. [PMID: 34866458 DOI: 10.1177/01455613211060167] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022] Open
Abstract
Carcinoma showing thymus-like differentiation (CASTLE) is a rare malignant tumor that accounts for 0.1%-0.15% of all thyroid cancers. More than half of the patients have tumor extension to adjacent organs, including the recurrent laryngeal nerve, trachea, and esophagus. The diagnosis of CASTLE is based on histology and immunohistochemistry. A 58-year-old female patient complained of hoarseness for one and half years. Right side vocal cord palsy was diagnosed by fiberscopy. Thyroid sonography revealed right thyroid tumors, which were reported to be papillary thyroid carcinoma through FNAC. Total thyroidectomy with central lymph node dissection was performed. Pathologist found 2 isolated malignancy tumors. One patient in the right thyroid lobe had papillary thyroid carcinoma features. The other extrathyroid tumor seemed to be separated from the first tumor and invaded the thyroid capsule. After multiple immunohistochemical studies, PTC synchronous CASTLE was the final diagnosis. Coexisting PTC and CASTLE is very rare. This is the first report to describe a case showing PTC at first, while subsequent pathologic examination revealed the presence of CASTLE in addition to PTC. Since the prognosis of CASTLE is favorable, the treatment is different from other aggressive thyroid cancers, such as poorly differentiated or anaplastic thyroid carcinoma.
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Affiliation(s)
- Yuan-Chin Hsu
- Department of Otolaryngology Head and Neck Surgecxry, Linkou Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Chuen Hsueh
- Department of Pathology, Linkou Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Wan-Ni Lin
- Department of Otolaryngology Head and Neck Surgecxry, Linkou Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Tsung-You Tsai
- Department of Otolaryngology Head and Neck Surgecxry, Linkou Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Shih-Yuan Hung
- Department of Endocrinology and Metabolism, Linkou Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Yi-An Lu
- Department of Otolaryngology Head and Neck Surgecxry, Linkou Chang Gung Memorial Hospital, Taoyuan, Taiwan
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Vajihinejad M, Ataei A, Pashmchi M, Aledavoud A, Zand V, Broomand MA, Mohammadi M, Reshkuiyeh NZ. Coexistence of intrathyroid thymic carcinoma and papillary thyroid carcinoma: a case report and literature review. Front Oncol 2024; 14:1394020. [PMID: 38764579 PMCID: PMC11099278 DOI: 10.3389/fonc.2024.1394020] [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: 02/29/2024] [Accepted: 04/23/2024] [Indexed: 05/21/2024] Open
Abstract
BACKGROUND Intrathyroid thymic carcinoma (ITTC) is a rare neoplasm of the thyroid, which accounts for less than 0.15% of all thyroid malignancies. The coexistence of ITTC and papillary thyroid carcinoma (PTC) is an extremely rare condition reported only in a limited number of cases. CASE SUMMARY A 26-year-old female presented with a growing neck mass, hoarseness, and dysphagia over four months. Ultrasonography revealed that the entire left lobe and the isthmus of the thyroid were replaced with a hypoechoic mass. Moreover, it revealed two hypoechoic nodules in the right thyroid. The patient underwent a total thyroidectomy and paratracheal lymph node dissection. Histopathological examinations revealed the coexistence of ITTC and PTC in the same thyroid. In immunohistochemical analyses, the ITTC was positive for CD5, P63, CD117, and CK 5/6 and negative for thyroglobulin, calcitonin, and TTF 1. At the same time, PTC was positive for TTF 1 and thyroglobulin and negative for CD5, P63, and CK 5/6. The patient received postoperative radiotherapy and remained well with no evidence of recurrence during one month follow-up. CONCLUSION Distinguishing ITTC from other thyroid malignancies before the surgery is challenging due to its non-specific presentations. Therefore, the diagnosis relies on postoperative studies, especially immunohistochemistry. The recommended treatment approach to improve survival in ITTC cases is total thyroidectomy combined with cervical lymph node dissection, followed by postoperative radiotherapy. The coexistence of ITTC and PTC may indicate the similarity in the underlying mechanisms of these tumors. However, further investigations are needed to understand this potential correlation.
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Affiliation(s)
- Maryam Vajihinejad
- Department of Pathology, Shahid Sadoughi University of Medical Sciences, Yazd, Iran
| | - Ali Ataei
- School of Medicine, Bam University of Medical Sciences, Bam, Iran
| | - Mohammad Pashmchi
- School of Medicine, Shahid Sadoughi University of Medical Sciences, Yazd, Iran
| | - Ali Aledavoud
- School of Medicine, Kerman University of Medical Sciences, Kerman, Iran
| | - Vahid Zand
- Department of Otolaryngology-Head and Neck Surgery, Otorhinolaryngology Research Center, Shahid Sadoughi University of Medical Sciences, Yazd, Iran
| | - Mohammad Ali Broomand
- Department of Clinical Oncology, Shahid Sadoughi University of Medical Sciences, Yazd, Iran
| | - Mohammad Mohammadi
- School of Medicine, Shahid Sadoughi University of Medical Sciences, Yazd, Iran
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Hamada M, Miyama Y, Matsumura S, Shintani‐Domoto Y, Urano M, Yasuda M. Carcinoma showing thymus-like elements (CASTLE) with amyloid deposition in the parotid gland. Pathol Int 2024; 74:227-233. [PMID: 38488752 PMCID: PMC11551826 DOI: 10.1111/pin.13420] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/03/2024] [Revised: 02/27/2024] [Accepted: 03/01/2024] [Indexed: 04/11/2024]
Abstract
Carcinoma showing thymus-like elements (CASTLE) is a rare tumor that commonly occurs in the thyroid gland. Extrathyroidal CASTLE is rarer, and only 11 cases of CASTLE of major salivary glands have been reported to date. We report the first case of amyloid deposition in parotid CASTLE. A 63-year-old man presented with a slowly growing mass in the left parotid region. Computed tomography revealed an approximately 28 × 23 mm mass lesion in the left parotid gland, and squamous cell carcinoma was suspected on biopsy. The patient underwent a parotidectomy with neck dissection. Morphologically, the tumor cells were squamoid and formed nests with lymphoid infiltration. Immunohistochemically, the tumor cells exhibited immunoreactivity for CD5, CD117/c-kit and Bcl-2, p40, and CK5 but not for p16. We diagnosed the tumor as parotid CASTLE. Amyloid deposition was also observed in the primary tumor and metastatic lymph node lesions, which were immunoreactive for cytokeratin 5. Tumor cytokeratin-derived amyloid deposition may be one of characteristics of parotid CASTLE.
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Affiliation(s)
- Mei Hamada
- Department of Pathology, Saitama International Medical CenterSaitama Medical UniversitySaitamaJapan
- Department of Diagnostic PathologySaitama Medical UniversitySaitamaJapan
| | - Yu Miyama
- Department of Pathology, Saitama International Medical CenterSaitama Medical UniversitySaitamaJapan
| | - Satoko Matsumura
- Department of Head and Neck Surgery, Saitama International Medical CenterSaitama Medical UniversitySaitamaJapan
| | | | - Makoto Urano
- Department of Diagnostic Pathology, Bantane HospitalFujita Health University School of MedicineNagoyaJapan
| | - Masanori Yasuda
- Department of Pathology, Saitama International Medical CenterSaitama Medical UniversitySaitamaJapan
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Gao Y, Pan Y, Luo Y, Cheng R, Zhai Q. Intrathyroid thymic carcinoma: A clinicopathological analysis of 22 cases. Ann Diagn Pathol 2023; 67:152221. [PMID: 37925863 DOI: 10.1016/j.anndiagpath.2023.152221] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/29/2023] [Revised: 10/19/2023] [Accepted: 10/21/2023] [Indexed: 11/07/2023]
Abstract
OBJECTIVE Intrathyroid thymic carcinoma (ITTC) is a rare malignancy. The current understanding of ITTC is inadequate, and there is no standard treatment for ITTC. In the present study, we aimed to explore the clinicopathological characteristics of ITTC and identify potential therapeutic targets. METHODS The clinicopathological characteristics of 22 ITTC patients at our institution were reviewed. The expression of DNA mismatch repair (MMR) proteins and PD-L1 in ITTC were assessed by immunohistochemistry (IHC). RESULTS All patients underwent surgery. There were nine females and 13 males, with a slight male predominance. Their ages ranged from 42 to 79 years (average, 54. 1 years). The diameters of the neck masses ranged from 10 to 100 mm (average, 39 mm). Ipsilateral lymph node (LN) dissection was performed in 18 patients: 12 demonstrated LN metastasis, six showed no LN metastasis, and no lymph nodes were dissected in four. One patient had liver metastasis. CK5/6, P63, CD5, and CD117 were expressed in all cases. All cases were negative for TTF1, PAX8, thyroglobulin, and BRAF V600E. DNA MMR protein expression was retained in all tested tumors, and EBV-encoded small RNA (EBER) in situ hybridization was consistently negative. The Ki67 proliferation index ranged from 10 to 70 %. All patients were followed-up for 14-134 months, four died, six were lost to follow-up, and the remaining patients survived without disease. The PD-L1 combined positive score ranged from 10 to 80 (average: 40). CONCLUSION Our results confirm that CD5 and CD117 co-expression support a diagnosis of ITTC. All tumors in this cohort were DNA MMR-proficient and were not associated with Epstein-Barr virus (EBV) infection. A high CPS for PD-L1 suggests that immune checkpoint inhibitor therapy may be worthy of further exploration in patients with ITTC.
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Affiliation(s)
- Yanan Gao
- Department of Pathology, National Clinical Research Centre for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Centre for Cancer, Tianjin Medical University Cancer Institute and Hospital, Huanhu West Road, Hexi District, Tianjin 300060, China.
| | - Yi Pan
- Department of Pathology, National Clinical Research Centre for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Centre for Cancer, Tianjin Medical University Cancer Institute and Hospital, Huanhu West Road, Hexi District, Tianjin 300060, China
| | - Ye Luo
- Department of Pathology, National Clinical Research Centre for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Centre for Cancer, Tianjin Medical University Cancer Institute and Hospital, Huanhu West Road, Hexi District, Tianjin 300060, China
| | - Runfen Cheng
- Department of Pathology, National Clinical Research Centre for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Centre for Cancer, Tianjin Medical University Cancer Institute and Hospital, Huanhu West Road, Hexi District, Tianjin 300060, China
| | - Qiongli Zhai
- Department of Pathology, National Clinical Research Centre for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Centre for Cancer, Tianjin Medical University Cancer Institute and Hospital, Huanhu West Road, Hexi District, Tianjin 300060, China
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Cui A, Du Y, Hou C, Zhang L, Sun L, He H. Case Reports: A role of postoperative radiation therapy in completely resected early stage intrathyroid thymic carcinoma: a case report and literature review of the diagnosis and treatment. Front Oncol 2023; 13:1234961. [PMID: 37849817 PMCID: PMC10577444 DOI: 10.3389/fonc.2023.1234961] [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: 06/05/2023] [Accepted: 09/08/2023] [Indexed: 10/19/2023] Open
Abstract
Background Intrathyroid thymic carcinoma (ITTC) is a rare malignant tumor of the thyroid, probably arising from ectopic thymus or branchial pouch remnants. Most of the literature recommended radical resection as the fundamental treatment for ITTC, and postoperative radiation appears to be able to reduce the recurrence rate in patients with advanced ITTC. However, the issue of adjuvant radiotherapy in completely resected early-stage ITTC has been controversial. Case presentation Here, we reported a new case of early-stage ITTC that treated with total thyroidectomy and the right central neck dissection. Postoperative external beam radiation therapy (50.0 Gy/25 fractions) was given to the thyroid bed and bilateral cervical lymph node area since the tumor involved part of the sternal thyroid muscle. At 4-year follow-up after completion of radiotherapy, she is without evidence of locally recurrent or distant disease. Conclusion Since there are no current guidelines for early-stage ITTC, in combination with this case and previous literature, we may suggest routine adjuvant radiotherapy should be considered in patients with incompletely resected tumors and extraparenchymal extension of ITTC. Moreover, we summarized comprehensive and advanced diagnosis, treatment, prognosis of ITTC and comparison between ITTC, primary squamous cell carcinoma of thyroid gland, differentiated thyroid cancer, and anaplastic thyroid cancer.
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Affiliation(s)
- Ailin Cui
- Cancer Center, Department of Ultrasound Medicine, Zhejiang Provincial People’s Hospital (Affiliated People’s Hospital), Hangzhou Medical College, Hangzhou, Zhejiang, China
| | - Yaoqiang Du
- Laboratory Medicine Center, Department of Transfusion Medicine, Zhejiang Provincial People’s Hospital (Affiliated People’s Hospital), Hangzhou Medical College, Hangzhou, Zhejiang, China
| | - Chunjie Hou
- Cancer Center, Department of Ultrasound Medicine, Zhejiang Provincial People’s Hospital (Affiliated People’s Hospital), Hangzhou Medical College, Hangzhou, Zhejiang, China
- Key Laboratory of Endocrine Gland Diseases of Zhejiang Province, Hangzhou, Zhejiang, China
| | - Lin Zhang
- Department of Ultrasound, Hangzhou Children’s Hospital, Hangzhou Children’s Hospital of Wenzhou Medical University, Hangzhou, Zhejiang, China
| | - Litao Sun
- Cancer Center, Department of Ultrasound Medicine, Zhejiang Provincial People’s Hospital (Affiliated People’s Hospital), Hangzhou Medical College, Hangzhou, Zhejiang, China
| | - Hongfeng He
- Cancer Center, Department of Ultrasound Medicine, Zhejiang Provincial People’s Hospital (Affiliated People’s Hospital), Hangzhou Medical College, Hangzhou, Zhejiang, China
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Zhao Q, Bian X. Two cases of concurrent carcinoma showing thymus-like differentiation (CASTLE) coexisting with papillary thyroid carcinoma. J Surg Case Rep 2023; 2023:rjad527. [PMID: 37771882 PMCID: PMC10532264 DOI: 10.1093/jscr/rjad527] [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: 07/09/2023] [Accepted: 09/04/2023] [Indexed: 09/30/2023] Open
Abstract
Carcinoma showing thymus-like differentiation (CASTLE), which emerges within the thyroid gland or the adjacent soft tissues of the neck, is a rare malignant neoplasm found globally. The occurrence of CASTLE in conjunction with papillary thyroid carcinoma is an even more infrequent phenomenon. The ensuing sections elaborate upon the clinical attributes characteristic of CASTLE.
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Affiliation(s)
- Qi Zhao
- Department of Thyroid Surgery, China–Japan Union Hospital of Jilin University, Jilin Provincial Key Laboratory of Surgical Translational Medicine, Changchun 130033, China
| | - Xuehai Bian
- Department of Thyroid Surgery, China–Japan Union Hospital of Jilin University, Jilin Provincial Key Laboratory of Surgical Translational Medicine, Changchun 130033, China
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Liang J, Huang M, Huang H, Li L, Luo H, Mao W, Gao S, Xu H. Intrathyroidal Thymic Carcinoma: A Retrospective Case Series Study. EAR, NOSE & THROAT JOURNAL 2023; 102:584-589. [PMID: 36408572 DOI: 10.1177/01455613221141225] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 09/09/2023] Open
Abstract
OBJECTIVE Intrathyroidal thymic carcinoma (ITC) is a rare low-grade malignant thyroid tumor. There is neither sufficient understanding of this tumor nor its clinical treatment. This study is to explore the clinicopathological features, treatment, and prognosis of ITC and thereby provide a reference for the diagnosis and treatment of the disease. METHODS The clinical, pathological, therapeutic, and prognostic data of 13 patients with ITC were retrospectively analyzed. RESULTS The case series comprised 7 males and 6 females, with an average age of 51.9 ± 10.1 years. After surgical resection, all patients received post-operative neck radiotherapy at dosages of 60-66 Gy. Five patients with level VI lymph node metastasis additionally received 6 courses of cisplatin chemotherapy. All patients were followed-up for 21-132 months (median = 66 months), and all of them survived without recurrence or metastasis. CONCLUSIONS The diagnosis of ITC depends mainly on pathological and immunohistochemical results, particularly CD5 positive staining. Surgical resection is the preferred primary treatment modality which can be supplemented with radiotherapy and chemotherapy to reduce the risk of recurrence and metastasis.
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Affiliation(s)
- Jinhui Liang
- Department of Otorhinolaryngology and Head & Neck Surgery, Hanzhong Central Hospital, Hanzhong, China
| | - Mei Huang
- Department of Otorhinolaryngology and Head & Neck Surgery, Hanzhong Central Hospital, Hanzhong, China
| | - Helang Huang
- Department of Otorhinolaryngology and Head & Neck Surgery, Hanzhong Central Hospital, Hanzhong, China
| | - Li Li
- Department of Otorhinolaryngology and Head & Neck Surgery, Hanzhong Central Hospital, Hanzhong, China
| | - Hailin Luo
- Department of Otorhinolaryngology and Head & Neck Surgery, Hanzhong Central Hospital, Hanzhong, China
| | - Weidong Mao
- Department of Pathology, Hanzhong Central Hospital, Hanzhong, China
| | - Shan Gao
- Department of Radiotherapy, Hanzhong Central Hospital, Hanzhong, China
| | - Haoxiang Xu
- Department of Dermatology, Plastic Surgery Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
- Jiangsu Key Laboratory of Molecular Biology for Skin Diseases and STIs, Chinese Academy of Medical Science & Peking Union Medical College, Nanjing, China
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Lu D, Zhang Y, Zhao R, Zhou E, Xue X, Li C, Huang S, Chen X. Spindle Epithelial Tumor With Thymus-Like Differentiation of Thyroid (SETTLE): A Case Report and Literature Review. EAR, NOSE & THROAT JOURNAL 2023:1455613231171826. [PMID: 37247430 DOI: 10.1177/01455613231171826] [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: 05/31/2023] Open
Abstract
Spindle epithelial tumor with thymus-like differentiation of thyroid (SETTLE) is very rare neoplasm with 2 cellular forms, epithelial cell and spindle cell, and most reported cases have been in young people. An 11-year-old boy presented with painless swelling of the right neck lasting for more than 2 months. A tumor size measuring approximately 3 × 3 cm was resected, and intraoperative frozen pathology suggested a spindle cell tumor, which was confirmed as SETTLE by immunohistochemical staining and external hospital consultation. The immunohistochemical staining profile of the resected tumor tissue was as follows: cytokeratin (CK) (+), smooth muscle actin (weak+), vimentin (+), CK7 (focal+), B-cell lymphoma 2 (partial+), CD99 (-), calcitonin (+), galectin-3 (+), CK19 (+), and Ki-67 (10%+). Ultrasound at 1-year postoperative follow-up revealed no local recurrence of the lesion or lymph node metastasis in the thyroid gland. We summarized the disease characteristics of SETTLE among 6 cases reported to date and found that SETTLE is associated with a good prognosis and low postoperative recurrence rate. Thus, for this type of malignant thyroid tumor, diagnosis depends mainly on postoperative pathology and immunohistochemical staining and simple surgical resection is recommended.
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Affiliation(s)
- Dan Lu
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
| | - Yi Zhang
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
| | - Runyu Zhao
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
| | - Enhui Zhou
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
| | - Xiaocheng Xue
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
| | - Can Li
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
| | - Shuixian Huang
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
| | - Xiaoping Chen
- Postgraduate Training Base at Shanghai Gongli Hospital, Ningxia Medical University, Shanghai, China
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Li J, Xiang R, Li Y, Liao Q, Liu Y. Intrathyroid thymic carcinoma: clinicopathological features and whole exome sequencing analysis. Virchows Arch 2023; 482:813-822. [PMID: 37016248 DOI: 10.1007/s00428-023-03536-3] [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: 11/20/2022] [Revised: 03/23/2023] [Accepted: 03/24/2023] [Indexed: 04/06/2023]
Abstract
Intrathyroid thymic carcinoma (ITC) is a rare malignant tumour. We present nine cases of ITC that were analysed by immunohistochemical staining, of which five were analysed using whole exome sequencing (WES). These cases included six women and three men with an age range of 31-66 years. The average postoperative follow-up term was 37.8 months (range, 7-95 months), and all patients survived well except for one case with lung metastasis. Microscopically, ITC showed solid islands of tumour cells separated by fibrous connective tissue containing lymphocytes and other inflammatory cells. Tumour cells strongly expressed Ckpan (AE1/AE3), P63, and CD117. And all cases but one were positive for CD5. The median value of Ki-67 was 32% (range 10-60%). We observed partial positivity of Syn and CgA in only one case. ITC shares morphological and immunohistochemical similarities with thymic squamous cell carcinoma. In situ hybridization of EBER showed negative results. All cases were microsatellite stable, and the tumour mutational burden of the 5 cases was all < 1 mutations/Mb. WES showed higher mutation rates for N4BP1 (2/5), and many genetic alterations were related to the NF-kB signalling pathway, which is crucial for insight into the molecular mechanisms of the occurrence and development of ITC.
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Affiliation(s)
- Jiayu Li
- Department of Pathology, Sichuan Clinical Research Center for Cancer, Sichuan Cancer Hospital & Institute, Sichuan Cancer Center, Affiliated Cancer Hospital of University of Electronic Science and Technology of China, No.55, Section 4, Renmin South Road, Chengdu, 610041, China
| | - Run Xiang
- Department of Thoracic Surgery, Sichuan Clinical Research Center for Cancer, Sichuan Cancer Hospital & Institute, Sichuan Cancer Center, Affiliated Cancer Hospital of University of Electronic Science and Technology of China, Chengdu, China
| | - Yunzhu Li
- Department of Pathology, Sichuan Clinical Research Center for Cancer, Sichuan Cancer Hospital & Institute, Sichuan Cancer Center, Affiliated Cancer Hospital of University of Electronic Science and Technology of China, No.55, Section 4, Renmin South Road, Chengdu, 610041, China
| | - Qiong Liao
- Department of Pathology, Sichuan Clinical Research Center for Cancer, Sichuan Cancer Hospital & Institute, Sichuan Cancer Center, Affiliated Cancer Hospital of University of Electronic Science and Technology of China, No.55, Section 4, Renmin South Road, Chengdu, 610041, China.
| | - Yang Liu
- Department of Pathology, Sichuan Clinical Research Center for Cancer, Sichuan Cancer Hospital & Institute, Sichuan Cancer Center, Affiliated Cancer Hospital of University of Electronic Science and Technology of China, No.55, Section 4, Renmin South Road, Chengdu, 610041, China.
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12
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Uchiyama T, Terada C, Tachibana Y, Nishiura H, Takeda M, Fujii T, Kimura T, Tsutsumi M, Ohbayashi C. Carcinoma showing thymus-like differentiation of the parotid gland: The brief report of cytomorphology and review of the literature. Diagn Cytopathol 2023; 51:E98-E104. [PMID: 36484759 DOI: 10.1002/dc.25088] [Citation(s) in RCA: 4] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/14/2022] [Revised: 11/28/2022] [Accepted: 11/29/2022] [Indexed: 12/13/2022]
Abstract
Here, we report an extremely rare case of carcinoma with thymus-like differentiation (CASTLE) of the parotid gland. CASTLE is a rare malignant epithelial tumor with thymic epithelial differentiation that arises in the thyroid gland or perithyroidal soft tissue. CASTLE of salivary gland origin is rare, with only nine published case reports to date (reported as "CASTLE" or "thymic carcinoma"). It is critical to diagnose salivary gland tumors using fine needle aspiration cytology. However, this tumor is rare, and there have been few studies on its cytomorphological features. Therefore, it is important to understand the cytological diagnostic characteristics of CASTLE. Herein, we review the cytological features and diagnostic characteristics of salivary gland CASTLE. We also report the genotype results obtained using targeted exome sequencing, which we analyzed with DNA extracted from formalin-fixed paraffin-embedded tissue.
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Affiliation(s)
- Tomoko Uchiyama
- Department of Diagnostic Pathology, Nara Medical University, Kashihara, Japan
| | - Chiyoko Terada
- Department of Diagnostic Pathology, Nara Medical University, Kashihara, Japan
| | - Yuma Tachibana
- Department of Clinical Technology, Yamatotakada Municipal Hospital, Yamatotakada, Japan
| | - Hirokazu Nishiura
- Department of Clinical Technology, Yamatotakada Municipal Hospital, Yamatotakada, Japan
| | - Maiko Takeda
- Department of Diagnostic Pathology, Nara Medical University, Kashihara, Japan
| | - Tomomi Fujii
- Department of Diagnostic Pathology, Nara Medical University, Kashihara, Japan
| | - Takahiro Kimura
- Department of Otolaryngology-Head and Neck Surgery, Nara Medical University, Kashihara, Japan
| | - Masahiro Tsutsumi
- Department of Clinical Technology, Yamatotakada Municipal Hospital, Yamatotakada, Japan
| | - Chiho Ohbayashi
- Department of Diagnostic Pathology, Nara Medical University, Kashihara, Japan
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Zhao Y, Liu J. Case report: Thymoid differentiated carcinoma of thyroid: Two cases. Front Surg 2023; 10:1112315. [PMID: 37181600 PMCID: PMC10174443 DOI: 10.3389/fsurg.2023.1112315] [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: 12/02/2022] [Accepted: 03/29/2023] [Indexed: 05/16/2023] Open
Abstract
Objective Thymoid carcinoma of the thyroid gland is a rare thyroid tumor, which is often presented in case reports. Methods The clinical data of two patients with thymic carcinoma of the thyroid gland were retrospectively reviewed. Results Case 1: a middle-aged woman who was admitted to the hospital because of "progressive enlargement of the anterior cervical mass for 8 months." Color Doppler ultrasound and CT showed malignant tumor with high possibility of bilateral cervical lymph node metastasis. Total thyroidectomy and bilateral central cervical lymph node dissection were performed. Lymph node biopsy showed the metastasis of small cell undifferentiated thyroid carcinoma. Because the biopsy pathological result was not consistent with the pathology of the primary lesion, immunohistochemistry was performed again, and the final diagnosis was thymic carcinoma in the thyroid gland. Case 2: the patient was an elderly man who was admitted to the hospital due to hoarseness for half a month. During the operation, the tumor invaded the trachea, esophagus, internal jugular vein, common carotid artery, and surrounding tissues. Palliative resection of the tumor was performed. The tumor postoperative pathology suggested thymoid carcinoma of the thyroid gland. It recurred and compressed the trachea 4 months after the operation, resulting in dyspnea of the patient, and finally tracheotomy was performed to alleviate the symptoms. Conclusion Case 1 showed multiple differences in pathological diagnosis, suggesting that the lack of specific imaging and clinical manifestations of thymoid-differentiated thyroid carcinoma made the diagnosis so difficult. Case 2 progressed rapidly, suggesting that thymoid-differentiated thyroid carcinoma was not always inert, and the treatment and follow-up should follow the principle of individualization.
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Ito Y, Miyauchi A, Hirokawa M, Kihara M, Onoda N, Miya A. Clinicopathological features and outcomes of intrathyroidal thymic carcinoma: a single institution study. Endocr J 2022; 69:1351-1356. [PMID: 35768280 DOI: 10.1507/endocrj.ej22-0141] [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] [Indexed: 11/23/2022] Open
Abstract
Intrathyroidal thymic carcinoma (ITTC) is a rare malignancy of the thyroid. It is thought to originate from ectopic thymic tissue or embryonic thymic rest, in, or adjacent to, the thyroid. We analyzed the backgrounds, clinicopathological features, and prognosis of 20 patients with ITTC, treated at our hospital. Thirteen of the 15 patients (86%) who underwent ultrasonography were diagnosed as malignant, based on imaging findings. 16 of the 17 patients (93%) who underwent cytology, were diagnosed or suspected to be malignant. Locally curative surgery (thyroidectomy and lymph node dissection) was performed for 19 patients. Large tumor size (>4 cm) was positively related to pathological node metastasis (p = 0.0389). Fourteen patients, including nine Ex-positive patients, underwent adjuvant external beam radiotherapy (EBRT) of the neck after surgery. Two patients showed recurrence of thyroid bed after and neither of them underwent adjuvant EBRT after surgery. Two patients who underwent EBRT showed recurrences of the lateral nodes (level V and level II), but they were easily dissected by re-operation. Ten- and 20-year local recurrence-free survival rates were 84.9% and 60.6%, respectively. To date, four patients showed distant recurrence, and 10- and 20-year distant recurrence-free survival rates were 75.0% and 75.0%, respectively. Our findings indicate that 1) the prognosis of ITTC is generally favorable, and 2) large tumor size is significantly related to lymph node metastasis. Two patients showing recurrence of the central region did not undergo EBRT; thus, further comparative studies are desirable to elucidate whether EBRT can prevent significant local recurrence.
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Affiliation(s)
- Yasuhiro Ito
- Department of Surgery, Kuma Hospital, Hyogo 650-0011, Japan
| | - Akira Miyauchi
- Department of Surgery, Kuma Hospital, Hyogo 650-0011, Japan
| | | | - Minoru Kihara
- Department of Surgery, Kuma Hospital, Hyogo 650-0011, Japan
| | - Naoyoshi Onoda
- Department of Surgery, Kuma Hospital, Hyogo 650-0011, Japan
| | - Akihiro Miya
- Department of Surgery, Kuma Hospital, Hyogo 650-0011, Japan
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15
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Kuroki M, Shibata H, Iinuma R, Okuda H, Ohashi T, Ogawa T, Horikawa Y. A Case of Thyroid Carcinoma Showing Thymus-Like Differentiation With Breast Cancer Susceptibility Gene 2 Mutation: A Case Report and Literature Review. Cureus 2022; 14:e30655. [DOI: 10.7759/cureus.30655] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 10/25/2022] [Indexed: 11/07/2022] Open
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16
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Stanciu M, Ristea RP, Popescu M, Vasile CM, Popa FL. Thyroid Carcinoma Showing Thymus-like Differentiation (CASTLE): A Case Report. Life (Basel) 2022; 12:life12091314. [PMID: 36143350 PMCID: PMC9503371 DOI: 10.3390/life12091314] [Citation(s) in RCA: 8] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/01/2022] [Revised: 08/22/2022] [Accepted: 08/24/2022] [Indexed: 11/16/2022] Open
Abstract
Background and Objectives: Carcinoma showing thymus-like differentiation (CASTLE) is a low-grade thyroid carcinoma, with an indolent clinical course and usually a favorable prognosis. The clinical and imagistic features are not specific for CASTLE but similar to other malignant lesions of the thyroid. Definite diagnosis is based on an immunohistochemical examination, as this carcinoma shows positive CD5 immunoreactivity when compared to other aggressive thyroid carcinomas. Case presentation: The main focus of this study is to outline a rare case of CASTLE compressing the trachea in a 50-year-old female patient who was initially diagnosed with undifferentiated thyroid carcinoma, for which she underwent unsuccessful surgery, as well as postoperative radiotherapy and chemotherapy. After receiving a second medical opinion, the patient underwent a challenging radical resection consisting in total thyroidectomy and central neck dissection, with no local recurrence after 6 months and 2 years of follow-up and negative metastatic follow-up. The correct diagnosis has been established based on pathological and immunohistochemical examinations. Conclusions: In summary, the diagnosis of CASTLE is difficult and requires an experienced histological analysis and CD5 immunoreactivity. Lack of metastasis, complete removal of the tumor, and a low degree of tumor infiltration into nearby structures are all associated with better long-term survival.
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Affiliation(s)
- Mihaela Stanciu
- Department of Endocrinology, Faculty of Medicine, Lucian Blaga University of Sibiu, 550169 Sibiu, Romania
| | - Ruxandra Paula Ristea
- Department of Endocrinology, County Clinical Emergency Hospital of Sibiu, 550245 Sibiu, Romania
| | - Mihaela Popescu
- Department of Endocrinology, University of Medicine and Pharmacy of Craiova, 200349 Craiova, Romania
- Correspondence: (M.P.); (C.M.V.)
| | - Corina Maria Vasile
- Department of Pediatric Cardiology, “Marie Curie” Emergency Children’s Hospital, 041451 Bucharest, Romania
- Correspondence: (M.P.); (C.M.V.)
| | - Florina Ligia Popa
- Department of Physical Medicine and Rehabilitation, Faculty of Medicine, Lucian Blaga University of Sibiu, 550169 Sibiu, Romania
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17
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Yuan Y, Ke C, Zhang G, Zhang J, Li Q. Case report and literature review: thyroid carcinoma showing intrathyroid thymic carcinoma. Front Oncol 2022; 12:923683. [PMID: 35992819 PMCID: PMC9389067 DOI: 10.3389/fonc.2022.923683] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/19/2022] [Accepted: 07/07/2022] [Indexed: 11/13/2022] Open
Abstract
Background Intrathyroid thymic carcinoma (ITTC) is a rare malignancy of the thyroid gland with histological and immunophenotypic resemblance to thymic carcinoma. Surgery combined with adjuvant radiotherapy improves the survival of patients with ITTC. However, for patients with extensive metastases, there is currently no effective treatment. Chemotherapy is an option but has not demonstrated improved patient survival. Methods and results A female patient presented with metastases to the pleura, lung, and bone 16 years after surgery for ITTC. As radiotherapy and chemotherapy failed to control the recurrent disease, lenvatinib treatment was initiated. After 3 months, positron emission tomography/computed tomography showed a substantial reduction of all metastatic lesions and decreased tumor metabolism. The patient continues to receive lenvatinib and remains well and symptom-free. Conclusion For patients with ITTC who have progressive, life-threatening metastases, lenvatinib represents a valuable salvage therapy that may offer a sustained reduction in tumor burden and maintenance of quality of life.
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Affiliation(s)
- Ye Yuan
- Department of Gastroenterology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
| | - Changshu Ke
- Department of Pathology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
| | - Guopeng Zhang
- Department of Nuclear Medicine, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
| | - Jun Zhang
- Department of Geriatrics, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
| | - Qianxia Li
- Department of Oncology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
- *Correspondence: Qianxia Li,
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18
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Jiang L, Zheng W, Chen C. Genomic variation associated with carcinoma showing thymus-like elements (CASTLE) in thyroid gland. Laryngoscope Investig Otolaryngol 2022; 7:894-900. [PMID: 35734076 PMCID: PMC9194970 DOI: 10.1002/lio2.805] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/08/2022] [Revised: 03/26/2022] [Accepted: 04/01/2022] [Indexed: 11/07/2022] Open
Abstract
Background Carcinoma showing thymus-like elements (CASTLE) is a rare kind of malignant tumor of thyroid gland. The genetic mutation characteristics of CASTLE are not clear. Methods We retrospectively analyzed seven patients diagnosed as CASTLE tumor in our hospital, and performed whole exome sequencing (WES) in five cases to analyze the genomic variation of CASTLE in thyroid gland. Results The diagnosis of CASTLE was confirmed by histopathological and immunohistochemical results. Immunohistochemical staining showed that cell membranes of tumor samples in all cases were moderately to strongly positive for CD5 and CD117. WES presented a large number of single nucleotide variants (SNVs), insertions and deletions (InDel), and copy number variations (CNVs). By comparing with the TCGA database, we found novel mutations in significantly mutated genes such as FBXL16, PAQR7, LEFTY1, UBA52, and FLNA, as well as in potential disease-related driver genes such as MLLT10, FLNA, CYLD, HLA-B, KMT2D, SFPQ, MUC16, EEF2, and KMT2C. Conclusions CASTLE tumors contain unique tumor driver gene mutations. The information about mutations in several novel genes obtained in this study may contribute to unraveling the molecular mechanisms responsible for the emergence of thyroid CASTLE tumors and help formulating possible in-roads for treatment.
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Affiliation(s)
- Lin Jiang
- The Cancer Hospital of the University of Chinese Academy of Sciences (Zhejiang Cancer Hospital)Institute of Basic Medicine and Cancer (IBMC), Chinese Academy of SciencesHangzhouChina
- Key Laboratory of Head and Neck Cancer Translational Research of Zhejiang ProvinceHangzhouChina
| | - Wei‐Hui Zheng
- The Cancer Hospital of the University of Chinese Academy of Sciences (Zhejiang Cancer Hospital)Institute of Basic Medicine and Cancer (IBMC), Chinese Academy of SciencesHangzhouChina
- Key Laboratory of Head and Neck Cancer Translational Research of Zhejiang ProvinceHangzhouChina
| | - Chao Chen
- The Cancer Hospital of the University of Chinese Academy of Sciences (Zhejiang Cancer Hospital)Institute of Basic Medicine and Cancer (IBMC), Chinese Academy of SciencesHangzhouChina
- Key Laboratory of Head and Neck Cancer Translational Research of Zhejiang ProvinceHangzhouChina
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19
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Baloch ZW, Asa SL, Barletta JA, Ghossein RA, Juhlin CC, Jung CK, LiVolsi VA, Papotti MG, Sobrinho-Simões M, Tallini G, Mete O. Overview of the 2022 WHO Classification of Thyroid Neoplasms. Endocr Pathol 2022; 33:27-63. [PMID: 35288841 DOI: 10.1007/s12022-022-09707-3] [Citation(s) in RCA: 535] [Impact Index Per Article: 178.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 01/27/2022] [Indexed: 12/15/2022]
Abstract
This review summarizes the changes in the 5th edition of the WHO Classification of Endocrine and Neuroendocrine Tumors that relate to the thyroid gland. The new classification has divided thyroid tumors into several new categories that allow for a clearer understanding of the cell of origin, pathologic features (cytopathology and histopathology), molecular classification, and biological behavior. Follicular cell-derived tumors constitute the majority of thyroid neoplasms. In this new classification, they are divided into benign, low-risk, and malignant neoplasms. Benign tumors include not only follicular adenoma but also variants of adenoma that are of diagnostic and clinical significance, including the ones with papillary architecture, which are often hyperfunctional and oncocytic adenomas. For the first time, there is a detailed account of the multifocal hyperplastic/neoplastic lesions that commonly occur in the clinical setting of multinodular goiter; the term thyroid follicular nodular disease (FND) achieved consensus as the best to describe this enigmatic entity. Low-risk follicular cell-derived neoplasms include non-invasive follicular thyroid neoplasm with papillary-like nuclear features (NIFTP), thyroid tumors of uncertain malignant potential, and hyalinizing trabecular tumor. Malignant follicular cell-derived neoplasms are stratified based on molecular profiles and aggressiveness. Papillary thyroid carcinomas (PTCs), with many morphological subtypes, represent the BRAF-like malignancies, whereas invasive encapsulated follicular variant PTC and follicular thyroid carcinoma represent the RAS-like malignancies. This new classification requires detailed subtyping of papillary microcarcinomas similar to their counterparts that exceed 1.0 cm and recommends not designating them as a subtype of PTC. The criteria of the tall cell subtype of PTC have been revisited. Cribriform-morular thyroid carcinoma is no longer classified as a subtype of PTC. The term "Hürthle cell" is discouraged, since it is a misnomer. Oncocytic carcinoma is discussed as a distinct entity with the clear recognition that it refers to oncocytic follicular cell-derived neoplasms (composed of > 75% oncocytic cells) that lack characteristic nuclear features of PTC (those would be oncocytic PTCs) and high-grade features (necrosis and ≥ 5 mitoses per 2 mm2). High-grade follicular cell-derived malignancies now include both the traditional poorly differentiated carcinoma as well as high-grade differentiated thyroid carcinomas, since both are characterized by increased mitotic activity and tumor necrosis without anaplastic histology and clinically behave in a similar manner. Anaplastic thyroid carcinoma remains the most undifferentiated form; squamous cell carcinoma of the thyroid is now considered as a subtype of anaplastic carcinoma. Medullary thyroid carcinomas derived from thyroid C cells retain their distinct section, and there is a separate section for mixed tumors composed of both C cells and any follicular cell-derived malignancy. A grading system for medullary thyroid carcinomas is also introduced based on mitotic count, tumor necrosis, and Ki67 labeling index. A number of unusual neoplasms that occur in the thyroid have been placed into new sections based on their cytogenesis. Mucoepidermoid carcinoma and secretory carcinoma of the salivary gland type are now included in one section classified as "salivary gland-type carcinomas of the thyroid." Thymomas, thymic carcinomas and spindle epithelial tumor with thymus-like elements are classified as "thymic tumors within the thyroid." There remain several tumors whose cell lineage is unclear, and they are listed as such; these include sclerosing mucoepidermoid carcinoma with eosinophilia and cribriform-morular thyroid carcinoma. Another important addition is thyroblastoma, an unusual embryonal tumor associated with DICER1 mutations. As in all the WHO books in the 5th edition, mesenchymal and stromal tumors, hematolymphoid neoplasms, germ cell tumors, and metastatic malignancies are discussed separately. The current classification also emphasizes the value of biomarkers that may aid diagnosis and provide prognostic information.
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Affiliation(s)
- Zubair W Baloch
- Department of Pathology & Laboratory Medicine, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
| | - Sylvia L Asa
- Department of Pathology, University Hospitals Cleveland Medical Center, Case Western Reserve University, Cleveland, OH, USA
| | - Justine A Barletta
- Department of Pathology, Brigham and Women's Hospital, Harvard Medical School, Boston, MA, USA
| | - Ronald A Ghossein
- Department of Pathology, Memorial Sloan Kettering Cancer Center, New York, NY, USA
| | - C Christofer Juhlin
- Department of Oncology-Pathology, Karolinska Institutet, Stockholm, Sweden
- Department of Pathology and Cancer Diagnostics, Karolinska University Hospital, Stockholm, Sweden
| | - Chan Kwon Jung
- Department of Hospital Pathology, College of Medicine, The Catholic University of Korea, Seoul, Republic of Korea
| | - Virginia A LiVolsi
- Department of Pathology & Laboratory Medicine, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA
| | | | - Manuel Sobrinho-Simões
- Department of Pathology, Institute of Molecular Pathology and Immunology, IPATIMUP, University of Porto, Porto, Portugal
| | - Giovanni Tallini
- Department of Experimental, Diagnostic and Specialty Medicine, University of Bologna, Bologna, Italy
- IRCCS Azienda Ospedaliero-Universitaria Di Bologna, Bologna, Italy
| | - Ozgur Mete
- Department of Pathology, University Health Network, University of Toronto, Toronto, ON, Canada
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Kimura T, Enomoto K, Kono M, Hiraoka M, Takeda S, Kumashiro N, Hirayama S, Kimura E, Tamagawa S, Ohtani M, Murata SI, Hotomi M. A case of concurrent occurrence of carcinoma showing thymus-like differentiation and follicular variant of papillary thyroid cancer in the same thyroid. J Surg Case Rep 2022; 2022:rjab570. [PMID: 35047168 PMCID: PMC8760851 DOI: 10.1093/jscr/rjab570] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/25/2021] [Revised: 11/18/2021] [Accepted: 11/22/2021] [Indexed: 11/12/2022] Open
Abstract
Carcinoma showing thymus-like differentiation (CASTLE) is a rare thyroid cancer. This is the first report of a case of concurrent occurrence of CASTLE with papillary thyroid carcinoma (PTC). A 66-year-old male patient had hoarseness with right vocal cord paralysis. Ultrasonography revealed a hypoechoic nodule in the inferior pole of the right thyroid lobe. Ultrasound-guided fine-needle aspiration cytology suggested differentiated thyroid cancer. The patient underwent total thyroidectomy with neck dissection. Pathological examination revealed two different thyroid cancers: a CASTLE and a follicular variant of PTC. Postoperative radiation therapy was performed. The patient was still alive after 5 year following the initial treatment without evidence of recurrence. The oncological management of patients with concurrent occurrence of different thyroid cancers should consider the biological behavior of both tumors.
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Affiliation(s)
- Takahito Kimura
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Keisuke Enomoto
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Masamitsu Kono
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Masanobu Hiraoka
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Saori Takeda
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Naoko Kumashiro
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Shun Hirayama
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Eri Kimura
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Shunji Tamagawa
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Makiko Ohtani
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Shin-Ichi Murata
- Department of Diagnostic Pathology, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
| | - Muneki Hotomi
- Department of Otorhinolaryngology-Head and Neck Surgery, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-8509, Japan
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21
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Yanhai WMD, Hua YMD, Hanqing LMD, Xiaoli LMD, Luying LBS, Pingting ZBS. Ultrasonographic Features of Intrathyroidal Thymic Carcinoma: Review and Analysis of 10 Cases. ADVANCED ULTRASOUND IN DIAGNOSIS AND THERAPY 2022. [DOI: 10.37015/audt.2022.220013] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
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22
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Yamamoto H, Kusafuka K, Nozaki Y, Iwasaki T, Nogami M, Hongo T, Yasumatsu R, Oda Y. Carcinoma showing thymus-like differentiation (CASTLE) of the salivary gland: Report of 2 cases of a hitherto under-recognized extrathyroid counterpart. Pathol Res Pract 2021; 227:153646. [PMID: 34649052 DOI: 10.1016/j.prp.2021.153646] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/04/2021] [Revised: 10/03/2021] [Accepted: 10/04/2021] [Indexed: 10/20/2022]
Abstract
Carcinoma showing thymus-like differentiation (CASTLE) outside the thyroid gland is extremely rare. Here we report two cases of CASTLE of the major salivary gland. The tumors occurred in the parotid gland of a 31-year-old female (Case 1) and in the submandibular gland of a 40-year-old female (Case 2). Both tumors showed a lobulated growth pattern, and were histologically composed of a nested or sheet-like proliferation of carcinoma cells with round- to oval-shaped nuclei, distinct nucleoli and pale eosinophilic cytoplasm, accompanied by various degrees of lymphocytic infiltration. Immunohistochemical staining revealed that the tumors were positive for pan-cytokeratin, p40, CD5, CD117 and bcl-2. In addition, PD-L1 expression was seen in 10-90% of tumor cells. After the initial surgery, Case 1 remained tumor-free for 20 months, while Case 2 suffered lymph node recurrence at 4 months, followed by lung metastasis, which was treated with chemoradiotherapy and anti-PD-1 immune checkpoint inhibitor, resulting in a partial response. The present findings indicate that an extrathyroid counterpart of CASTLE can occur as a primary salivary gland neoplasm. Salivary CASTLEs seem to show a wide range of biological behavior, and long-term follow-up may be needed. Immune checkpoint inhibitor targeting PD-1 might become a promising treatment option in patients with CASTLE; however, further study with a larger number of cases is necessary to establish the optimal therapeutic strategy and prognostic factors for this rare cancer.
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Affiliation(s)
- Hidetaka Yamamoto
- Department of Anatomic Pathology, Kyushu University, Graduate School of Medical Sciences, Fukuoka, Japan; Division of Pathology, Kyushu University Hospital, Fukuoka, Japan.
| | | | - Yui Nozaki
- Department of Anatomic Pathology, Kyushu University, Graduate School of Medical Sciences, Fukuoka, Japan; Division of Pathology, Kyushu University Hospital, Fukuoka, Japan
| | - Tomohiro Iwasaki
- Department of Pathology, Shizuoka General Hospital, Shizuoka, Japan
| | - Miwako Nogami
- Division of Pathology, Kyushu University Hospital, Fukuoka, Japan
| | - Takahiro Hongo
- Department of Anatomic Pathology, Kyushu University, Graduate School of Medical Sciences, Fukuoka, Japan
| | - Ryuji Yasumatsu
- Department of Otorhinolaryngology, Kyushu University, Graduate School of Medical Sciences, Fukuoka, Japan
| | - Yoshinao Oda
- Department of Anatomic Pathology, Kyushu University, Graduate School of Medical Sciences, Fukuoka, Japan
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23
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Dang NV, Son LX, Hong NTT, Nhung NTT, Tung NT, Quang LV. Recurrence of carcinoma showing thymus-like differentiation (CASTLE) involving the thyroid gland. Thyroid Res 2021; 14:20. [PMID: 34399813 PMCID: PMC8365953 DOI: 10.1186/s13044-021-00111-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/04/2021] [Accepted: 08/06/2021] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Carcinoma showing thymus-like differentiation (CASTLE) in the thyroid gland is a rare disease with generally a favorable prognosis. Treatment with surgery and adjuvant radiotherapy has been shown to improve local control and long-term survival rates. In this report, we present a case of a recurrent thyroid gland CASTLE and review the literature on the diagnosis and treatment of this disease. CASE PRESENTATION A 60-year-old woman, who was diagnosed with a CASTLE thyroid tumor in 2015, had a total thyroidectomy and was maintained on thyroid hormone replacement (levothyroxine). After 5 years, the patient had a recurrence, in an advanced stage unsuitable for surgery. As the patient declined to undergo radiotherapy, she was followed up without intervention and is currently stable after 15 months. CONCLUSIONS CASTLE is a rare disease, diagnosed based on postoperative pathology and immunohistochemistry analysis, especially upon CD5 marker. In case of relapse, treatment options include surgery and radiotherapy; however conservative management without intervention is an acceptable alternative in some cases.
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Affiliation(s)
- N V Dang
- Department of Oncology, Hanoi Medical University, 01 Ton That Tung Street, Dong Da District, Hanoi, Vietnam. .,Department of Head and Neck Radiation Oncology, Vietnam National Cancer Hospital, Hanoi, Vietnam.
| | - L X Son
- Department of Oncology, Hanoi Medical University, 01 Ton That Tung Street, Dong Da District, Hanoi, Vietnam
| | - N T T Hong
- Department of Radiation Physics, Vietnam National Cancer Hospital, Hanoi, Vietnam
| | - N T T Nhung
- Department of Head and Neck Radiation Oncology, Vietnam National Cancer Hospital, Hanoi, Vietnam
| | - N T Tung
- Department of Head and Neck Radiation Oncology, Vietnam National Cancer Hospital, Hanoi, Vietnam
| | - L V Quang
- Department of Oncology, Hanoi Medical University, 01 Ton That Tung Street, Dong Da District, Hanoi, Vietnam
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24
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Inoperable CASTLE of the thyroid gland treated with radical radiotherapy with complete remission. Strahlenther Onkol 2021; 197:847-853. [PMID: 34160632 DOI: 10.1007/s00066-021-01800-6] [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: 03/26/2021] [Accepted: 05/25/2021] [Indexed: 10/21/2022]
Abstract
Carcinoma showing thymus-like elements (CASTLE) is an extremely rare malignant tumor of the thyroid gland and soft tissues of the neck with favorable prognosis. Histological features of the CASTLE are similar to thymic carcinoma, and it is assumed that it arises from the ectopic thymic tissue or the remnants of branchial pouches. The optimal treatment strategy is still uncertain because of the rarity of the tumor. The mainstay of treatment is surgery. The role of other modalities is unclear. We present a case report of a patient with locally advanced CASTLE of the thyroid gland who was not suitable for surgery and underwent radical radiotherapy with subsequent achievement of complete remission. We also present a literature review.
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25
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Kimura E, Enomoto K, Kono M, Tamagawa S, Takeda S, Kumashiro N, Hirayama S, Kimura T, Hotomi M. A Rare Case of Thyroid Carcinoma Showing Thymus-Like Differentiation in a Young Adult. Case Rep Oncol 2021; 14:671-675. [PMID: 33976651 PMCID: PMC8077592 DOI: 10.1159/000514271] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/14/2020] [Accepted: 01/06/2021] [Indexed: 11/19/2022] Open
Abstract
Thyroid carcinoma showing thymus-like differentiation (CASTLE) is thought to originate from ectopic thymic tissue or remnants of the developing thymus within or adjacent to the thyroid. This case report describes a mass located on the left thyroid of a 28-year-old man. Fine-needle aspiration cytology revealed a number of lymphoid cells without atypia that were similar to those seen in a malignant lymphoma of the thyroid, and surgery was performed. Based on additional histopathological findings, the tumor was finally diagnosed as a CASTLE. It is difficult to diagnose this neoplasm using fine-needle aspiration cytology. However, it is possible to differentially diagnose CASTLE based on its histological features. CD5 is useful for diagnosing CASTLE with immunohistochemical staining.
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Affiliation(s)
- Eri Kimura
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Keisuke Enomoto
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Masamitsu Kono
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Shunji Tamagawa
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Saori Takeda
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Naoko Kumashiro
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Shun Hirayama
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Takahito Kimura
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
| | - Muneki Hotomi
- Department of Otolaryngology-Head and Neck Surgery, Wakayama Medical University, Wakayama, Japan
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26
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Misfeld M, Borger MA, Steinert F. Resection of a Tumor With Thymic-like Differentiation and Reconstruction of the Innominate Artery. Ann Thorac Surg 2021; 112:e99-e100. [PMID: 33444581 DOI: 10.1016/j.athoracsur.2020.12.037] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/10/2020] [Revised: 12/16/2020] [Accepted: 12/26/2020] [Indexed: 10/22/2022]
Abstract
Carcinomas showing thymic-like differentiation (CASTLE tumors) are very rare entities requiring an individualized therapeutic plan. This report describes a case of reconstruction of the innominate artery after radical resection of the tumor in a patient in whom a custom-made cerebral shunt was used for continuous cerebral perfusion.
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Affiliation(s)
- Martin Misfeld
- University Department of Cardiac Surgery, Leipzig Heart Center, Leipzig, Germany; Department of Cardiothoracic Surgery, Royal Prince Alfred Hospital, Sydney, Australia; Institute of Academic Surgery, Royal Prince Alfred Hospital, Sydney, Australia; The Baird Institute of Applied Heart and Lung Surgical Research, Sydney, Australia; Sydney Medical School, University of Sydney, Sydney, Australia.
| | - Michael A Borger
- University Department of Cardiac Surgery, Leipzig Heart Center, Leipzig, Germany
| | - Frank Steinert
- Department of General- and Visceral Surgery, Helios Clinic Schkeuditz, Schkeuditz, Germany
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27
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Whole exome sequencing and establishment of an organoid culture of the carcinoma showing thymus-like differentiation (CASTLE) of the parotid gland. Virchows Arch 2021; 478:1149-1159. [PMID: 33415446 DOI: 10.1007/s00428-020-02981-8] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/16/2020] [Revised: 11/04/2020] [Accepted: 11/25/2020] [Indexed: 10/22/2022]
Abstract
Carcinoma showing thymus-like differentiation (CASTLE) is a rare tumor, especially in the parotid gland. We encountered a CASTLE of the parotid gland and analyzed its clinicopathological features, as well as the genotype using whole exome sequencing (WES). Moreover, we successfully established an organoid culture cell line from the primary tumor tissue. The patient was a 23-year-old woman who underwent superficial parotidectomy with peripheral neck dissection, followed by radiotherapy. Pathologically, the resected specimen showed atypical epithelioid nests and trabeculae with squamous differentiation, separated by thick fibrous septa, accompanied by dense lymphocytes and plasma cell infiltration. Immunohistochemistry revealed that the tumor cells were positive for AE1/AE3, p40, p63, p16, CK5/6, and CD5, and the background lymphocytes were positive for CD5 and CD99. Based on these findings, the tumor was diagnosed as CASTLE. WES uncovered five nonsynonymous and splicing somatic mutations, namely, FREM2 p.Val861Phe, CLK3 p.Phe376Leu, DLGAP1 p.Lys294Asn, NOX1 p.Val165Met, and PSG9 c.430 + 4A > T. Organoid culture cells preserved the histopathological characteristics of the epithelioid component of CASTLE and harbored all five somatic mutations detected in the primary tumor. In conclusion, for the first time to the best of our knowledge, we successfully analyzed a comprehensive genotype and established an organoid culture cell line of a parotid gland CASTLE, which should serve for analyzing the nature of this rare tumor.
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28
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Cameselle-Teijeiro JM, Eloy C, Sobrinho-Simões M. Pitfalls in Challenging Thyroid Tumors: Emphasis on Differential Diagnosis and Ancillary Biomarkers. Endocr Pathol 2020; 31:197-217. [PMID: 32632840 PMCID: PMC7395918 DOI: 10.1007/s12022-020-09638-x] [Citation(s) in RCA: 19] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 07/01/2020] [Indexed: 02/06/2023]
Abstract
Thyroid pathology encompasses a heterogenous group of clinicopathological entities including rare and diagnostically challenging neoplasms. The review is focused on morphological, immunohistochemical, and molecular features of rare thyroid neoplasms that can pose diagnostic problems. The tumors are organized based on growth patterns including thyroid neoplasms with predominantly papillary, follicular, solid, and spindle cell growth pattern, as well as neoplasms with distinct cytological characteristics. A special section is also dedicated to rare thyroid tumors with peculiar patterns including thyroid carcinoma with Ewing family tumor elements and intrathyroidal thymic-related neoplasms.
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Affiliation(s)
- José Manuel Cameselle-Teijeiro
- Department of Pathology, Clinical University Hospital, Health Research Institute of Santiago de Compostela (IDIS), Galician Healthcare Service (SERGAS), Santiago de Compostela, Spain.
- Medical Faculty, University of Santiago de Compostela, Santiago de Compostela, Spain.
- Department of Anatomic Pathology, Clinical University Hospital, Travesía Choupana s/n, 15706, Santiago de Compostela, Spain.
| | - Catarina Eloy
- i3S Instituto de Investigação e Inovação em Saúde, Porto, Portugal
- Institute of Molecular Pathology and Immunology, University of Porto, Porto, Portugal
- Medical Faculty, University of Porto, Porto, Portugal
| | - Manuel Sobrinho-Simões
- i3S Instituto de Investigação e Inovação em Saúde, Porto, Portugal
- Institute of Molecular Pathology and Immunology, University of Porto, Porto, Portugal
- Medical Faculty, University of Porto, Porto, Portugal
- Department of Pathology, Centro Hospitalar S. João, Porto, Portugal
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29
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Tahara I, Oishi N, Mochizuki K, Oyama T, Miyata K, Miyauchi A, Hirokawa M, Katoh R, Kondo T. Identification of Recurrent TERT Promoter Mutations in Intrathyroid Thymic Carcinomas. Endocr Pathol 2020; 31:274-282. [PMID: 32594366 DOI: 10.1007/s12022-020-09635-0] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/10/2023]
Abstract
Intrathyroid thymic carcinoma (ITTC) is a rare malignant neoplasm considered to be a eutopic thymic carcinoma (TC) arising ectopically in the thyroid. Histopathologically, ITTC resembles squamous cell carcinoma of the thymus with positive TC markers such as CD5 and c-KIT. Despite these similar histological findings, ITTC is clinically less aggressive than TC. In this study, we compared clinical, histological, and genetic characteristics of ITTCs and TCs. We collected 9 ITTCs and 8 TCs with their clinicopathological profiles. Immunohistochemistry for CD5, p63, CD117/c-KIT, Ki-67, p53, TTF-1, thyroglobulin, PAX8, EGFR, and PD-L1/CD274 plus in situ hybridization for EBER was performed. We further investigated mutation status of KIT, EGFR, BRAF, and TERT promoter using Sanger sequencing. In our study, ITTCs affected significantly younger patients than TCs. After a mean follow-up of 86 months, all patients with ITTC were alive, while two patients with TC had died. Immunohistochemistry showed ITTCs and TCs had a similar immunophenotype except for EGFR and p53. Genetic analysis did not identify KIT or BRAF mutations in any ITTCs or TCs. EGFR mutations were positive in 11% (1/9) of ITTCs and 25% (2/8) of TCs. Notably, TERT promoter C228T mutation was identified in 22% (2/9) of ITTCs but none of the TCs. There were no significant differences in age, tumor size, or sex between TERT-mutated and TERT-wild-type ITTCs. Collectively, ITTC and TC have similar histopathologic and immunophenotypic features but different clinical outcomes. Recurrent TERT promoter mutation may be a key event related to cancer progression in ITTCs and warrants further investigation.
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Affiliation(s)
- Ippei Tahara
- Department of Pathology, University of Yamanashi, Chuo, Yamanashi, Japan
| | - Naoki Oishi
- Department of Pathology, University of Yamanashi, Chuo, Yamanashi, Japan
| | - Kunio Mochizuki
- Department of Pathology, University of Yamanashi, Chuo, Yamanashi, Japan
| | - Toshio Oyama
- Department of Pathology, Yamanashi Prefectural Central Hospital, Kofu, Yamanashi, Japan
| | - Kazuyuki Miyata
- Department of Pathology, Kofu Municipal Hospital, Kofu, Yamanashi, Japan
| | - Akira Miyauchi
- Department of Surgery, Kuma Hospital, Kobe, Hyogo, Japan
| | | | - Ryohei Katoh
- Department of Pathology, Ito hospital, Tokyo, Japan
| | - Tetsuo Kondo
- Department of Pathology, University of Yamanashi, Chuo, Yamanashi, Japan.
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30
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Ardighieri L, Tomasoni M, Battocchio S, Facchetti F, Maroldi R, Nicolai P, Lombardi D. Carcinoma Showing Thymus-Like Differentiation (CASTLE) Arising in the Sublingual Gland. Int J Surg Pathol 2020; 29:301-307. [PMID: 32672084 DOI: 10.1177/1066896920941604] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/12/2023]
Abstract
Carcinoma showing thymic-like differentiation (CASTLE) is a rare tumor most commonly occurring in the thyroid and soft tissues of the neck. We report the first case of CASTLE occurring in the sublingual gland. The patient, a 35-year-old healthy man, presented with a submucosal lesion located in the anterior right floor of the oral cavity and an ipsilateral neck mass. The lesion had been previously investigated by neck computed tomography and ultrasound-guided fine needle aspiration cytology and diagnosed as metastatic squamous cell carcinoma. After oral cavity magnetic resonance imaging, positron emission tomography, and a non-diriment, fine needle aspiration cytology of the sublingual mass, the patient was treated as affected by a sublingual gland malignancy with removal of primary tumor and neck dissection. Morphological and immunohistochemical findings were diagnostic for primary sublingual gland CASTLE. The patient received adjuvant radiotherapy and is free of disease 2 years after treatment. We describe the pathological features of the lesion and discuss the possible differential diagnoses.
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31
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Jiang L, Zheng WH, Xi Y, Chen C. An extrathyroid CASTLE tumor in the left neck. Oral Oncol 2020; 109:104656. [PMID: 32245700 DOI: 10.1016/j.oraloncology.2020.104656] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/04/2020] [Revised: 03/10/2020] [Accepted: 03/16/2020] [Indexed: 11/16/2022]
Affiliation(s)
- Lin Jiang
- Institute of Cancer and Basic Medicine (ICBM) of Chinese Academy of Sciences, Hangzhou, PR China; Department of Head and Neck Surgery, Cancer Hospital of the University of Chinese Academy of Sciences, Hangzhou, PR China; Department of Head and Neck Surgery, Zhejiang Cancer Hospital, Hangzhou, PR China
| | - Wei-Hui Zheng
- Institute of Cancer and Basic Medicine (ICBM) of Chinese Academy of Sciences, Hangzhou, PR China; Department of Head and Neck Surgery, Cancer Hospital of the University of Chinese Academy of Sciences, Hangzhou, PR China; Department of Head and Neck Surgery, Zhejiang Cancer Hospital, Hangzhou, PR China
| | - Yun Xi
- Institute of Cancer and Basic Medicine (ICBM) of Chinese Academy of Sciences, Hangzhou, PR China; Department of Pathology, Cancer Hospital of the University of Chinese Academy of Sciences, Hangzhou, PR China; Department of Pathology, Zhejiang Cancer Hospital, Hangzhou, PR China
| | - Chao Chen
- Institute of Cancer and Basic Medicine (ICBM) of Chinese Academy of Sciences, Hangzhou, PR China; Department of Head and Neck Surgery, Cancer Hospital of the University of Chinese Academy of Sciences, Hangzhou, PR China; Department of Head and Neck Surgery, Zhejiang Cancer Hospital, Hangzhou, PR China.
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32
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Fung ACH, Tsang JS, Lang BHH. Thyroid Carcinoma Showing Thymus-Like Differentiation (CASTLE) with Tracheal Invasion: A Case Report. AMERICAN JOURNAL OF CASE REPORTS 2019; 20:1845-1851. [PMID: 31819030 PMCID: PMC6913266 DOI: 10.12659/ajcr.919208] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/09/2022]
Abstract
Patient: Male, 49-year-old Final Diagnosis: Thyroid carcinoma showing thymus-like differentiation (CASTLE) Symptoms: Hoarseness Medication: — Clinical Procedure: Thyroidectomy Specialty: Surgery
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Affiliation(s)
- Adrian Chi Heng Fung
- Department of Surgery, The University of Hong Kong, Hong Kong, Hong Kong S.A.R., China (mainland)
| | - Julian Shun Tsang
- Department of Surgery, The University of Hong Kong, Hong Kong, Hong Kong S.A.R., China (mainland)
| | - Brian Hung Hin Lang
- Department of Surgery, The University of Hong Kong, Hong Kong, Hong Kong S.A.R., China (mainland)
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33
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Wang M, Kundu U, Gong Y. Pitfalls of FNA diagnosis of thymic tumors. Cancer Cytopathol 2019; 128:57-67. [DOI: 10.1002/cncy.22211] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/31/2019] [Revised: 09/30/2019] [Accepted: 10/21/2019] [Indexed: 01/16/2023]
Affiliation(s)
- Minhua Wang
- Department of Pathology The University of Texas MD Anderson Cancer Center Houston Texas
| | - Uma Kundu
- Department of Pathology The University of Texas MD Anderson Cancer Center Houston Texas
| | - Yun Gong
- Department of Pathology The University of Texas MD Anderson Cancer Center Houston Texas
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34
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Lorenz L, von Rappard J, Arnold W, Mutter N, Schirp U, Scherr A, Jehle AW. Pembrolizumab in a Patient With a Metastatic CASTLE Tumor of the Parotid. Front Oncol 2019; 9:734. [PMID: 31475102 PMCID: PMC6702522 DOI: 10.3389/fonc.2019.00734] [Citation(s) in RCA: 12] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/05/2019] [Accepted: 07/22/2019] [Indexed: 11/25/2022] Open
Abstract
Carcinoma showing thymus-like elements (CASTLE) is a rare tumor, most commonly found in the thyroid gland. Here we report a case of CASTLE tumor localized to the parotid gland, recognized in retrospect after a late manifestation of symptomatic pleural carcinomatosis. The original tumor in the parotid gland was treated by surgery followed by radiotherapy. Ten years later, a metastatic disease with recurrent pleural effusions occurred. Pleural carcinomatosis was strongly positive for CD5, CD117, and p63 as was the original tumor of the parotid, which allowed the diagnosis of a CASTLE tumor. Additionally, the pleural tumor expressed high levels of programmed death ligand 1 (PD-L1), and the patient underwent treatment with the monoclonal PD-L1 inhibitor pembrolizumab achieving a partial remission. To the best of our knowledge, this is the first patient with a metastatic CASTLE tumor treated with a PD-L1 inhibitor.
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Affiliation(s)
- Lisa Lorenz
- Department of Internal Medicine, Hirslanden Klinik St. Anna, Lucerne, Switzerland
| | - Joscha von Rappard
- Department of Internal Medicine, Hirslanden Klinik St. Anna, Lucerne, Switzerland
| | - Walter Arnold
- Department of Pathology, Cantonal Hospital Lucerne, Lucerne, Switzerland
| | - Nicole Mutter
- Department of Oncology, Hirslanden Klinik St. Anna, Lucerne, Switzerland
| | - Udo Schirp
- Institute of Radiology and Nuclear Medicine, Hirslanden Klinik St. Anna, Lucerne, Switzerland
| | - Andreas Scherr
- Department of Pneumology, Hirslanden Klinik St. Anna, Lucerne, Switzerland
| | - Andreas Werner Jehle
- Department of Internal Medicine, Hirslanden Klinik St. Anna, Lucerne, Switzerland.,Transplantation Immunology and Nephrology, University Hospital Basel, Basel, Switzerland
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35
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Dualim DM, Loo GH, Suhaimi SNA, Md Latar NH, Muhammad R, Abd Shukor N. The 'CASTLE' tumour: An extremely rare presentation of a thyroid malignancy. A case report. Ann Med Surg (Lond) 2019; 44:57-61. [PMID: 31312445 PMCID: PMC6609788 DOI: 10.1016/j.amsu.2019.06.013] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/23/2019] [Revised: 06/22/2019] [Accepted: 06/26/2019] [Indexed: 11/24/2022] Open
Abstract
Thyroid carcinoma showing thymic-like differentiation (CASTLE) is a rare malignancy of the thyroid gland, and it accounts for 0.1–0.15% of all thyroid cancers. As the name suggests, it has a histological and immunophenotypic resemblance to thymic carcinoma. Preoperative diagnosis of CASTLE can be difficult as its clinical manifestations, and histological characteristic resembles other aggressive and advanced thyroid carcinomas. It is essential to distinguish CASTLE from other aggressive neoplasms as the former has a more favourable prognosis. Immunohistochemical staining with CD5 can help to differentiate thyroid CASTLE from other aggressive thyroid neoplasms. Due to the rarity of this disease, there is no clear definitive treatment strategy. Surgical resection of CASTLE is usually attempted initially. Nodal involvement and extrathyroidal extension are shown to be the main prognostic factors that influenced the survival of patients. Therefore, complete resection of the tumour is vital to reduce local recurrence rates and to improve the chance of long-term survival. Radiotherapy (RT) for CASTLE is an effective treatment. Curative surgery followed by adjuvant RT should be considered in cases with extrathyroidal extension and nodal metastases. With RT, shrinkage of the tumour and reduction of local recurrence rate is possible. With that in mind, we present a case of CASTLE who presented with airway compression symptoms three years after thyroid surgery. He subsequently underwent tumour debulking surgery and a tracheostomy. The patient refused adjuvant chemoradiotherapy, and during our serial follow-up, he is well and symptom-free.
Thyroid carcinoma showing thymic-like differentiation (CASTLE) is a rare malignancy of the thyroid gland. Diagnosis of CASTLE can be difficult as it resembles other aggressive and advanced thyroid carcinomas. Immunohistochemical staining with CD5 can help to differentiate thyroid CASTLE from other aggressive thyroid neoplasms. Complete surgical resection is vital to reduce local recurrence rates and to improve the chance of long-term survival. Adjuvant radiotherapy should be considered in cases with extrathyroidal extension and nodal metastases.
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Affiliation(s)
- Diana Mellisa Dualim
- The National University of Malaysia, Jalan Yaacob Latiff, Bandar Tun Razak, Postcode 56000, Selangor, Malaysia
| | - Guo Hou Loo
- The National University of Malaysia, Jalan Yaacob Latiff, Bandar Tun Razak, Postcode 56000, Selangor, Malaysia
| | | | - Nani Harlina Md Latar
- The National University of Malaysia, Jalan Yaacob Latiff, Bandar Tun Razak, Postcode 56000, Selangor, Malaysia
| | - Rohaizak Muhammad
- The National University of Malaysia, Jalan Yaacob Latiff, Bandar Tun Razak, Postcode 56000, Selangor, Malaysia
| | - Nordashima Abd Shukor
- The National University of Malaysia, Jalan Yaacob Latiff, Bandar Tun Razak, Postcode 56000, Selangor, Malaysia
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36
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Suzuki A, Hirokawa M, Takada N, Higuchi M, Tanaka A, Hayashi T, Kuma S, Miyauchi A. Utility of monoclonal PAX8 antibody for distinguishing intrathyroid thymic carcinoma from follicular cell-derived thyroid carcinoma. Endocr J 2018; 65:1171-1175. [PMID: 30210064 DOI: 10.1507/endocrj.ej18-0282] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022] Open
Abstract
Follicular cell-derived thyroid carcinomas, including thyroid squamous cell carcinomas (SCCs) and anaplastic carcinomas, are immunoreactive for paired-box gene 8 (PAX8), while non-follicular cell-derived thyroid carcinomas stain negative for the PAX8 antibody. Intrathyroid thymic carcinoma (ITTC) arising from the intrathyroidal ectopic thymus exhibits moderate-to-strong nuclear reactivity for polyclonal PAX8. This is difficult to understand given that PAX8 is not associated with embryonic thymic development. We aimed to determine the diagnostic significance of monoclonal PAX8 antibody in distinguishing ITTCs from follicular cell-derived thyroid carcinomas. Ten ITTCs, 14 poorly differentiated thyroid carcinomas (PDTCs), 14 thyroid SCCs, 7 thymic tissue specimens, 7 thymomas, and 1 thymic carcinoma were analyzed using antibodies against polyclonal and monoclonal PAX8, thyroid transcription factor-1, p63, and CD5. Four ITTCs (40.0%) stained positive for polyclonal PAX8; none stained positive for monoclonal PAX8. All PDTCs and 92.9% of SCCs were immunoreactive for both polyclonal and monoclonal PAX8. All PDTCs, 46.2% of SCCs, and none of the ITTCs were immunoreactive for thyroid transcription factor-1. Eight ITTCs (80.0%), but none of the PDTCs and SCCs, were immunoreactive for CD5. We are the first to show that ITTCs stain negative for monoclonal PAX8. Monoclonal PAX8 is a more reliable marker than polyclonal PAX8 for determining follicular cell origin. We conclude that monoclonal PAX8 is a useful marker for distinguishing ITTCs from PDTCs and SCCs. Monoclonal PAX8 negativity is additional evidence in support of ITTCs not being follicular cell-derived thyroid carcinomas, but having a thymic origin.
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Affiliation(s)
- Ayana Suzuki
- Department of Clinical Laboratory, Kuma Hospital, Kobe, Hyogo 650-0011, Japan
| | - Mitsuyoshi Hirokawa
- Department of Diagnostic Pathology and Cytology, Kuma Hospital, Kobe, Hyogo 650-0011, Japan
| | - Nami Takada
- Department of Diagnostic Pathology, Faculty of Medicine, Oita University, Yufu, Oita 879-5593, Japan
| | - Miyoko Higuchi
- Department of Clinical Laboratory, Kuma Hospital, Kobe, Hyogo 650-0011, Japan
| | - Aki Tanaka
- Department of Clinical Laboratory, Kuma Hospital, Kobe, Hyogo 650-0011, Japan
| | - Toshitetsu Hayashi
- Department of Diagnostic Pathology and Cytology, Kuma Hospital, Kobe, Hyogo 650-0011, Japan
| | - Seiji Kuma
- Department of Diagnostic Pathology and Cytology, Kuma Hospital, Kobe, Hyogo 650-0011, Japan
| | - Akira Miyauchi
- Department of Surgery, Kuma Hospital, Kobe, Hyogo 650-0011, Japan
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37
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Gao R, Jia X, Ji T, Feng J, Yang A, Zhang G. Management and Prognostic Factors for Thyroid Carcinoma Showing Thymus-Like Elements (CASTLE): A Case Series Study. Front Oncol 2018; 8:477. [PMID: 30416983 PMCID: PMC6212596 DOI: 10.3389/fonc.2018.00477] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/31/2018] [Accepted: 10/08/2018] [Indexed: 11/20/2022] Open
Abstract
Introduction: This study aims to identify the prognostic factors that influence therapeutic modalities for thyroid carcinoma showing thymus-like elements (CASTLE). Materials and Methods: Reported studies with CASTLE patients published between 2004 and 2018 were retrieved from a cross-database literature search. Three patients treated in our institute were also included as one case series. Standardized data collection was performed; data pertaining to clinical stages, treatment regimens, and survival time were collected and statistically analyzed. Results: Up to 26 case series of CASTLE were selected, including 51 males and 38 females with a median age of 48 years. Follow-up time ranged from 2 to 362 months and the median survival time was 158.03 months. Lymph node metastasis and tumor invasion of adjacent tissue both showed a significant negative effect on survival (p = 0.001 and 0.013, respectively). Radiotherapy significantly improved survival (p = 0.034), while neck dissection significantly prolonged survival only in patients with extrathyroidal extension (p = 0.043). Conclusions: Extrathyroidal infiltration and nodal metastasis are important factors in cancer outcomes. Radiation therapy appears to be important for better outcomes in CASTLE patients, and neck dissection is recommended for patients with extrathyroidal extension.
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Affiliation(s)
- Rui Gao
- Department of Nuclear Medicine, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, China
| | - Xi Jia
- Department of Nuclear Medicine, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, China
| | - Ting Ji
- Department of Nuclear Medicine, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, China
| | - Jinteng Feng
- Department of Thoracic Surgery, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, China
| | - Aimin Yang
- Department of Nuclear Medicine, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, China
| | - Guangjian Zhang
- Department of Thoracic Surgery, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, China
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Ota H, Hirokawa M, Suzuki A, Oshita M, Ito A, Fukushima M, Kobayashi K, Miyauchi A. Phantom Nodules Detected by Ultrasound Examination of the Neck: The Possibility of Ectopic Cervical Thymic Tissue in Adults. Ultrasound Int Open 2018; 4:E119-E123. [PMID: 30349900 PMCID: PMC6195431 DOI: 10.1055/a-0747-6453] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/07/2018] [Revised: 09/05/2018] [Accepted: 09/15/2018] [Indexed: 12/01/2022] Open
Abstract
Purpose The aim of this study was to investigate the ultrasound characteristics and clinical significance of slightly hyperechoic lesions, referred to as phantom nodules, in the perithyroidal area in patients. Materials and Methods A total of 128 patients who underwent thyroidectomy with central neck lymph node dissection at Kuma Hospital in Hyogo, Japan were included in the study. We detected 16 phantom nodules during preoperative ultrasound examinations, defined as slightly hyperechoic masses located in the perithyroidal areas, in 13 of these 128 patients (10.2%; mean age: 55.6 years, range: 36–75 years). Results All phantom nodules were located in the caudal region of the thyroid gland, and the mean maximum dimension was 7.2 mm. 12 of the 16 nodules were round or oval, while the remaining 4 were fusiform and molded by the surrounding tissue. All nodules were well-defined, solid, homogeneous, hyperechoic masses. No speckled echo pattern, internal linear echo, or vascular flow signal was observed. All 4 nodules subjected to histological examination were composed of ectopic thymic tissue. In 2 of these 4, the parenchyma was severely involuted and almost entirely replaced by adipose tissue. Conclusion To the best of our knowledge, this is the first report wherein some of the detected hyperechoic perithyroidal masses were composed of ectopic thymic tissue, and some were primarily composed of adipose tissue that completely replaced involuted ectopic thymic tissue. The results of the study suggest that these so-called phantom nodules are clinically insignificant and do not require fine needle aspiration cytology or further investigation.
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Affiliation(s)
- Hisashi Ota
- Kuma Hospital, Department of Clinical Laboratory, Kobe, Japan
| | | | - Ayana Suzuki
- Kuma Hospital, Department of Clinical Laboratory, Kobe, Japan
| | - Maki Oshita
- Kuma Hospital, Department of Clinical Laboratory, Kobe, Japan
| | - Aki Ito
- Kuma Hospital, Department of Clinical Laboratory, Kobe, Japan
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Dennis K, O'Neil M, Harrington A. Not all neck mass fine-needle aspirations with squamous cells are squamous cell carcinoma; report of a case of recurrent thyroid carcinoma with papillary and squamous components. Cytojournal 2018; 15:23. [PMID: 30294355 PMCID: PMC6161500 DOI: 10.4103/cytojournal.cytojournal_19_17] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2017] [Accepted: 06/11/2017] [Indexed: 11/04/2022] Open
Abstract
We report a case of a 65-year-old female who had a total thyroidectomy 12 years ago for papillary thyroid carcinoma (PTC) who presented with a recurrent thyroid bed mass. Fine-needle aspiration biopsy yielded malignant cells, consistent with squamous cell carcinoma (SCCa). Surgical resection was performed, and histologic evaluation of the mass showed mixed PTC and SCCa. The tumor cells were positive for BRAF V600E mutation. Thyroid carcinomas with admixed papillary carcinoma and SCCa are rare and are associated with aggressive behavior, high rates of metastasis, and poor outcomes. Although SCCa presenting as a neck mass is relatively common, clinical history and appropriate workup are essential for accurate diagnosis and determination of origin.
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Affiliation(s)
- Katie Dennis
- Address: Department of Pathology and Laboratory Medicine, University of Kansas School of Medicine, 3901 Rainbow Blvd, Kansas City, KS 66160, USA
| | - Maura O'Neil
- Address: Department of Pathology and Laboratory Medicine, University of Kansas School of Medicine, 3901 Rainbow Blvd, Kansas City, KS 66160, USA
| | - Anthony Harrington
- Address: Department of Pathology and Laboratory Medicine, University of Kansas School of Medicine, 3901 Rainbow Blvd, Kansas City, KS 66160, USA
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Abstract
The case presented herein was a 70-year-old woman who had no compliant, but had a mass in the lower part of the right lobe of the thyroid detected by ultrasound (US). The US image of the tumor, measuring 13 mm in diameter, showed a low and heterogeneous internal echo level with calcification and an irregular margin. The tumor appeared to extend to the adjacent sternothyroid muscle, and cervical lymph node swelling was detected in a computer tomography (CT) image, but no metastatic lesion was found by positron emission tomography (PET)-CT. In a fine needle aspiration cytology of the tumor, papillary thyroid carcinoma was suggested because of the atypical epithelial cells having some changes other than intranuclear inclusion bodies. A subtotal thyroidectomy and central neck lymph node dissection were performed. The excised tumor was histologically composed of irregular nests or sheets of atypical squamoid epithelial cells with some ductal structures that leached to the sternothyroid muscle and involved the right lower parathyroid gland. Carcinoma showing thymus-like differentiation (CASTLE) was diagnosed histopathologically and immunohistochemically with the following immunohistochemical results: Cluster of differentiation 5 (CD5) (+), tumor protein p63 (p63) (+), KIT proto-oncogene receptor tyrosine kinase (c-KIT(CD117)) (+), thyroglobulin (-), and thyroid transcription factor-1 (TTF-1) (-). CASTLE is a rare carcinoma of the thyroid that architecturally resembles thymic epithelial tumors. Many CASTLE patients have been misdiagnosed as other carcinomas, such as anaplastic carcinoma, poorly differentiated carcinoma or squamous cell carcinoma of the thyroid. Immunohistochemical examination, including CD5 played an important role in the final diagnosis of CASTLE, although the distinction from diagnosis as squamous cell carcinoma or mucoepidermoid carcinoma in Hematoxylin-Eosin staining was challenging in our case. Nodal metastasis and perithyroidal tumor extension of CASTLE can predict its worse prognosis. Thus, at least careful follow-up studies are mandatory in cases of CASTLE.
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Dong W, Zhang P, Li J, He L, Wang Z, Zhang T, Shao L, Zhang H. Outcome of Thyroid Carcinoma Showing Thymus-Like Differentiation in Patients Undergoing Radical Resection. World J Surg 2018; 42:1754-1761. [PMID: 29134304 DOI: 10.1007/s00268-017-4339-2] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/13/2023]
Abstract
BACKGROUND Carcinoma showing thymus-like differentiation (CASTLE) is a rare malignant tumor of the thyroid. It is difficult to diagnose, and there is no universally recognized therapeutic regimen. This study aims to define the clinicopathological features and discuss the optimal management of CASTLE. METHODS We retrospectively analyzed six patients with CASTLE who accepted surgery at the First Hospital of China Medical University between January 2010 and December 2015. RESULTS The six patients (three women and three men) had median age of 53 years (range 47-61 years). All patients presented with a slow-growing, painless neck mass; three patients also had hoarseness. All tumors were located in middle-lower or lower lobe, and two tumors extended to the substernal region. All patients underwent radical surgery without postoperative radiotherapy or chemotherapy. Five patients had extrathyroidal extension and two had lymph node metastasis. All six tumors were positive for CD5 and negative for thyroglobulin (Tg) and thyroid transcription factor (TTF)-1. Median follow-up was 32 months (range 23-81 months). Lateral cervical lymph node metastasis occurred in one patient at 26 months after initial treatment. CONCLUSIONS CASTLE is a rare, aggressive malignant tumor of the thyroid. Ultrasound, computed tomography, and fine-needle aspiration biopsy may not be sufficient to establish the diagnosis preoperatively; pathological examination and immunohistochemistry, particularly positive CD5 staining, are necessary to establish the diagnosis. Patients with CASTLE can yield a favorable outcome after radical surgery.
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Affiliation(s)
- Wenwu Dong
- Department of Thyroid Surgery, The First Hospital of China Medical University, Shenyang, 110001, People's Republic of China
| | - Ping Zhang
- Department of Thyroid Surgery, The First Hospital of China Medical University, Shenyang, 110001, People's Republic of China
| | - Jian Li
- Department of Surgery, Jinqiu Hospital of Liaoning, Shenyang, 110016, People's Republic of China
| | - Liang He
- Department of Thyroid Surgery, The First Hospital of China Medical University, Shenyang, 110001, People's Republic of China
| | - Zhihong Wang
- Department of Thyroid Surgery, The First Hospital of China Medical University, Shenyang, 110001, People's Republic of China
| | - Ting Zhang
- Department of Thyroid Surgery, The First Hospital of China Medical University, Shenyang, 110001, People's Republic of China
| | - Liang Shao
- Department of Thyroid Surgery, The First Hospital of China Medical University, Shenyang, 110001, People's Republic of China
| | - Hao Zhang
- Department of Thyroid Surgery, The First Hospital of China Medical University, Shenyang, 110001, People's Republic of China.
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Kong F, Ying H, Zhai R, Du C, Huang S, Zhou J, He X, Hu C, Wang Z, Sun T, Ji Q. Clinical outcome of intensity modulated radiotherapy for carcinoma showing thymus-like differentiation. Oncotarget 2018; 7:81899-81905. [PMID: 27626313 PMCID: PMC5348440 DOI: 10.18632/oncotarget.11914] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/15/2016] [Accepted: 09/02/2016] [Indexed: 11/25/2022] Open
Abstract
PURPOSE To evaluate the efficacy and toxicity of adjuvant intensity-modulated radiotherapy (IMRT) after surgery for carcinoma showing thymus-like differentiation (CASTLE). METHODS Between September 2008 and June 2015, 14 CASTLE patients were retrospectively enrolled. The clinical features, treatment procedure and clinical outcomes were reviewed. All patients received postoperative IMRT. The radiation doses ranged from 56Gy/28 fractions to 66Gy/33 fractions. Treatment-related toxicities were graded by National Cancer Institute Common Toxicity Criteria (NCI-CTC) version 3.0. RESULTS After a median follow-up period of 42 months, only one patient suffered local recurrence and distant metastasis. The most frequently seen acute toxicities were mucositis and dermatitis (grade 1-2). No grade 3-4 toxicities were observed. CONCLUSIONS Although based upon a small series of consecutively treated patients, our study showed that adjuvant IMRT provides satisfactory local-regional control for CASTLE, with acceptable toxicities. Further studies are still warranted to clarify our findings.
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Affiliation(s)
- Fangfang Kong
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China
| | - Hongmei Ying
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China
| | - Ruiping Zhai
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China
| | - Chengrun Du
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China
| | - Shuang Huang
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China.,Department of Radiation Oncology, Zhejiang Cancer Hospital, Hangzhou, P. R. China
| | - Junjun Zhou
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China
| | - Xiayun He
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China
| | - Chaosu Hu
- Department of Radiation Oncology, Fudan University Shanghai Cancer Center, Shanghai, P.R. China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China
| | - Zhuoying Wang
- Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China.,Department of Head and Neck Surgery, Fudan University Shanghai Cancer Center, Shanghai, P.R. China
| | - Tuanqi Sun
- Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China.,Department of Head and Neck Surgery, Fudan University Shanghai Cancer Center, Shanghai, P.R. China
| | - Qinghai Ji
- Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, P.R. China.,Department of Head and Neck Surgery, Fudan University Shanghai Cancer Center, Shanghai, P.R. China
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Wong EHC, Tetter N, Tzankov A, Muller L. CASTLE tumor of the parotid: First documented case, literature review, and genetic analysis of the cancer. Head Neck 2017; 40:E1-E4. [PMID: 29120527 DOI: 10.1002/hed.24985] [Citation(s) in RCA: 17] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/28/2017] [Revised: 07/20/2017] [Accepted: 09/15/2017] [Indexed: 01/07/2023] Open
Abstract
BACKGROUND Carcinoma showing thymus-like elements (CASTLE) is a rare tumor that normally affects the thyroid gland and was thought to arise from either the remnants of the branchial pouch or the ectopic cervical thymic tissue. These tumors show strong immunopositivity for CD5, P63, and CD117. Most CASTLE of the thyroid gland is treated with surgery with or without adjuvant radiotherapy or chemotherapy. METHOD A 55-year-old woman presented with a slow-growing right parotid mass. A right total parotidectomy and ipsilateral selective neck dissection were performed and the diagnosis of CASTLE was made after confirmation with an immunohistochemistry test. She received radiotherapy postoperatively. RESULTS Genetic sequencing showed alterations in the PPARG, BRCA2, and NOTCH1 genes. She remained disease free for >1 year after treatment. CONCLUSION We believe that this is the first reported case of CASTLE in the parotid gland. Clinicians should be aware of this rare entity and consider this differential diagnosis after ruling out other common parotid lesions.
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Affiliation(s)
- Eugene Hung Chih Wong
- Department of Otolaryngology, Head and Neck Surgery, University Hospital of Basel, Basel, Switzerland
| | - Nora Tetter
- Department of Otolaryngology, Head and Neck Surgery, University Hospital of Basel, Basel, Switzerland
| | - Alexandar Tzankov
- Institute of Medical Genetics and Pathology, University Basel, Basel, Switzerland
| | - Laurent Muller
- Department of Otolaryngology, Head and Neck Surgery, University Hospital of Basel, Basel, Switzerland
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Rajeshwari M, Singh V, Nambirajan A, Mridha AR, Jain D. Carcinoma showing thymus like elements: Report of a case with EGFR T790M mutation. Diagn Cytopathol 2017; 46:413-418. [PMID: 29115061 DOI: 10.1002/dc.23859] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/23/2017] [Revised: 10/25/2017] [Accepted: 10/26/2017] [Indexed: 11/05/2022]
Abstract
Carcinoma showing thymus-like differentiation of the thyroid (CASTLE) is a rare tumor involving the thyroid and perithyroidal soft tissues. It shares morphological, immunohistochemical and molecular similarities with thymic carcinomas. Due to its relatively better prognosis, it needs differentiation from other primary and metastatic tumors of this region. A 40-year-old lady presented with a gradually progressive anterior neck swelling for one year. Imaging showed bulky right and left lobes of thyroid along with a solid soft tissue mass in the pretracheal region. Fine needle aspiration smears showed features of poorly differentiated carcinoma. Total thyroidectomy with excision of the mass revealed histopathological features characteristic of CASTLE, with evidence of thyroiditis in adjoining thyroid. Epidermal growth factor receptor (EGFR) assay revealed presence of EGFR T790M somatic mutation in exon 20. The same was not detectable on direct sequencing. We present a rare case of CASTLE, occurring in association with Hashimoto thyroiditis, with emphasis on cytological features and report for the first time the presence of a low level somatic mutation in EGFR (EGFR T790M mutation).
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Affiliation(s)
- Madhu Rajeshwari
- Department of Pathology, All India Institute of Medical Sciences, New Delhi 110029, India
| | - Varsha Singh
- Department of Pathology, All India Institute of Medical Sciences, New Delhi 110029, India
| | - Aruna Nambirajan
- Department of Pathology, All India Institute of Medical Sciences, New Delhi 110029, India
| | - Asit Ranjan Mridha
- Department of Pathology, All India Institute of Medical Sciences, New Delhi 110029, India
| | - Deepali Jain
- Department of Pathology, All India Institute of Medical Sciences, New Delhi 110029, India
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45
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Lominska C, Estes CF, Neupane PC, Shnayder Y, TenNapel MJ, O'Neil MF. CASTLE Thyroid Tumor: A Case Report and Literature Review. Front Oncol 2017; 7:207. [PMID: 28955658 PMCID: PMC5602351 DOI: 10.3389/fonc.2017.00207] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/06/2017] [Accepted: 08/24/2017] [Indexed: 11/21/2022] Open
Abstract
Carcinoma showing thymus-like differentiation is a rare tumor of the thyroid gland, which is structurally similar to thymic tissue. Overall, it has a favorable prognosis. Radiotherapy has been shown to be an effective local treatment, but there have been reports of distant recurrence. It has been suggested that adding chemotherapy may decrease the risk of recurrence. Here, we present a case report of a patient with a large tumor and extrathyroidal extension. The patient was treated with surgery, radiotherapy, and cisplatin with acceptable toxicity. The patient is free of locally recurrent or distant disease at 3 years.
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Affiliation(s)
- Chris Lominska
- Radiation Oncology, University of Kansas Medical Center, Kansas City, KS, United States
| | | | - Prakash C Neupane
- Medical Oncology, University of Kansas Medical Center, Westwood, KS, United States
| | - Y Shnayder
- Otolaryngology, University of Kansas Medical Center, Kansas City, KS, United States
| | - Mindi J TenNapel
- Radiation Oncology, University of Kansas Medical Center, Kansas City, KS, United States
| | - Maura F O'Neil
- Pathology, University of Kansas Medical Center, Kansas City, KS, United States
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46
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Collins JA, Ping B, Bishop JA, Ali SZ. Carcinoma Showing Thymus-Like Differentiation (CASTLE): Cytopathological Features and Differential Diagnosis. Acta Cytol 2016; 60:421-428. [PMID: 27643719 DOI: 10.1159/000448836] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/19/2016] [Accepted: 07/28/2016] [Indexed: 01/27/2023]
Abstract
BACKGROUND CASTLE (carcinoma showing thymus-like differentiation) is a rare tumor (accounting for less than 0.15% of thyroid neoplasms) with most of the reported cases from Asia. Although this tumor is rare, distinguishing it from more aggressive neoplasms is critical because of its improved prognosis. To date, there have been limited studies on its cytomorphological features. Herein, we review the cytomorphological features of 10 fine-needle aspiration (FNA) cases of histologically confirmed CASTLE and discuss the findings in light of the current literature. METHODS We retrospectively (1989-2016) identified 10 cases of CASTLE from 17,415 surgical cases of thyroid carcinoma from The Johns Hopkins Hospital and Fudan University Shanghai Cancer Center. The cases were reviewed for discrete cytomorphological characteristics. RESULTS All of the cases displayed predominantly single (dishesive) epithelial cell populations, high nuclear/cytoplasmic ratios, hyperchromatic to open/vesicular nuclei and macronucleoli. The majority of cases additionally showed hypercellular, three-dimensional fragments in a background of lymphocytes. The fragments displayed syncytial architecture without molding and rarely squamous differentiation. All cases lacked follicular differentiation, intranuclear inclusions, nuclear molding, or neuroendocrine-type chromatin. CONCLUSIONS Cytopathological features alone do not appear to be distinctive or definitive of CASTLE but rather allow for the inclusion of CASTLE within the differential diagnosis. Procurement of cell block material is important and resulted in the single case of CASTLE accurately diagnosed on FNA. Cytopathological features that favored CASTLE included syncytial fragments of malignant cells with pleomorphic large nuclei, vesicular chromatin, and prominent nucleoli in a background of lymphocytes. These cytopathological findings in the appropriate clinicoradiological context warrant its inclusion in the reported differential diagnosis in order to be able to implement appropriate clinical management.
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Affiliation(s)
- Jennifer A Collins
- Department of Pathology, The Johns Hopkins Hospital, Baltimore, Md., USA
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47
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Sheikh S, Picken C, Montgomery E. Ectopic Hamartomatous Thymoma. Int J Surg Pathol 2016. [DOI: 10.1177/106689699800600305] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
The authors report an example of an unusual lower neck lesion, ectopic hamartomatous thymoma. Like other examples of this entity, the lesion was well marginated and characterized by cysts lined by squamous epithelium, adipose tissue, and spindle cells. The lesion was initially mistaken for a malignant neoplasm (at the time of frozen section) and the correct interpretation was rendered upon review of the permanent sections. They review the literature and clinicopathologic differential diagnosis of this entity.
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Affiliation(s)
- Salwa Sheikh
- Department of Pathology and Otolaryngology, Georgetown University, Basic Sciences Building, 3900 Reservoir Road, NW, Washington, DC 20007
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48
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Piacentini MG, Romano F, De Fina S, Sartori P, Leone EB, Rubino B, Uggeri F. Carcinoma of the Neck Showing Thymic-like Elements (CASTLE): Report of a Case and Review of the Literature. Int J Surg Pathol 2016; 14:171-5. [PMID: 16703183 DOI: 10.1177/106689690601400214] [Citation(s) in RCA: 22] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
Carcinoma showing thymic-like elements (CASTLE) is a rare tumor affecting thyroid and neck soft tissues, which has to be distinguished from squamous cell and anaplastic thyroid carcinoma, because it has a better prognosis. We report a new case of CASTLE which occurred in a patient submitted to total thyroidectomy with central neck dissection. The tumor stained positively for CD5, which seems to be the most useful marker in the differential diagnosis. By the analysis of the 18 cases reported in literature, total thyroidectomy with selective modified neck dissection should be the treatment of choice and radiotherapy should be considered for patients with positive nodal status.
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Affiliation(s)
- Maria Gaia Piacentini
- Department of Surgery, San Gerardo Hospital, II University of Milan-Bicocca, Monza, Italy
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Srivastava A, Padilla O, Alroy J, Ucci A, Pilichowska M, Daley B, Wolfe HJ. Primary Intrapulmonary Spindle Cell Thymoma with Marked Granulomatous Reaction: Report of a Case with Review of Literature. Int J Surg Pathol 2016; 11:353-6. [PMID: 14615837 DOI: 10.1177/106689690301100419] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
Primary intrapulmonary thymoma is a rare lesion with around 20 cases reported so far in the literature. A pure spindle cell morphology in these lesions is rarer still with only a single case recorded to date. We report herein an interesting case of a 47-yearold-man, status post surgical resection and radiotherapy for a squamous cell carcinoma of the floor of mouth, who was being followed up for a radiologic opacity in the right lower lobe of the lung. The lesion remained stable in size for almost 5 years and then an increase in size was noted. A right lower lobectomy was performed with a preoperative suspicion of metastasis. Histologic and immunohistochemical evaluation revealed a primary intrapulmonary spindle cell thymoma that displayed a prominent granulomatous reaction, a phenomenon not described so far in the literature. We discuss the possible embryologic origins and the pitfalls in diagnosis of these rare neoplasms. The remarkable granulomatous response observed in the tumor raises the possibility that similar lesions might conceivably occur in the thymus as well.
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Affiliation(s)
- Amitabh Srivastava
- Department of Pathology, New England Medical Center, Boston, MA 02111, USA
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50
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Saw D, Wu D, Chess Q, Shemen L. Spindle Epithelial Tumor with Thymus-like Element (SETTLE), a Primary Thyroid Tumor. Int J Surg Pathol 2016. [DOI: 10.1177/106689699600400306] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
A 49-year-old female presented with a nontender mass in the neck. Fine-needle aspirate revealed spindle cells in a vaguely whorled pattern. The cells were fairly uniform and had elongated nuclei with fine chromatin. It was diagnosed as a cellular spindle cell neoplasm of uncertain histogenesis. Total thyroidectomy was performed revealing a well-circumscribed mass in the right lower pole extending substernally for 2 cm. The tumor was highly cellular and characterized by fascicles of spindle cells, focally displaying a biphasic pattern. The glandular component had a tubopapillary pattern. Occasional small mucous cysts were found adjacent to the tumor and in the opposite lobe, suggesting that the tumor might have arisen from the epithelial inclusions or remnants of branchial pouch within the thyroid. The spindle cells were immunoreactive for cytokeratin, epithelial membrane antigen, and vimentin and negative for thyroglobulin, calcitonin, chromogranin, desmin, myoglobin, and actin. Ultrastructurally, the spindle cells had abundant tonofilaments and well-developed desmosomes. These findings are consistent with a newly described entity, spindle epithelial tumor with thymus-like element (SETTLE), an intrathyroid lesion with a potential for delayed metastasis. Int J Surg Pathol 4(3):00-00, 1997
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Affiliation(s)
- Daisy Saw
- New York Hospital Medical Center of Queens, Flushing, Memorial Sloan Kettering Cancer Center; New York
| | - Derrina Wu
- Mount Sinai Medical Center, New York, New York
| | | | - Larry Shemen
- New York Hospital Medical Center of Queens, Flushing
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