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Singh S, Sahni M, Gothwal R. Low-Grade Myofibroblastic Sarcoma of Hard Palate-A Diagnostic Challenge with Review of Literature. Indian J Surg Oncol 2024; 15:497-500. [PMID: 39328731 PMCID: PMC11422317 DOI: 10.1007/s13193-024-02046-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/22/2024] [Accepted: 07/21/2024] [Indexed: 09/28/2024] Open
Abstract
Low-grade myofibroblastic sarcoma (LGMS) is categorized as an extremely rare malignant neoplasia of myofibroblasts. To date, only 38 cases describing patients diagnosed with LGMS in the oro-maxillofacial region have been reported in the scientific literature. Here, we introduce a rare case report and, for the first time, also provide a recent update of the literature and a clear review regarding the immunohistochemical panel to diagnose this entity.
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Affiliation(s)
- Suresh Singh
- Department of Surgical Oncology, SMS Medical College and Hospital, Jaipur, Rajasthan India
| | - Manish Sahni
- Department of Surgical Oncology, SMS Medical College and Hospital, Jaipur, Rajasthan India
| | - Ravindra Gothwal
- Department of Radiation Oncology, SMS Medical College and Hospital, Jaipur, Rajasthan India
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Kito M, Ae K, Okamoto M, Endo M, Ikuta K, Takeuchi A, Yasuda N, Yasuda T, Imura Y, Morii T, Kikuta K, Kawamoto T, Nezu Y, Baba I, Ohshika S, Uehara T, Ueda T, Takahashi J, Kawano H. Clinical Outcome of Low-Grade Myofibroblastic Sarcoma in Japan: A Multicenter Study from the Japanese Musculoskeletal Oncology Group. Cancers (Basel) 2023; 15:cancers15082314. [PMID: 37190242 DOI: 10.3390/cancers15082314] [Citation(s) in RCA: 5] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/17/2023] [Revised: 04/10/2023] [Accepted: 04/14/2023] [Indexed: 05/17/2023] Open
Abstract
This retrospective multicenter study aimed to analyze the clinical features and prognosis of 24 patients diagnosed with LGMS between 2002 and 2019 in the Japanese sarcoma network. Twenty-two cases were surgically treated and two cases were treated with radical radiotherapy (RT). The pathological margin was R0 in 14 cases, R1 in 7 cases, and R2 in 1 case. The best overall response in the two patients who underwent radical RT was one complete response and one partial response. Local relapse occurred in 20.8% of patients. Local relapse-free survival (LRFS) was 91.3% at 2 years and 75.4% at 5 years. In univariate analysis, tumors of 5 cm or more were significantly more likely to cause local relapse (p < 0.01). In terms of the treatment of relapsed tumors, surgery was performed in two cases and radical RT was performed in three cases. None of the patients experienced a second local relapse. Disease-specific survival was 100% at 5 years. A wide excision aimed at the microscopically R0 margin is considered the standard treatment for LGMS. However, RT may be a viable option in unresectable cases or in cases where surgery is expected to cause significant functional impairment.
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Affiliation(s)
- Munehisa Kito
- Department of Orthopaedic Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto 390-8621, Japan
| | - Keisuke Ae
- Department of Orthopaedic Surgery, Cancer Institute Hospital, Japanese Foundation for Cancer Research, 3-8-31 Ariake, Koto-ku, Tokyo 135-8550, Japan
| | - Masanori Okamoto
- Department of Orthopaedic Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto 390-8621, Japan
| | - Makoto Endo
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan
| | - Kunihiro Ikuta
- Department of Orthopaedic Surgery, Nagoya University Graduate School of Medicine, 65 Tsurumai, Showa-ku, Nagoya 466-8560, Japan
| | - Akihiko Takeuchi
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takaramachi, Kanazawa 920-8641, Japan
| | - Naohiro Yasuda
- Department of Orthopaedic Surgery, Osaka University Graduate School of Medicine, 2-2 Yamadaoka, Suita 565-0871, Japan
- Department of Orthopaedic Surgery, National Hospital Organization Osaka National Hospital, 2-1-14 Houenzaka, Chuo-ku, Osaka 540-0006, Japan
| | - Taketoshi Yasuda
- Department of Orthopaedic Surgery, University of Toyama, 2630 Sugitani, Toyama 930-0194, Japan
| | - Yoshinori Imura
- Department of Orthopaedic Surgery, Osaka International Cancer Institute, 3-1-69 Otemae, Chuo-ku, Osaka 540-0008, Japan
| | - Takeshi Morii
- Department of Orthopaedic Surgery, Kyorin University Faculty of Medicine, 6-20-2 Shinkawa, Tokyo 181-8621, Japan
| | - Kazutaka Kikuta
- Department of Musculoskeletal Oncology and Orthopaedic Surgery, Tochigi Cancer Center, 4-9-13 Yonan, Utsunomiya 320-0834, Japan
| | - Teruya Kawamoto
- Department of Orthopaedic Surgery, Kobe University Graduate School of Medicine, 7-5-1 Kusunoki-cho, Chuo-ku, Kobe 650-0017, Japan
| | - Yutaka Nezu
- Department of Musculoskeletal Oncology and Rehabilitation, National Cancer Center Hospital, 5-1-1 Tsukigi, Chuo-ku, Tokyo 104-0045, Japan
| | - Ichiro Baba
- Department of Orthopaedic Surgery, Osaka Medical and Pharmaceutical University, 2-7 Daigakumachi, Takatsuki 569-8686, Japan
| | - Shusa Ohshika
- Department of Orthopaedic Surgery, Hirosaki University Graduate School of Medicine, 5 Zaifu-cho, Hirosaki 036-8562, Japan
| | - Takeshi Uehara
- Department of Laboratory Medicine, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto 390-8621, Japan
| | - Takafumi Ueda
- Department of Orthopaedic Surgery, Kodama Hospital, 1-3-2 Gotenyama, Takarazuka 665-0841, Japan
| | - Jun Takahashi
- Department of Orthopaedic Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto 390-8621, Japan
| | - Hirotaka Kawano
- Department of Orthopaedic Surgery, Teikyo University School of Medicine, 2-11-1 Kaga, Itabashi-ku, Tokyo 173-0806, Japan
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Modaboyina S, Pushker N, Agarwal S, Bakhshi S, Sen S, Ray R, Kashyap S. Recurrent periorbital low-grade myofibroblastic sarcoma in an infant. Indian J Ophthalmol 2022; 70:2744-2746. [PMID: 35791232 PMCID: PMC9426097 DOI: 10.4103/ijo.ijo_2686_21] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/24/2022] Open
Affiliation(s)
- Sujeeth Modaboyina
- Oculoplastics, Tumor and Pediatric Ophthalmology Services, New Delhi, India
| | - Neelam Pushker
- Oculoplastics, Tumor and Pediatric Ophthalmology Services, New Delhi, India
| | - Sahil Agarwal
- Oculoplastics, Tumor and Pediatric Ophthalmology Services, New Delhi, India
| | - Sameer Bakhshi
- Department of Medical Oncology, Dr BRAIRCH, All India Institute of Medical Sciences, New Delhi, India
| | - Seema Sen
- Department of Ocular Pathology, Dr. R. P. Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, India
| | - Ruma Ray
- Department of Pathology, All India Institute of Medical Sciences, New Delhi, India
| | - Seema Kashyap
- Department of Ocular Pathology, Dr. R. P. Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, India
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Gonçalves JM, Marola LHG, Vieira DSC, Modolo F, Gondak R. The challenging diagnosis of low-grade myofibroblastic sarcoma: A case report and literature update. Oral Oncol 2022; 126:105762. [DOI: 10.1016/j.oraloncology.2022.105762] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/19/2022] [Revised: 01/26/2022] [Accepted: 01/31/2022] [Indexed: 11/15/2022]
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Xu Y, Xu G, Wang X, Mao M, Wu H, Baklaushev VP, Chekhonin VP, Peltzer K, Wang G, Zhang C. Is there a role for chemotherapy and radiation in the treatment of patients with low-grade myofibroblastic sarcoma? Clin Transl Oncol 2021; 23:344-352. [PMID: 32607812 DOI: 10.1007/s12094-020-02425-4] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/23/2020] [Accepted: 06/10/2020] [Indexed: 10/24/2022]
Abstract
PURPOSE Low-grade myofibroblastic sarcoma (LGMS) is a rare entity with a predilection for the head and neck. There are still no optimal treatment strategies for patients with LGMS. We retrospectively investigated the efficacies of chemotherapy and radiation treatment for patients with LGMS. METHODS/PATIENTS We obtained data from the Surveillance, Epidemiology, and End Result (SEER) database for 96 patients diagnosed with LGMS between 2001 and 2015. We used Kaplan-Meier curves and log-rank tests to estimate overall survival (OS) and Cox proportional hazard regression to identify prognostic factors. RESULTS The median age of the patients was 55.0 years. Twenty-two of the patients had LGMS in the head and neck region. Of the 96 patients, 86 (89.6%) received surgical treatment, 28 (29.2%) received radiation treatment, and 20 (10.4%) received chemotherapy. The mean OS was 125.2 [95% confidence interval (CI) 106.3-144.2] months. The 1, 3, 5, and 10-year OS rates were 88%, 77%, 70%, and 59%, respectively. Age greater than 60 years, positive nodal status, and no surgical treatment were independent prognostic factors for patients with LGMS, whereas chemotherapy and radiation treatment were not. CONCLUSIONS Surgical resection is the most effective therapy for LGMS. Chemotherapy and radiation had limited effects on survival improvement for patients with LGMS. Therefore, chemotherapy and/or radiation therapy should not be routinely performed in LGMS, especially for those with negative margins after surgery.
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Affiliation(s)
- Y Xu
- Department of Bone and Soft Tissue Tumors, Tianjin Medical University Cancer Institute and Hospital, National Clinical Research Center for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Center for Cancer, Tianjin, China
| | - G Xu
- Department of Bone and Soft Tissue Tumors, Tianjin Medical University Cancer Institute and Hospital, National Clinical Research Center for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Center for Cancer, Tianjin, China
- Department of Orthopaedics, Tianjin Hospital, Tianjin, China
| | - X Wang
- Department of Epidemiology and Biostatistics, First Affiliated Hospital, Army Medical University, Chongqing, China
| | - M Mao
- Department of Pathology and Southwest Cancer Center, First Affiliated Hospital, Army Medical University, Chongqing, China
| | - H Wu
- Department of Bone and Soft Tissue Tumors, Tianjin Medical University Cancer Institute and Hospital, National Clinical Research Center for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Center for Cancer, Tianjin, China
| | - V P Baklaushev
- Federal Research and Clinical Center of Specialized Medical Care and Medical Technologies, Federal Biomedical Agency of the Russian Federation, Moscow, Russian Federation
| | - V P Chekhonin
- Department of Basic and Applied Neurobiology, Federal Medical Research Center for Psychiatry and Narcology, Moscow, Russian Federation
| | - K Peltzer
- Department of Research and Innovation, University of Limpopo, Turfloop, South Africa
| | - G Wang
- Department of Bone and Soft Tissue Tumors, Tianjin Medical University Cancer Institute and Hospital, National Clinical Research Center for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Center for Cancer, Tianjin, China.
| | - C Zhang
- Department of Bone and Soft Tissue Tumors, Tianjin Medical University Cancer Institute and Hospital, National Clinical Research Center for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin's Clinical Research Center for Cancer, Tianjin, China.
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Yonezawa H, Yamamoto N, Hayashi K, Takeuchi A, Miwa S, Igarashi K, Langit MB, Kimura H, Shimozaki S, Kato T, Morinaga S, Araki Y, Asano Y, Ikeda H, Nojima T, Tsuchiya H. Low-grade myofibroblastic sarcoma of the levator scapulae muscle: a case report and literature review. BMC Musculoskelet Disord 2020; 21:836. [PMID: 33302922 PMCID: PMC7731512 DOI: 10.1186/s12891-020-03857-3] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/08/2020] [Accepted: 12/03/2020] [Indexed: 01/15/2023] Open
Abstract
BACKGROUND Low-grade myofibroblastic sarcoma (LGMS) is described as a distinct atypical myofibroblastic tumor often with fibromatosis-like features and predilection for the head and neck, especially the oral cavity and larynx. LGMS arising in the levator scapulae muscle is extremely rare. CASE PRESENTATION A 69-year-old woman was admitted to our hospital because she noticed a hard mass in her left neck six months prior. Magnetic resonance images (MRI) showed a soft tissue tumor of the left levator scapulae muscle. A core needle biopsy showed cellular fascicles or a storiform growth pattern of spindle-shaped tumor cells with minimally atypia. Immunohistochemistry revealed focally positive for α-smooth muscle actin (α-SMA), negative for S-100, and a low-grade spindle cell sarcoma was suspected. Following a biopsy, the tumor was resected with a wide surgical margin. Immunohistochemical staining was a positive for vimentin and α-SMA and negative for desmin, CD34, nuclear β-catenin, and h-caldesmon. LGMS diagnosis was determined based on the histopathological findings. The patient was alive with no evidence of disease eight years after the surgery. CONCLUSIONS To the best of our knowledge, this is the first case report of LGMS arising in the levator scapulae muscle. In addition to the case report, 48 reports with 103 LGMS cases are reviewed and discussed. In previous reports of LGMS, there were 43 females and 60 males, with a mean age of 43.0 years (range, 2-75). There were 13 (12.6%) patients aged < 18 years, 67 (65.1%) patients aged 18 to 59 years, and 23 (22.3%) patients aged ≥60 years. The average tumor size was 4.4 cm (range: 0.4-22.0). The commonest sites of LGMS was the tongue. Tumor growth patterns were evaluated in 52 cases, and 44 cases (84.6%) showed infiltrative growth patterns. Local recurrence was 26.7%, and distant metastasis was 4.4%. Because of the locally aggressive feature, it is important to diagnose LGMS with biopsy and to excise the tumor with an adequately wide margin.
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Affiliation(s)
- Hirotaka Yonezawa
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Norio Yamamoto
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan.
| | - Katsuhiro Hayashi
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Akihiko Takeuchi
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Shinji Miwa
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Kentaro Igarashi
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Mickhael Bang Langit
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan.,Department of Orthopedics, Philippine Orthopedic Center, Quezon City, Manilla, Philippines
| | - Hiroaki Kimura
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Shingo Shimozaki
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Takashi Kato
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Sei Morinaga
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Yoshihiro Araki
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Yohei Asano
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
| | - Hiroko Ikeda
- Department of Diagnostic Pathology, Kanazawa University Hospital, Kanazawa, Japan
| | - Takayuki Nojima
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan.,Department of Diagnostic Pathology, Kanazawa University Hospital, Kanazawa, Japan
| | - Hiroyuki Tsuchiya
- Department of Orthopaedic Surgery, Graduate School of Medical Sciences, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8640, Japan
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7
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Zhao R, Wang J, Zhang H, Chi Y, Bi N. High-grade myofibroblastic sarcoma of the pleura: A case report and literature review. Thorac Cancer 2020; 11:3011-3014. [PMID: 32815307 PMCID: PMC7529570 DOI: 10.1111/1759-7714.13613] [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: 06/01/2020] [Revised: 07/22/2020] [Accepted: 07/23/2020] [Indexed: 11/28/2022] Open
Abstract
High‐grade myofibroblastic sarcoma (HGMS) is a rare cancer that has high recurrence and metastatic rates. Here, we report the first case of HGMS originating from the pleura. Based on the findings of pleural biopsy, pathological examination and immunohistochemical staining, grade III myofibroblastic sarcoma (MS) was diagnosed. The patient underwent eight cycles of chemotherapy (epirubicin and ifosfamide), followed by radiotherapy. As of May 2020, the patient had been followed for six months and no tumor progression had occurred. Key points This is the first report of high‐grade myofibroblastic sarcoma originating from the pleura. The patient was treated via nonsurgical strategies, including chemotherapy and radiotherapy.
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Affiliation(s)
- Ruizhi Zhao
- Department of Radiation Oncology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Jianyang Wang
- Department of Radiation Oncology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Hongtu Zhang
- Department of Pathology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Yihebali Chi
- Department of Medical Oncology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
| | - Nan Bi
- Department of Radiation Oncology, National Cancer Center/National Clinical Research Center for Cancer/Cancer Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China
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8
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Pereira de Oliveira DHI, da Silveira ÉJD, de Souza LB, Caro-Sanchez CHS, Dominguez-Malagon H, Mosqueda Taylor A, Queiroz LMG. Myofibroblastic lesions in the oral cavity: Immunohistochemical and ultrastructural analysis. Oral Dis 2018; 25:174-181. [PMID: 30168888 DOI: 10.1111/odi.12972] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/27/2018] [Revised: 08/09/2018] [Accepted: 08/22/2018] [Indexed: 12/15/2022]
Abstract
OBJECTIVE To immunohistochemically characterize a group of oral myofibroblastic lesions (MLs) and to evaluate the ultrastructural features of myofibroblasts. MATERIAL AND METHODS Using a tissue microarray technique (TMA), cases of myofibroma (MF), of nodular fasciitis (NF), of desmoplastic fibroma (DF), and of myofibroblastic sarcoma (MS) from the Universidad Autónoma Metropolitana Xochimilco, and a Private Oral Pathology Service in Mexico City were stained with antibodies against alpha-smooth muscle actin (α-SMA), H-caldesmon, vimentin, desmin, β-catenin, CD34, anaplastic lymphoma protein kinase (ALK-1), and Ki-67. RESULTS Nineteen of the 22 MF cases, 2/5 of the NF cases, 1/10 of the DF cases, and 1/2 of the MS cases were positive for α-SMA. 1/2 of the MS cases were positive for desmin; 6/10 of the DF cases were positive for β-catenin, and 2 of the MF cases were positive for ALK-1. All of the MLs were positive for vimentin and negative for H-caldesmon and CD-34. The Ki-67 labeling index in all of the 8/22 MF, 3/5 NF, and 2/2 MS cases was ≥10%. For all of the MLs evaluated, ultrastructural analysis revealed spindle-shaped cells containing endoplasmic reticulum and peripheral actin filament bundles. CONCLUSION In certain myofibroblastic lesions, the use of auxiliary techniques (such as immunohistochemistry) can be critical for differential diagnosis.
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Affiliation(s)
| | | | - Lélia Batista de Souza
- Postgraduate Program, Oral Pathology, Federal University of Rio Grande do Norte, Natal, Brazil
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Wen J, Zhao W, Li C, Shen JY, Wen TF. High-grade myofibroblastic sarcoma in the liver: A case report. World J Gastroenterol 2017; 23:7054-7058. [PMID: 29097878 PMCID: PMC5658323 DOI: 10.3748/wjg.v23.i38.7054] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/23/2017] [Revised: 06/18/2017] [Accepted: 08/15/2017] [Indexed: 02/06/2023] Open
Abstract
Only two cases of myofibroblastic sarcoma in the liver have been reported in the literature. Here, we report the case of a male patient with high-grade myofibroblastic sarcoma mimicking echinococcosis in the liver. The 25-year-old male patient complained of right upper quadrant swelling pain for one week and was initially diagnosed with echinococcosis. He was then scheduled for an exploratory laparotomy. During the operation, a huge mass exceeding 16 cm in diameter was found to occupy nearly the entire right trisegment of the liver, with a clear boundary and a round shape, and the mass was resected by right hepatic trisegmentectomy. Immunohistochemical staining revealed that the tumor tissue was positive for desmin, α-smooth muscle actin, CD56, and vimentin and negative for ALK-1, myogenin, calponin, β-catenin, S100, and glypican-3, with a Ki-67 (MIB-1) index of approximately 20%. Based on the histological manifestations and immunohistochemical staining, a diagnosis of myofibroblastic sarcoma was established. The postoperative recovery was uneventful. There was no evidence of recurrence or metastasis through the last follow-up, 6 mo after surgery, despite a lack of postoperative chemotherapy or radiotherapy. To the best of our knowledge, the present case is the first reported case of high-grade myofibroblastic sarcoma in the liver, and it is also the first reported case in a male patient.
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Affiliation(s)
- Jun Wen
- Department of Liver Surgery and Liver Transplantation Center, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
- Department of General Surgery, The Third People’s Hospital of Chengdu, Chengdu 610031, Sichuan Province, China
| | - Wei Zhao
- Department of Pathology, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Chuan Li
- Department of Liver Surgery and Liver Transplantation Center, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Jun-Yi Shen
- Department of Liver Surgery and Liver Transplantation Center, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Tian-Fu Wen
- Department of Liver Surgery and Liver Transplantation Center, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China
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10
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Guillen Astete CA, Larena Grijalva C. Myofibroblastic sarcoma of the trapezius muscle. ACTA ACUST UNITED AC 2017; 15:e47-e48. [PMID: 28754518 DOI: 10.1016/j.reuma.2017.06.011] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/08/2017] [Revised: 06/08/2017] [Accepted: 06/15/2017] [Indexed: 11/17/2022]
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11
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Han SR, Yee GT. Low Grade Myofibroblastic Sarcoma Occurred in the Scalp. J Korean Neurosurg Soc 2015; 58:385-8. [PMID: 26587195 PMCID: PMC4652002 DOI: 10.3340/jkns.2015.58.4.385] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/09/2015] [Revised: 06/28/2015] [Accepted: 07/03/2015] [Indexed: 11/27/2022] Open
Abstract
Low-grade myofibroblastic sarcoma (LGMS) is a rare sarcoma with myofibroblastic differentiation. LGMS has a propensity for local recurrence and is associated with a low risk of metastatic spread. A 26-year-old man presented with a 12-month history of a slow growing palpable hard mass in the right parietal scalp. Enhanced CT scan of head showed a 3×4 cm sized well-defined and heterogeneously enhancing scalp mass. The patient underwent excision of the tumor. The histological and immunohistochemical features were consistent with a LGMS. We performed re-operation for remnant tumor removal after diagnosis. After 14 months of surgery, the patient was well-being state.
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Affiliation(s)
- Seong Rok Han
- Department of Neurosurgery, Ilsan Paik Hospital, College of Medicine, Inje University, Goyang, Korea
| | - Gi Taek Yee
- Department of Neurosurgery, Gil Medical Center, Gachon University, Incheon, Korea
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12
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Yamada T, Yoshimura T, Kitamura N, Sasabe E, Ohno S, Yamamoto T. Low-grade myofibroblastic sarcoma of the palate. Int J Oral Sci 2012; 4:170-3. [PMID: 22935748 PMCID: PMC3465011 DOI: 10.1038/ijos.2012.49] [Citation(s) in RCA: 30] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/20/2022] Open
Abstract
Low-grade myofibroblastic sarcoma (LGMS) is a rare, malignant tumor with myofibroblastic differentiation. Despite it being classified as a distinct entity by the World Health Organization, a few cases were reported in the oral and maxillofacial region. Here, a LGMS developed on the palate of a 73-year-old man who presented with a 1-cm tumor on the posterior border of the palate. Based on the histological and immunohistochemical features, a diagnosis of LGMS was established. The tumor was resected, and no recurrence was observed over 2 years. Although the tongue is the most preferred site for LGMS, it may occur in any region of the oral cavity.
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Affiliation(s)
- Tomohiro Yamada
- Department of Oral and Maxillofacial Surgery, Kochi Medical School, Kochi University, Kochi, Japan.
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Shenjere P, Eyden B, Banerjee SS, Chakrabarty B, Shanks JH, Sikand KA, Menasce LP. Ultrastructurally confirmed myofibrosarcoma: a series of 10 new cases, with a discussion on diagnostic criteria. Int J Surg Pathol 2012; 21:29-36. [PMID: 22843641 DOI: 10.1177/1066896912454568] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
Some view ultrastructure as key to myofibrosarcoma diagnosis, whereas others argue that electron microscopy is too little used in contemporary practice to be considered an important diagnostic tool. These views are discussed in the context of 10 ultrastructurally confirmed cases of myofibrosarcoma, some occurring at rare sites such as skin and penis. Patient age ranged from 21 to 83 years, with a 6:4 male to female ratio. Size ranged from 2 to 7.5 cm and all had infiltrative margins. Histologically, all consisted of variably cellular fascicles of spindle cells with mild to moderately pleomorphic nuclei, small punctate nucleoli, and eosinophilic cytoplasm. All cases showed α-smooth muscle actin positivity and 2 showed very focal weak positivity for desmin. Ultrastructurally, the tumor cells contained rough endoplasmic reticulum, mainly peripheral smooth-muscle myofilaments, and fibronectin fibrils or fibronexus junctions at the cell surface. The most confident diagnosis of myofibrosarcoma is provided by ultrastructural examination. However, given the right histological appearance, use of a panel of antibodies that includes α-smooth muscle actin, desmin, and h-caldesmon, serves as an acceptable practical way of diagnosing myofibrosarcoma.
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14
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Park KR, Jang HW, Won JH, Kim HS, Cha IH, Kim HJ. Myofibroblastic sarcoma of the mandible: a case report. J Korean Assoc Oral Maxillofac Surg 2012. [DOI: 10.5125/jkaoms.2012.38.4.240] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/07/2022] Open
Affiliation(s)
- Kyung-Ran Park
- Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea
| | - Hyo Won Jang
- Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea
| | - Ji-Hoon Won
- Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea
| | - Hyun-Sil Kim
- Department of Oral Pathology, College of Dentistry, Yonsei University, Seoul, Korea
- Oral Cancer Research Institute, College of Dentistry, Yonsei University, Seoul, Korea
| | - In-Ho Cha
- Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea
- Oral Cancer Research Institute, College of Dentistry, Yonsei University, Seoul, Korea
| | - Hyung Jun Kim
- Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea
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Ni C, Xu YY, Zhou SH, Wang SQ. Differential diagnosis of inflammatory myofibroblastic tumour and low-grade myofibroblastic sarcoma: two case reports with a literature review. J Int Med Res 2011; 39:311-20. [PMID: 21672335 DOI: 10.1177/147323001103900134] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/22/2023] Open
Abstract
Inflammatory myofibroblastic tumour (IMT) and low-grade myofibroblastic sarcoma (LGMS) have similar morpho logical and immunophenotypic features, but LGMS is more malignant than IMT and the treatment requires a wider surgical margin plus post-operative chemotherapy or radiotherapy. To date, only 28 cases of IMT and two cases of LGMS have been reported in the laryngopharynx. Recent studies have suggested that anaplastic lymphoma kinase (ALK) and cytokeratin are important markers for differentiating between the two tumours. Here, two cases involving different myofibroblastic tumours of the larynx are reported. Based on the histological and immunohistochemical results, case 1 was diagnosed as IMT involving the right arytenoepiglottic fold, while case 2 was diagnosed as LGMS involving the epiglottic-glossal surface. There was no recurrence or metastasis in either case after post-operative follow-up (12 and 14 months, respectively). It is difficult to distinguish IMT from LGMS; both morphological and immunohistological analyses are required.
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Affiliation(s)
- C Ni
- Department of Otolaryngology, The First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, China
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