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Kinkopf P, Bowen RC, Laurenti K. Ocular inflammatory myofibroblastic tumor in a pediatric patient. J AAPOS 2024; 28:103821. [PMID: 38242228 DOI: 10.1016/j.jaapos.2023.10.008] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/17/2023] [Revised: 10/16/2023] [Accepted: 10/20/2023] [Indexed: 01/21/2024]
Abstract
Inflammatory myofibroblastic tumor (IMT) is a mesenchymal neoplasm that rarely metastasizes and is more commonly seen in children, adolescents, and young adults than older adults. These tumors, composed of myofibroblasts and inflammatory cells, are often confused for a local infection due to the inflammatory cell infiltration, and they form in mucosal surfaces but rarely arise in the orbit. We present the case of a 6-year-old girl with excisional biopsy-confirmed conjunctival stromal IMT. There was no evidence of recurrence 2 years following resection with no subsequent medical therapy.
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Affiliation(s)
- Paul Kinkopf
- Department of Ophthalmology, Northwestern University Feinberg School of Medicine, Chicago, Illinois.
| | - Randy Christopher Bowen
- Department of Ophthalmology, Northwestern University Feinberg School of Medicine, Chicago, Illinois
| | - Kelly Laurenti
- Department of Ophthalmology, Northwestern University Feinberg School of Medicine, Chicago, Illinois; Division of Ophthalmology, Department of Surgery, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, Illinois
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Benedetti Pedroza J, Carrasco García I, Martínez Bernal G, Miras Rodriguez I. New Perspectives in the Treatment of Inflammatory Myofibroblastic Tumor with ALK Translocation: Case Report. Case Rep Oncol 2024; 17:763-772. [PMID: 39144250 PMCID: PMC11324221 DOI: 10.1159/000539739] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/19/2024] [Accepted: 06/03/2024] [Indexed: 08/16/2024] Open
Abstract
Introduction Inflammatory myofibroblastic tumor (IMT) is a rare entity, classified within soft tissue sarcomas. It is an intermediate malignancy tumor, which seldom presents as metastatic disease. The treatment of choice is surgery, except in cases where surgery is not possible due to localization or if it presents with metastatic disease. Approximately 50% of IMTs will exhibit ALK translocation, providing a therapeutic target for these patients. Case Presentation A case is presented of a patient with metastatic IMT in complete response to treatment with alectinib, maintained for over 4 years. Conclusion This case showed a long time complete response in patient with IMT treated with alectinib.
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Affiliation(s)
| | | | - Gala Martínez Bernal
- Medical Oncology Department, Virgen del Rocio University Hospital, Seville, Spain
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Kawakita Y, Anan K, Kurata K, Koga K, Saimura M, Tamiya S, Nishihara K, Mitsuyama S, Nakano T. Anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the breast: a case report and review of the literature. Surg Case Rep 2023; 9:152. [PMID: 37656266 PMCID: PMC10474000 DOI: 10.1186/s40792-023-01732-6] [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: 04/11/2023] [Accepted: 08/20/2023] [Indexed: 09/02/2023] Open
Abstract
BACKGROUND Few reports of inflammatory myofibroblastic tumor (IMT) of the breast have been published worldwide. Furthermore, primary anaplastic lymphoma kinase (ALK)-positive IMT of the breast is extremely rare. To date, only six patients with ALK-positive IMT have been reported in the literature. CASE PRESENTATION A 52-year-old woman underwent a medical examination, and a left breast mass was detected. She did not feel a mass in her chest. Mammography showed a focal asymmetric density at the lower outer portion of the left breast. Breast ultrasonography showed a 1.2-cm hypoechoic lesion with relatively clear boundaries and poor blood flow. Magnetic resonance imaging and computed tomography revealed a solitary heterogeneous mass in the left breast. Pathologic examination revealed a fibrosing lesion with proliferation of fibroblastic cells arranged in a storiform pattern and admixed inflammatory cells. Immunohistochemical examination showed that the tumor cells were positive for ALK. Under the preoperative diagnosis of IMT, we performed partial mastectomy with adequate margins. The postoperative diagnosis was pathologically confirmed as IMT. Immunohistochemical staining also showed overexpression of ALK-1 in the tumor. The patient had a good clinical course for 24 months postoperatively, without recurrence or metastasis. CONCLUSIONS IMT of the breast shows nonspecific imaging findings, making preoperative diagnosis difficult. Nevertheless, IMT has the characteristics of low-grade neoplasms with recurrence, invasion, and metastatic potential. Our report emphasizes the importance of determining a treatment plan as soon as possible based on an accurate diagnosis to improve the prognosis of this disease.
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Affiliation(s)
- Yasutaka Kawakita
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan.
- Department of Surgery 1, School of Medicine, University of Occupational and Environmental Health, 1-1 Iseigaoka Yahatanishi-Ku, Kitakyushu, Fukuoka, 807-8555, Japan.
| | - Keisei Anan
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
| | - Kanako Kurata
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
| | - Kenichiro Koga
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
| | - Michiyo Saimura
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
| | - Sadafumi Tamiya
- Department of Pathology, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
| | - Kazuyoshi Nishihara
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
| | - Shoshu Mitsuyama
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
| | - Toru Nakano
- Department of Surgery, Kitakyushu Municipal Medical Center, 2-1-1 Bashaku Kokurakita-Ku, Kitakyushu, Fukuoka, 802-0077, Japan
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18F-FDG PET/CT Imaging of G-CSF-Producing Inflammatory Myofibroblastic Tumor of the Pleura. Clin Nucl Med 2023; 48:e84-e86. [PMID: 36607379 DOI: 10.1097/rlu.0000000000004457] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/07/2023]
Abstract
ABSTRACT We report a case of granulocyte colony-stimulating factor (G-CSF)-producing inflammatory myofibroblastic tumor of the pleura in a 71-year-old man. Contrast-enhanced CT demonstrated multiple pleural masses with pulmonary hilar lymph nodes swelling. 18F-FDG PET/CT demonstrated marked focal FDG uptake in the thoracic masses with diffuse uptake in the bone marrow. Based on the pathological findings and elevated serum G-CSF level, the patient was diagnosed as G-CSF-producing inflammatory myofibroblastic tumor. Because G-CSF-producing tumors demonstrate aggressive clinical course, early and accurate diagnosis is important.
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Armijo Sánchez A, Ramírez Muñoz B, Aguilar Gálvez I, García Vidal E, García Mejido J, Iglesias Bravo E. Tumor miofibroblástico inflamatorio tras cáncer de mama. Un reto en el diagnóstico diferencial. Revisión de la literatura. CLINICA E INVESTIGACION EN GINECOLOGIA Y OBSTETRICIA 2022. [DOI: 10.1016/j.gine.2022.100793] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/14/2022]
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Clinicopathological Features of Inflammatory Myofibroblastic Tumor in the Breast. Breast J 2022; 2022:1863123. [PMID: 36304484 PMCID: PMC9578916 DOI: 10.1155/2022/1863123] [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: 05/09/2022] [Accepted: 07/11/2022] [Indexed: 11/17/2022]
Abstract
Inflammatory myofibroblastic tumor (IMT) is a mesenchymal spindle cell tumour with low malignant potential which is extremely rare in breasts. Because of the lack of typical imaging and clinical characteristics of IMT, it is easy to misdiagnose before operation. We now report a case of a 37-year-old woman presenting with a mass in her left breast. Ultrasound showed a well-circumscribed lesion in the lower outer quadrant. The patient underwent lumpectomy, and histopathology revealed a tumor which was composed of fusiform cells and inflammatory cells. Immunohistochemistry (IHC) showed tumor cells are positive for vimentin, ALK, BCL2, and SMA. The FISH test demonstrated ALK (2p23) chromosomal translocation (ALK positive). The final diagnosis of breast IMT was rendered with nonclassical morphology. Postoperative 30-month follow-up no evidence showed residual tumor or recurrence. As a very rare tumor, breast IMT could be easily misdiagnosed clinically and pathologically. Complete surgical resection of the tumor is preferred, and it has the risk of recurrence and metastasis.
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Huang K, Zhao P, Zhao J, Zhao P, Yang J. A unique case of inflammatory myofibroblastic tumor of the liver manifesting with biloma: A case report. Oncol Lett 2022; 24:227. [PMID: 35720485 PMCID: PMC9185161 DOI: 10.3892/ol.2022.13348] [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: 12/24/2021] [Accepted: 04/22/2022] [Indexed: 02/05/2023] Open
Abstract
Anaplastic lymphoma kinase (ALK)-negative hepatic inflammatory myofibroblastic tumors (IMTs) harboring the ETS variant transcription factor 6-neurotrophic receptor tyrosine kinase 3 (ETV6-NTRK3) fusion gene and manifesting with biloma are extremely rare, and their biological behavior is unclear. The present study reports the case of a 45-year-old female with ALK-negative IMT of the liver harboring the ETV6-NTRK3 fusion gene and manifesting with biloma. Computed tomography of the abdomen confirmed the lesions to be a low-density mass, measuring 11.2×8.5×10.5 cm, located in the left lobe of the liver, and a lower-density mass, measuring 8.5×6.1×5.9 cm, located in the interior of the tumor. As the suspicion of a malignancy remained high, surgical resection of the left hepatic lobe, including the tumor, was undertaken. Intraoperatively, a tumor (12×10×9 cm), with an unclear boundary, incomplete capsule and fish-like texture, was found in the left lateral lobe of the liver, and a biloma, measuring 8×6 cm, was identified inside the tumor. Pathological examination revealed spindle cell proliferation with infiltration of chronic inflammatory cells and mucinous degeneration. Immunohistochemical studies showed negativity for ALK, CD117, CD34, discovered on GIST-1, desmin, smooth muscle actin, S-100, CD21, pan-cytokeratin, epithelial membrane antigen, CD23 and CD35, but positivity for vimentin staining, and 5% Ki-67-positive cells. Fluorescence in situ hybridization studies assessing characteristic genetic rearrangements using ALK, RET, ROS1, MDM2, MGEA5 and ETV6 break-apart assays, revealed the presence of the ETV6-NTRK3 fusion oncogene and negativity for ALK, RET, ROS1, MDM2 and MGEA5. The patient was discharged 7 days post-operatively, without any adjuvant treatment. No recurrence of symptoms was noted at the 3-year follow-up. To the best of our knowledge, this is the first report of biloma in an ALK-negative IMT of the liver, which may increase our understanding of hepatic IMT.
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Affiliation(s)
- Kun Huang
- Department of General Surgery, Mianyang Hospital of Traditional Chinese Medicine, Mianyang, Sichuan 621000, P.R. China
| | - Pingwu Zhao
- Department of General Surgery, Mianyang Hospital of Traditional Chinese Medicine, Mianyang, Sichuan 621000, P.R. China
| | - Jiangying Zhao
- Department of Pathology, Mianyang Hospital of Traditional Chinese Medicine, Mianyang, Sichuan 621000, P.R. China
| | - Pan Zhao
- Department of General Surgery, Mianyang Hospital of Traditional Chinese Medicine, Mianyang, Sichuan 621000, P.R. China
| | - Jian Yang
- Department of Liver Surgery, West China Hospital, Sichuan University, Chengdu, Sichuan 610000, P.R. China
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Zhou P, Chen YH, Lu JH, Jin CC, Xu XH, Gong XH. Inflammatory myofibroblastic tumor after breast prosthesis: A case report and literature review. World J Clin Cases 2022; 10:1432-1440. [PMID: 35211580 PMCID: PMC8855174 DOI: 10.12998/wjcc.v10.i4.1432] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/05/2021] [Revised: 10/21/2021] [Accepted: 12/23/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Inflammatory myofibroblastic tumors (IMTs) are defined as tumors composed of differentiated myofibroblastic spindle cells, usually accompanied by numerous plasma cells and lymphocytes, and classified as intermediate (occasionally metastatic) by the World Health Organization. Its pathogenesis and biological behavior have not yet been elucidated. Breast IMT is extremely rare, and prosthesis implantation combined with IMT has not been reported. This study reports a case of IMT following resection of a malignant phyllodes tumor of the left breast and implantation of a prosthesis.
CASE SUMMARY A 41-year-old female presented to our hospital with a mass in the left breast for 3 mo. The patient had undergone resection of a large mass in her left breast pathologically diagnosed as a malignant phyllodes tumor and implantation of a prosthesis five years prior. Ultrasonic examination revealed an oval mass in the left breast, and the patient underwent left breast mass resection and prosthesis removal. Light microscopy revealed the spindle cells to be diffusely proliferated, with a large number of neutrophils, lymphocytes, and plasma cell infiltration. Immunohistochemical staining revealed that the spindle cells were partially positive for smooth muscle actin, which is positive for BCL-2 and cluster of differentiation (CD) 99 but were negative for anaplastic lymphoma kinase, cytokeratin, S-100 protein, desmin, and CD34. The final diagnosis was IMT. No recurrence or metastasis was observed during the 5-year postoperative follow-up.
CONCLUSION Prosthesis implantation may be one of the causes of IMT, but further investigation is necessary to prove it.
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Affiliation(s)
- Peng Zhou
- Department of Ultrasound, Shenzhen Second People’s Hospital, The First Affiliated Hospital of Shenzhen University, Shenzhen 518035, Guangdong Province, China
| | - Yi-Hao Chen
- Department of Ultrasound, Shenzhen Second People’s Hospital, The First Affiliated Hospital of Shenzhen University, Shenzhen 518035, Guangdong Province, China
- Graduate School, Guangdong Medical University, Zhanjiang 524023, Guangdong Province, China
| | - Jiang-Hao Lu
- Department of Ultrasound, Shenzhen Second People’s Hospital, The First Affiliated Hospital of Shenzhen University, Shenzhen 518035, Guangdong Province, China
| | - Chun-Chun Jin
- Department of Ultrasound, Shenzhen Second People’s Hospital, The First Affiliated Hospital of Shenzhen University, Shenzhen 518035, Guangdong Province, China
| | - Xiao-Hong Xu
- Graduate School, Guangdong Medical University, Zhanjiang 524023, Guangdong Province, China
- Department of Ultrasound, Guangdong Medical University Affiliated Hospital, Zhanjiang 524001, Guangdong Province, China
| | - Xue-Hao Gong
- Department of Ultrasound, Shenzhen Second People’s Hospital, The First Affiliated Hospital of Shenzhen University, Shenzhen 518035, Guangdong Province, China
- Graduate School, Guangdong Medical University, Zhanjiang 524023, Guangdong Province, China
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Chen ZT, Lin YX, Li MX, Zhang T, Wan DL, Lin SZ. Inflammatory myofibroblastic tumor of the pancreatic neck: A case report and review of literature. World J Clin Cases 2021; 9:6418-6427. [PMID: 34435007 PMCID: PMC8362560 DOI: 10.12998/wjcc.v9.i22.6418] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/18/2021] [Revised: 03/23/2021] [Accepted: 06/01/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Pancreatic inflammatory myofibroblastic tumor (IMT) is a relatively rare disease that is often confused with pancreatic cancer or pancreatic neuroendocrine tumors. The histological features of IMTs show that tissue from this type of tumor contains an intermingling of fibroblast and myofibroblast proliferation, accompanied by a varying degree of inflammatory cell infiltration.
CASE SUMMARY The management of an IMT occurring at the neck of the pancreas is presented in this paper. A 66-year-old female patient was diagnosed with a pancreatic neck mass after a series of tests. The patient underwent enucleation of the pancreatic neck tumor after a pathological diagnosis of IMT. Previous research on the clinical features, pathological diagnosis and treatment of pancreatic IMTs was reviewed. Compared with previous reports, this is a unique case of enucleation of a pancreatic IMT.
CONCLUSION The enucleation of pancreatic IMTs may be a safe and efficient surgical method for managing such tumors with a better prognosis. Further cases are required to explore surgical measures for pancreatic IMTs.
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Affiliation(s)
- Zhi-Tao Chen
- Department of Hepatobiliary and Pancreatic Surgery, Shulan (Hangzhou) Hospital Affiliated to Zhejiang Shuren University Shulan International Medical College, Hangzhou 310000, Zhejiang Province, China
- School of Medicine, Zhejiang University, Hangzhou 310000, Zhejiang Province, China
| | - Yao-Xiang Lin
- School of Medicine, Hangzhou Normal University, Hangzhou 310003, Zhejiang Province, China
| | - Meng-Xia Li
- School of Medicine, Zhejiang University, Hangzhou 310000, Zhejiang Province, China
- Department of Hepatobiliary and Pancreatic Surgery, First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
| | - Ting Zhang
- Department of Pathology, Shulan (Hangzhou) Hospital Affiliated to Zhejiang Shuren University Shulan International Medical College, Hangzhou 310000, Zhejiang Province, China
| | - Da-Long Wan
- Department of Hepatobiliary and Pancreatic Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
| | - Sheng-Zhang Lin
- Department of Hepatobiliary Surgery, Shulan (Hangzhou) Hospital Affiliated to Zhejiang Shuren University Shulan International Medical College, Hangzhou 310000, Zhejiang Province, China
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Liu R, Li H, Deng J, Wu Q, Liao C, Xiao Q, Chang Q. QKI 6 ameliorates CIRI through promoting synthesis of triglyceride in neuron and inhibiting neuronal apoptosis associated with SIRT1-PPARγ-PGC-1α axis. Brain Behav 2021; 11:e2271. [PMID: 34227244 PMCID: PMC8413718 DOI: 10.1002/brb3.2271] [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] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/05/2021] [Revised: 06/17/2021] [Accepted: 06/17/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND The stroke induced by ischemia of brain remains high incidence and death rate. The study wanted to confirm the effects of Quaking 6 (QKI 6) on the protection role in neurons of rat model of cerebral ischemia/reperfusion injury (CIRI). MATERIAL AND METHODS The rat model with CIRI induced by middle cerebral artery occlusion was well established and rat neurons were isolated to characterize the effects of QKI 6 mediated by sirtuin 1 (SIRT1) on synthesis of triglyceride in neuron and neuronal apoptosis via activation of SIRT1-peroxisome proliferater-activated receptor (PPAR)γ- peroxisome proliferator-activated receptor coactivator (PGC)-1α signaling pathway. RESULTS The expression levels of SIRT1 or QKI 6, and acetylation level of QKI 6 were decreased in neurons of rat model with CIRI. QKI 6 deacetylated and mediated by SIRT1 that contributed to suppressing the progression of neuronal apoptosis in rat through promoting synthesis of triglyceride in vivo and in vitro via SIRT1-PPARγ-PGC-1α signaling pathway, then inhibiting CIRI. CONCLUSIONS Our results demonstrated SIRT1 deacetylates QKI 6, the RNA-binding protein, that affects significantly the synthesis of triglyceride in neurons of CIRI rat model. Moreover, it activated transcription factor peroxisome proliferator-activated receptorγ coactivator-1α (PGC-1α) through post-transcriptional regulation of the expression of PPARγ, and further enhanced synthesis of triglyceride, thereby restrained the progression of neural apoptosis and CIRI.
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Affiliation(s)
- Rui Liu
- Department of Rehabilitation, Tangdu Hospital, Fourth Military Medical University, Xi'an, PR China
| | - Hongzeng Li
- Department of Gerontology, Tangdu Hospital, Fourth Military Medical University, Xi'an, PR China
| | - Jingyuan Deng
- Department of Encephalology, the First Affiliated Hospital, Medical School of Xi'an Jiaotong University, Xi'an, PR China
| | - Qunqiang Wu
- Department of Rehabilitation, Tangdu Hospital, Fourth Military Medical University, Xi'an, PR China
| | - Chunhua Liao
- Department of Rehabilitation, Tangdu Hospital, Fourth Military Medical University, Xi'an, PR China
| | - Qun Xiao
- Department of Rehabilitation, Tangdu Hospital, Fourth Military Medical University, Xi'an, PR China
| | - Qi Chang
- Department of Orthopaedics, Xijing Hospital, Fourth Military Medical University, Xi'an, PR China.,Department of Orthopaedics, The 150th Central Hospital of Chinese People's Liberation Army, Luoyang, PR China
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Preclinical In Vivo Modeling of Pediatric Sarcoma-Promises and Limitations. J Clin Med 2021; 10:jcm10081578. [PMID: 33918045 PMCID: PMC8069549 DOI: 10.3390/jcm10081578] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/27/2021] [Revised: 04/05/2021] [Accepted: 04/06/2021] [Indexed: 02/07/2023] Open
Abstract
Pediatric sarcomas are an extremely heterogeneous group of genetically distinct diseases. Despite the increasing knowledge on their molecular makeup in recent years, true therapeutic advancements are largely lacking and prognosis often remains dim, particularly for relapsed and metastasized patients. Since this is largely due to the lack of suitable model systems as a prerequisite to develop and assess novel therapeutics, we here review the available approaches to model sarcoma in vivo. We focused on genetically engineered and patient-derived mouse models, compared strengths and weaknesses, and finally explored possibilities and limitations to utilize these models to advance both biological understanding as well as clinical diagnosis and therapy.
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Magro G, Salvatorelli L, Puzzo L, Piombino E, Bartoloni G, Broggi G, Vecchio GM. Practical approach to diagnosis of bland-looking spindle cell lesions of the breast. Pathologica 2020; 111:344-360. [PMID: 31965112 PMCID: PMC8145669 DOI: 10.32074/1591-951x-31-19] [Citation(s) in RCA: 17] [Impact Index Per Article: 3.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/24/2019] [Accepted: 10/28/2019] [Indexed: 12/22/2022] Open
Abstract
The diagnosis of bland-looking spindle cell lesions of the breast is often challenging because there is a close morphological and immunohistochemical overlap among the different entities. The present review will discuss reactive spindle cell nodule/exuberant scar, nodular fasciitis, inflammatory pseudotumor, myofibroblastoma (classic type), lipomatous myofibroblastoma, palisaded myofibroblastoma, benign fibroblastic spindle cell tumor, spindle cell lipoma, fibroma, leiomyoma, solitary fibrous tumor, myxoma, schwannoma/neurofibroma, desmoid-type fibromatosis, dermatofibrosarcoma protuberans, low-grade fibromatosis-like spindle cell carcinoma, inflammatory myofibroblastic tumor and low-grade myofibroblastic sarcoma arising in the breast parenchyma. The pathologist should be aware of each single lesion to achieve a correct diagnosis to ensure patient a correct prognostic information and therapy. Accordingly representative illustrations and morphological/immunohistochemical diagnostic clues will be provided.
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Affiliation(s)
- G Magro
- Department of Medical and Surgical Sciences and Advanced Technologies, G.F. Ingrassia, Azienda Ospedaliero-Universitaria "Policlinico Vittorio Emanuele", Anatomic Pathology, School of Medicine, University of Catania, Italy
| | - L Salvatorelli
- Department of Medical and Surgical Sciences and Advanced Technologies, G.F. Ingrassia, Azienda Ospedaliero-Universitaria "Policlinico Vittorio Emanuele", Anatomic Pathology, School of Medicine, University of Catania, Italy
| | - L Puzzo
- Department of Medical and Surgical Sciences and Advanced Technologies, G.F. Ingrassia, Azienda Ospedaliero-Universitaria "Policlinico Vittorio Emanuele", Anatomic Pathology, School of Medicine, University of Catania, Italy
| | - E Piombino
- Department of Medical and Surgical Sciences and Advanced Technologies, G.F. Ingrassia, Azienda Ospedaliero-Universitaria "Policlinico Vittorio Emanuele", Anatomic Pathology, School of Medicine, University of Catania, Italy
| | - G Bartoloni
- Anatomic Pathology, A.R.N.A.S. Garibaldi-Nesima, Catania, Italy
| | - G Broggi
- Department of Medical and Surgical Sciences and Advanced Technologies, G.F. Ingrassia, Azienda Ospedaliero-Universitaria "Policlinico Vittorio Emanuele", Anatomic Pathology, School of Medicine, University of Catania, Italy
| | - G M Vecchio
- Department of Medical and Surgical Sciences and Advanced Technologies, G.F. Ingrassia, Azienda Ospedaliero-Universitaria "Policlinico Vittorio Emanuele", Anatomic Pathology, School of Medicine, University of Catania, Italy
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Liu Q, Wei J, Liu X, Wang J. Anaplastic lymphoma kinase-negative pulmonary inflammatory myofibroblastic tumor with multiple metastases and its treatment by Apatinib: A case report. Medicine (Baltimore) 2019; 98:e18414. [PMID: 31876714 PMCID: PMC6946427 DOI: 10.1097/md.0000000000018414] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/30/2022] Open
Abstract
RATIONALE Primary pulmonary inflammatory myofibroblastic tumor (IMT) with distant metastasis is extremely rare. Moreover, metastasis of pulmonary IMT to bone marrow has never been reported in previous studies. Therapeutic approaches for anaplastic lymphoma kinase (ALK)-negative pulmonary IMT with metastasis are limited. Yet there is no report on the treatment of advanced IMT cases with anti-angiogenesis drugs. PATIENT CONCERNS We described a patient with a complaint of fatigue, with the chest computed tomography (CT) scan revealing 2 masses in bilateral lung. DIAGNOSES The CT-guided lung biopsy examined 1 lesion in the right lung, and the post-operative pathological diagnosis of ALK-negative pulmonary IMT was recommended. However, the lung lesions were found significantly enlarged during the subsequent visit 8 months later, along with multiple metastases to the bone and abdominal cavity. A bone marrow biopsy revealed bone marrow infiltration by spindle cells. INTERVENTIONS The patient began to take Celecoxib due to the rapid progression of IMT, however, resulting in the aggravated gastric ulcer. He stopped taking the medicine 1 month later, with no remarkable change in the lesions by CT. Apatinib was administrated instead of Celecoxib. OUTCOMES After the 5-month treatment of Apatinib, the mass in the abdominal cavity significantly shrank and the lung lesions slightly decreased in size. With the 9-month administration of Apatinib, the lung lesions and the abdominal mass kept stable, compared with the situation in the 5-month follow-up. LESSONS Although pulmonary IMT shows the potential of metastasis, its metastasizing to bone marrow is a highly unusual event. Apatinib is effective for pulmonary IMT, and should be taken into consideration for the treatment of inoperable pulmonary IMT patients who lack ALK rearrangement.
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Affiliation(s)
| | - Jianguo Wei
- Department of Pathology, Shaoxing People's Hospital, Shaoxing Hospital of Zhejiang University, Zhejiang
| | - Xizhong Liu
- Institute of Nonlinear Science, Shaoxing University, Shaoxing, China
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Zeng J, He RQ, Mo WG, Peng ZG, Ma J, Zhong JC, Mo CH, Qin MJ, Hu XH. Inflammatory myofibroblastic tumor of the prostate after transurethral resection of the prostate with negative expression of anaplastic lymphoma kinase: a case report. SAO PAULO MED J 2018; 136:484-487. [PMID: 29898011 PMCID: PMC9907760 DOI: 10.1590/1516-3180.2017.0079070417] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/16/2017] [Accepted: 04/07/2017] [Indexed: 12/16/2022] Open
Abstract
CONTEXT Inflammatory myofibroblastic tumors are a rare type of soft-tissue tumor. Inflammatory myofibroblastic tumors are characterized by rearrangements involving the anaplastic lymphoma kinase gene locus on 2p23. CASE REPORT We report the case of a 67-year-old Chinese male who presented with dysuria and fever. Magnetic resonance imaging showed an irregular prostatic mass with an isointense signal and obscure boundary. Histopathological evaluation showed that the mass consisted mainly of spindle-shaped cells. Immunohistochemical evaluation showed that the tumor cells were negative for anaplastic lymphoma kinase. CONCLUSIONS Inflammatory myofibroblastic prostate tumors are rare lesions with unclear etiology. The pathological diagnosis is very important.
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Affiliation(s)
- Jie Zeng
- MD, MSc. Postgraduate Student, Department of Medical Oncology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Rong-Quan He
- MD. Postgraduate Student, Department of Medical Oncology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Wei-Guang Mo
- MSc. Technician, Department of Pathology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Zhi-Gang Peng
- MD, MSc. Professor, Department of Medical Oncology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Jie Ma
- MD, MSc. Professor, Department of Medical Oncology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Jin-Cai Zhong
- MD, MSc. Professor, Department of Medical Oncology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Chao-Hua Mo
- MD, MSc. Attending Physician and Postgraduate Student, Department of Pathology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Mei-Jiao Qin
- MD, MSc. Postgraduate Student, Department of Medical Oncology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
| | - Xiao-Hua Hu
- MD, MSc. Professor, Department of Medical Oncology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, China.
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15
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Mao X, Liu H, Du J, Yu N, Chen L, Zhang L. Imaging findings of inflammatory myofibroblastic tumor in breast: A case report. Medicine (Baltimore) 2018; 97:e11804. [PMID: 30095645 PMCID: PMC6133435 DOI: 10.1097/md.0000000000011804] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/26/2018] [Accepted: 07/17/2018] [Indexed: 12/15/2022] Open
Abstract
RATIONALE Inflammatory myofibroblastic tumors (IMTs), particularly breast IMTs, are rare neoplastic lesions typically associated with a favorable prognosis. Breast IMTs are easily misdiagnosed as other types of malignant lesions, and therefore there is a significant unmet need for a better preoperational differential diagnosis based on imaging manifestations. Here, we report the imaging findings of a breast IMT and compare our findings with previously published features of breast IMTs. PATIENT CONCERNS The patient, a 43-year-old female, reported the presence of a palpable lump within her left breast. An ultrasound examination revealed an irregular hypoechoic mass with unclear boundaries. Mammography demonstrated a mass of heterogeneous and striped density with granular calcification. Magnetic resonance image (MRI) inspection displayed an irregular tissue lump with an undistinguishable boundary and a further dynamic contrast-enhanced MRI disclosed an associated efflux change. DIAGNOSES Breast inflammatory myofibroblastic tumors. INTERVENTIONS Breast needle biopsy and mammary resection were performed. Pathological staining of the bulk resected tumor after preoperative preparation revealed that the tumor-like tissue was enriched for spindle cells arranged in fascicular clusters. Histopathological diagnosis and immunohistochemistry confirmed the mass as being a breast IMT. OUTCOMES No metastatic recurrence was found during 6-month or 1-year follow-ups. LESSONS Breast IMTs commonly develop in elderly women with atypical imaging features. They are primarily composed of lobular soft tissues infiltrated with an abundant focal blood supply and granular calcification. Development of breast IMTs is closely related to trauma. A preliminary diagnosis of such masses can be made based on combined manifestations of both clinical and imaging features, while a final confirmation still requires pathological staining. Imaging examinations are of value for such tumors to define the lesion edges and their associations with adjacent tissues.
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Affiliation(s)
| | | | | | - Ning Yu
- Department of Pathology, Binzhou Medical University Hospital, Binzhou, Shandong, China
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16
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Fernández-Aceñero MJ, Rejas M, Vázquez Á, Varela S, Jiménez-Ayala B. Tumor miofibroblástico inflamatorio de mama: una entidad poco frecuente. REVISTA ESPAÑOLA DE PATOLOGÍA 2018; 51:193-196. [PMID: 30012314 DOI: 10.1016/j.patol.2017.09.003] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Received: 07/02/2017] [Revised: 09/10/2017] [Accepted: 09/16/2017] [Indexed: 01/14/2023]
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17
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Whorms DS, Fishman MDC, Slanetz PJ. Mesenchymal Lesions of the Breast: What Radiologists Need to Know. AJR Am J Roentgenol 2018; 211:224-233. [PMID: 29792741 DOI: 10.2214/ajr.17.19020] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/18/2022]
Abstract
OBJECTIVE Mesenchymal breast tumors originate from the various components of mammary stroma. The aim of this review is to discuss the clinical presentation, imaging appearance, and management of mesenchymal breast lesions. CONCLUSION Although many mesenchymal tumors exhibit characteristic findings on imaging, others show nonspecific characteristics and require tissue biopsy for diagnosis. An awareness of the clinical and imaging presentation is essential in guiding the differential diagnosis and patient management.
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Affiliation(s)
- Debra S Whorms
- 1 Harvard Medical School, 25 Shattuck St, Boston, MA 02215
| | - Michael D C Fishman
- 1 Harvard Medical School, 25 Shattuck St, Boston, MA 02215
- 2 Department of Radiology, Division of Breast Imaging, Beth Israel Deaconess Medical Center, Boston, MA
- 3 Present address: Department of Radiology, Division of Breast Imaging, Boston Medical Center, Boston, MA
| | - Priscilla J Slanetz
- 1 Harvard Medical School, 25 Shattuck St, Boston, MA 02215
- 2 Department of Radiology, Division of Breast Imaging, Beth Israel Deaconess Medical Center, Boston, MA
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18
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Abstract
Spindle cell lesions of the breast cover a wide spectrum of diseases ranging from reactive tumor-like lesions to high-grade malignant tumors. The recognition of the benign spindle cell tumor-like lesions (nodular fasciitis; reactive spindle cell nodule after biopsy, inflammatory pseudotumor/inflammatory myofibroblastic tumor; fascicular variant of pseudoangiomatous stromal hyperplasia) and tumors (myofibroblastoma, benign fibroblastic spindle cell tumor, leiomyoma, schwannoma, spindle cell lipoma, solitary fibrous tumor, myxoma) is crucial to avoid confusion with morphologically similar but more aggressive bland-appearing spindle cell tumors, such as desmoid-type fibromatosis, low-grade (fibromatosis-like) spindle cell carcinoma, low-grade fibrosarcoma/myofibroblastic sarcoma and dermatofibrosarcoma protuberans.
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Affiliation(s)
- Gaetano Magro
- Department of Medical and Surgical Sciences and Advanced Technologies, G.F. Ingrassia, Anatomic Pathology, University of Catania, Via S. Sofia 87, Catania 95123, Italy.
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19
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Inoue M, Ohta T, Shioya H, Sato S, Takahashi H, Nakata N, Taniguchi C, Hirano M, Nishioka M, Yamakawa H. Inflammatory myofibroblastic tumors of the breast with simultaneous intracranial, lung, and pancreas involvement: ultrasonographic findings and a review of the literature. J Med Ultrason (2001) 2017; 45:331-335. [PMID: 29027063 DOI: 10.1007/s10396-017-0829-y] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/09/2017] [Accepted: 08/21/2017] [Indexed: 12/21/2022]
Abstract
We encountered a case of inflammatory myofibroblastic tumor (IMT) of the breast with simultaneous intracranial, lung, and pancreas involvement. Here, we present the clinical imaging results and report the significance of sonographic findings of breast IMT along with a review of the literature. A 16-year-old girl with a history of subarachnoidal hemorrhage was admitted to our hospital due to tonic-clonic seizure. Computed tomography (CT) and magnetic resonance imaging (MRI) showed multiple intracranial, lung, and pancreas mass lesions and a solitary mass lesion in the right breast. Breast ultrasonography showed a circumscribed oval-shaped hypoechoic mass with a central hyperechoic region. Power Doppler sonography revealed an unusual spiral-shaped flow signal. Breast tumorectomy was performed for definitive diagnosis, and pathological analysis indicated IMT. A literature review indicated that ultrasonographic findings of IMT of the breast are nonspecific, as in other systems or organs. It would be difficult to make a diagnosis of IMT of the breast preoperatively due to its rarity and the lack of specificity of clinical imaging findings. In addition, it is better to consider the possibility of IMT of the breast especially in younger patients without an obvious family history of hereditary breast cancer.
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Affiliation(s)
- Mari Inoue
- Department of Radiology, Diagnostic Ultrasound Center, Jikei University School of Medicine, 3-19-18 Nishi-Shinbashi, Minato-ku, Tokyo, 105-8471, Japan.
| | - Tomoyuki Ohta
- Department of Radiology, Diagnostic Ultrasound Center, Jikei University School of Medicine, 3-19-18 Nishi-Shinbashi, Minato-ku, Tokyo, 105-8471, Japan
| | - Hisashi Shioya
- Departments of Breast and Endocrine Surgery, Jikei University School of Medicine, Tokyo, Japan
| | - Shun Sato
- Department of Pathology, Jikei University School of Medicine, Tokyo, Japan
| | - Hiroyuki Takahashi
- Department of Pathology, Jikei University School of Medicine, Tokyo, Japan
| | - Norio Nakata
- Department of Radiology, Diagnostic Ultrasound Center, Jikei University School of Medicine, 3-19-18 Nishi-Shinbashi, Minato-ku, Tokyo, 105-8471, Japan
| | - Chiaki Taniguchi
- Department of Radiology, Diagnostic Ultrasound Center, Jikei University School of Medicine, 3-19-18 Nishi-Shinbashi, Minato-ku, Tokyo, 105-8471, Japan
| | - Megumi Hirano
- Department of Radiology, Diagnostic Ultrasound Center, Jikei University School of Medicine, 3-19-18 Nishi-Shinbashi, Minato-ku, Tokyo, 105-8471, Japan
| | - Makiko Nishioka
- Department of Radiology, Diagnostic Ultrasound Center, Jikei University School of Medicine, 3-19-18 Nishi-Shinbashi, Minato-ku, Tokyo, 105-8471, Japan
| | - Hironori Yamakawa
- Department of Radiology, Diagnostic Ultrasound Center, Jikei University School of Medicine, 3-19-18 Nishi-Shinbashi, Minato-ku, Tokyo, 105-8471, Japan
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20
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Siraj F, Kaur M, Dalal V, Sonam J. Inflammatory Myofibroblastic Tumor of the Breast Mimicking Malignancy in an Elderly Male. Ochsner J 2017; 17:277-279. [PMID: 29026362 PMCID: PMC5625989] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/07/2023] Open
Abstract
BACKGROUND Inflammatory myofibroblastic tumor (IMT) is a rare, distinctive lesion composed of a proliferation of myofibroblastic spindle cells accompanied by an inflammatory infiltrate. It was first described in the lung, but its occurrence at various extrapulmonary sites has also been reported. The literature mentions only a handful of cases of IMT in the breast and only 1 case in the male breast. We report the second case of IMT in the male breast. CASE REPORT A 60-year-old male presented with a large, lobulated lump in the left breast that had progressively increased in size during the past year. The lump measured 15 × 10 cm. Ultrasonography revealed a solid mass lesion with regular borders in the subcutaneous plane of the left anterior chest wall. Fine-needle aspiration cytology showed a cellular mesenchymal tumor. Macroscopically, the nodule was firm, circumscribed, and yellow. On microscopic examination, the tumor was composed of bland spindle cells arranged in sheets and short fascicles along with a rich inflammatory infiltrate comprising predominantly plasma cells, admixed with lymphocytes, neutrophils, and eosinophils. On immunohistochemistry, the tumor cells were positive for vimentin, focally positive for smooth muscle antigen, and negative for anaplastic lymphoma kinase, CD34, S100, β-catenin, and cytokeratin. Thus, a final diagnosis of IMT was rendered. CONCLUSION IMT is a rare entity with intermediate clinical behavior. Knowledge of this entity and its recurrence and metastatic potential is of paramount significance to guide appropriate treatment and follow-up.
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Affiliation(s)
- Fouzia Siraj
- National Institute of Pathology, Indian Council of Medical Research, Safdarjung Hospital, New Delhi, India
| | - Manveen Kaur
- National Institute of Pathology, Indian Council of Medical Research, Safdarjung Hospital, New Delhi, India
| | - Varsha Dalal
- National Institute of Pathology, Indian Council of Medical Research, Safdarjung Hospital, New Delhi, India
| | - Jain Sonam
- National Institute of Pathology, Indian Council of Medical Research, Safdarjung Hospital, New Delhi, India
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21
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Marylilly S, Subachitra T, Ramya V. Inflammatory Myofibroblastic Tumour of Thyroid with its Prominent Spindle Cell Pattern: A Rare Case Report. J Clin Diagn Res 2016; 10:ED05-7. [PMID: 27190815 DOI: 10.7860/jcdr/2016/15159.7558] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/09/2015] [Accepted: 01/31/2016] [Indexed: 12/26/2022]
Abstract
Inflammatory myofibroblastic tumour of thyroid is very rare. Only 18 cases reported so far. Here we report a case of Inflammatory myofibroblastic tumour with its prominent spindle cell (fibrohistiocytic) pattern in a 61-year-old male patient. The dominant histological pattern in our case was myofibroblastic in contrast to prominent lymphoplasmocytic pattern in other previously reported cases. The tumour was strongly positive for vimentin, Anaplastic lymphoma kinase and showed focal positivity for Smooth Muscle Actin. The patient was treated with total thyroidectomy and he is comfortable after surgery.
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Affiliation(s)
- S Marylilly
- Professor, Department of Pathology, Stanley Medical College , Chennai, Tamil Nadu, India
| | - T Subachitra
- Assistant Professor, Department of Pathology, Stanley Medical College , Chennai, Tamil Nadu, India
| | - V Ramya
- Postgraduate, Department of Pathology, Stanley Medical College , Chennai, Tamil Nadu, India
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22
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ZHANG TIAN, YUAN YAWEI, REN CHEN, DU SHASHA, CHEN JIARONG, SUN QUANQUAN, LIU ZHENGJUN. Recurrent inflammatory myofibroblastic tumor of the inguinal region: A case report and review of the literature. Oncol Lett 2015; 10:675-680. [PMID: 26622552 PMCID: PMC4509014 DOI: 10.3892/ol.2015.3297] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/08/2014] [Accepted: 04/24/2015] [Indexed: 12/17/2022] Open
Abstract
Inflammatory myofibroblastic tumors (IMTs) of the inguinal region are exceptionally rare. The current study reported the case of a 49 year-old male patient with IMT, who presented with a fever, night sweats, anorexia, loss of weight and frequent urination. Computed tomography (CT) revealed a lesion occupying the soft tissue of the right inguinal region and surgery was performed to resect the lesion. Histopathological analysis of the lesion revealed a composition of spindle and inflammatory cells, including plasma cells and lymphocytes. In addition, immunohistochemical analysis demonstrated that the tumor cells were positive for CD34, vimentin, actin, Ki-67, B cell lymphoma-2, CD99, epithelial membrane antigen and CD38; however, tumor cells were negative for CD117, desmin, anaplastic lymphoma kinase and creatine kinase. Thus, the patient was diagnosed with IMT and was advised to return for regular follow-up appointments. Subsequently, the patient developed a local recurrence 12 months following the initial surgery. Of note, the histopathological characteristics of the recurrent lesions were consistent with those of the initial specimen. Thus, a second surgery was performed, followed by fractionated radiotherapy (FRT). At 3 and 6 months following the FRT, magnetic resonance imaging scans did not indicate tumor recurrence or metastasis. In conclusion, surgical excision is the current recommended treatment for IMT; however, for cases similar to that of the current study, which are not successfully controlled by surgical excision, radiotherapy should be considered and long-term follow-up is essential.
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Affiliation(s)
- TIAN ZHANG
- Department of Radiation Oncology, Nanfang Hospital, Southern Medical University, Guangzhou, Guangdong 510515, P.R. China
- Department of Radiology, Tianjin Medical University Cancer Institute and Hospital, National Clinical Research Center for Cancer, Key Laboratory of Cancer Prevention and Therapy, Tianjin 300060, P.R. China
| | - YAWEI YUAN
- Department of Radiation Oncology, Nanfang Hospital, Southern Medical University, Guangzhou, Guangdong 510515, P.R. China
| | - CHEN REN
- Department of Radiation Oncology, Nanfang Hospital, Southern Medical University, Guangzhou, Guangdong 510515, P.R. China
| | - SHASHA DU
- Department of Radiation Oncology, Nanfang Hospital, Southern Medical University, Guangzhou, Guangdong 510515, P.R. China
| | - JIARONG CHEN
- Department of Radiation Oncology, Nanfang Hospital, Southern Medical University, Guangzhou, Guangdong 510515, P.R. China
| | - QUANQUAN SUN
- Department of Radiation Oncology, Nanfang Hospital, Southern Medical University, Guangzhou, Guangdong 510515, P.R. China
| | - ZHENGJUN LIU
- Department of Vascular Surgery, Nanfang Hospital, Southern Medical University, Guangzhou, Guangdong 510515, P.R. China
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23
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Choi EJ, Jin GY, Chung MJ, Moon WS, Youn HJ. Primary Inflammatory Myofibroblastic Tumors of the Breast with Metastasis: Radiographic and Histopathologic Predictive Factors. J Breast Cancer 2015; 18:200-5. [PMID: 26155298 PMCID: PMC4490271 DOI: 10.4048/jbc.2015.18.2.200] [Citation(s) in RCA: 17] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/01/2015] [Accepted: 03/04/2015] [Indexed: 12/13/2022] Open
Abstract
Primary inflammatory myofibroblastic tumors (IMTs) of the breast are uncommon and metastasis of IMTs is extremely rare. To date, the natural course of this disease is not fully understood. Although patients with IMTs should undergo regular follow-up after complete surgical resection of the tumor, the appropriate interval and method of follow-up are unclear. We report the case of a patient with an IMT of the breast that metastasized 2 years after complete surgical resection. This unusual case emphasizes the importance of preoperative examinations to determine whether the IMT has atypical features that should guide the interval and method of follow-up.
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Affiliation(s)
- Eun Jung Choi
- Department of Radiology, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea
| | - Gong Yong Jin
- Department of Radiology, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea
| | - Myoung Ja Chung
- Department of Pathology, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea
| | - Woo Sung Moon
- Department of Pathology, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea
| | - Hyun Jo Youn
- Department of Surgery, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea
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24
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Wu S, Xu R, Wan Q, Zhu X, Zhang L, Jiang H, Zhao X. Assessment of the potential diagnostic role of anaplastic lymphoma kinase for inflammatory myofibroblastic tumours: a meta-analysis. PLoS One 2015; 10:e0125087. [PMID: 25910080 PMCID: PMC4409171 DOI: 10.1371/journal.pone.0125087] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/06/2014] [Accepted: 03/20/2015] [Indexed: 12/15/2022] Open
Abstract
Objective To assess the value of anaplastic lymphoma kinase for the diagnosis of inflammatory myofibroblastic tumours using a comprehensive meta-analysis. Methods We searched the related literature using electronic databases and manual searches. Approximately 454 cases from several countries were included in this analysis. The quality of studies included was assessed by QUADAS (quality assessment of studies of diagnostic accuracy). The diagnostic odds ratio (DOR), positive likelihood ratio (PLR), negative likelihood ratio (NLR), sensitivity and specificity were calculated to assess the role of anaplastic lymphoma kinase in the diagnosis of inflammatory myofibroblastic tumours. The overall test performance was summarised by an SROC (summary receiver operating characteristic curve). The heterogeneity and publication bias were analysed using Meta-regression and Deeks' test. All data were analysed by Stata 12.0 software. Results Eight studies were included according to our inclusion criteria. The overall results for the specificity, sensitivity, PLR, NLR, DOR and area under the curve (AUC) were 0.99 (95% CI 0.82-1.00), 0.67 (95% CI 0.46-0.83), 0.67 (95% CI 0.46-0.83), 60.6 (95% CI 3.3-1112.4), 0.33 (95% CI 0.19-0.60), 181 (95% CI 9-3684) and 0.95 (95% CI 0.93-0.97), respectively, while the specificity, sensitivity, PLR, NLR, DOR and AUC for bladder IMTs were 0.99 (95% CI 0.67-1.00), 0.86 (95% CI 0.58-0.96), 95.6 (95% CI 2.0-4616.2), 0.14 (95% CI 0.04-0.50), 671 (95% CI 16-28913) and 0.99 (95% CI 0.97-0.99), respectively. Conclusion The present meta-analysis indicated that anaplastic lymphoma kinase plays a significant role in the differential diagnosis of inflammatory myofibroblastic tumours, particularly for inflammatory myofibroblastic tumours of the urinary bladder.
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Affiliation(s)
- Shuiqing Wu
- Department of Urology, The Second Xiangya Hospital of Central South University, Changsha, China
| | - Ran Xu
- Department of Urology, The Second Xiangya Hospital of Central South University, Changsha, China
| | - Qi Wan
- Neural Medical Center of the First Hospital in Changsha City, Changsha, China
| | - Xuan Zhu
- Department of Urology, The Second Xiangya Hospital of Central South University, Changsha, China
| | - Lei Zhang
- Department of Urology, The Second Xiangya Hospital of Central South University, Changsha, China
| | - Hongyi Jiang
- Department of Urology, The Second Xiangya Hospital of Central South University, Changsha, China
| | - Xiaokun Zhao
- Department of Urology, The Second Xiangya Hospital of Central South University, Changsha, China
- * E-mail:
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25
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Kovács A, Máthé G, Mattsson J, Stenman G, Kindblom LG. ALK-Positive Inflammatory Myofibroblastic Tumor of the Nipple During Pregnancy-An Unusual Presentation of a Rare Disease. Breast J 2015; 21:297-302. [DOI: 10.1111/tbj.12404] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/23/2022]
Affiliation(s)
- Anikó Kovács
- Department of Pathology; Sahlgrenska University Hospital; Gothenburg Sweden
| | - Gyöngyvér Máthé
- Department of Pathology; Sahlgrenska University Hospital; Gothenburg Sweden
| | - Jan Mattsson
- Department of Surgery; Sahlgrenska University Hospital; Gothenburg Sweden
| | - Göran Stenman
- Department of Pathology; Sahlgrenska Cancer Center; University of Gothenburg; Gothenburg Sweden
| | - Lars-Gunnar Kindblom
- Department of Musculosceletal Pathology; Royal Orthopaedic Hospital NHS Trust Foundation and Division of Cancer Studies; Medical School; Birmingham University; Birmingham UK
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26
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Inflammatory myofibroblastic tumor of the breast. Case Rep Surg 2015; 2015:705127. [PMID: 25767734 PMCID: PMC4341847 DOI: 10.1155/2015/705127] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/26/2014] [Accepted: 02/03/2015] [Indexed: 02/08/2023] Open
Abstract
Inflammatory myofibroblastic tumors (IMTs) of the breast represent extremely rare lesions. Due to the scarcity of reports, their natural history, recurrence, and metastatic potential remain poorly defined. We report on a case of a primary breast IMT in a postmenopausal female patient treated successfully with breast conserving surgery and review the literature pertaining to this rare entity.
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27
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Xing P, Li J, Jin F, Wu Y, Zheng X, Chen B, Yao F, Wei X. Metaplastic breast carcinoma development following surgical resection of an inflammatory myofibroblastic tumor in the right breast: A case report. Oncol Lett 2014; 8:1345-1347. [PMID: 25120721 PMCID: PMC4114583 DOI: 10.3892/ol.2014.2261] [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: 11/18/2013] [Accepted: 05/29/2014] [Indexed: 11/06/2022] Open
Abstract
Inflammatory myofibroblastic tumors (IMTs) are uncommon, mesenchymal lesions, and malignant transformation is extremely rare. The current study presents the case of a 56-year-old female with a rapidly growing mass in the right breast, which was diagnosed as IMT. Immunohistochemically, the mass was positive for smooth muscle actin (SMA) and Ki-67 (positive staining in 30% of the cells), and negative for S-100, cluster of differentiation (CD)34, p63 and cytokeratin. Malignant transformation to metaplastic carcinoma of the spindle-cell type was observed following surgical resection. This metaplastic carcinoma demonstrated positive immunoreactivity for cytokeratin, vimentin, CD34, p63 and Ki-67 (>30%), and was negative for cytokeratin 7, SMA, desmin and S-100. The patient underwent total mastectomy of the right breast, followed by palliative chemotherapy with capecitabine; however, the patient succumbed to the disease after 12 weeks. The unusual case presented in the current study emphasizes the importance of pre-operative examinations to determine the benign or malignant nature of IMTs, which aids in the choice of appropriate surgical procedures.
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Affiliation(s)
- Peng Xing
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
| | - Jiguang Li
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
| | - Feng Jin
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
| | - Yunfei Wu
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
| | - Xinyu Zheng
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
| | - Bo Chen
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
| | - Fan Yao
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
| | - Xiaolin Wei
- Department of Breast Surgery, The First Affiliated Hospital of China Medical University, Shenyang, Liaoning 110001, P.R. China
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An Unusual Case of Systemic Inflammatory Myofibroblastic Tumor with Successful Treatment with ALK-Inhibitor. Case Rep Pathol 2014; 2014:470340. [PMID: 25045570 PMCID: PMC4087275 DOI: 10.1155/2014/470340] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/30/2014] [Accepted: 06/02/2014] [Indexed: 02/07/2023] Open
Abstract
Systemic inflammatory myofibroblastic tumor is an exceedingly rare entity. A 45-year-old Hispanic female presented with a 6-month history of left-sided thigh pain, low back pain, and generalized weakness. PET/CT scan revealed abnormal activity in the liver, adrenal gland, and pancreas. MRI of the abdomen demonstrated two 6-7 cm masses in the liver. MRI of the lumbar spine demonstrated lesions in the L2 to L4 spinous processes, paraspinal muscles, and subcutaneous tissues, as well as an 8 mm enhancing intradural lesion at T11, all thought to be metastatic disease. A biopsy of the liver showed portal tract expansion by a spindle cell proliferation rich in inflammation. Tumor cells showed immunoreactivity for smooth muscle actin and anaplastic lymphoma kinase 1 (ALK1). Tissue from the L5 vertebra showed a process histologically identical to that seen in the liver. FISH analysis of these lesions demonstrated an ALK (2p23) gene rearrangement. The patient was successfully treated with an ALK-inhibitor, Crizotinib, and is now in complete remission. We present the first reported case, to our knowledge, of inflammatory myofibroblastic tumor with systemic manifestations and ALK translocation. This case is a prime example of how personalized medicine has vastly improved patient care through the use of molecular-targeted therapy.
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Zhou R, Xiang J, Chen Z, Li Z, Hong J. Fever of unknown origin as a presentation of colonic inflammatory myofibroblastic tumor in a 36-year-old female: A case report. Oncol Lett 2014; 7:1566-1568. [PMID: 24765177 PMCID: PMC3997664 DOI: 10.3892/ol.2014.1942] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/14/2013] [Accepted: 01/23/2014] [Indexed: 01/21/2023] Open
Abstract
Inflammatory myofibroblastic tumor is a rare type of lesion that mimics malignancy and has various clinical manifestations. The current study presents a 36-year-old female with a colonic mass, which closely resembled a stromal tumor during imaging. The patient experienced intermittent fever and slight abdominal pain for one month. The fever remained at ≤38.5°C until the day of surgery. The patient underwent a right hemicolectomy and the preoperative fever disappeared and did not recur until the patient was discharged.
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Affiliation(s)
- Ru Zhou
- Department of General Surgery, Huashan Hospital, Fudan University, Shanghai 200040, P.R. China
| | - Jianbin Xiang
- Department of General Surgery, Huashan Hospital, Fudan University, Shanghai 200040, P.R. China
| | - Zongyou Chen
- Department of General Surgery, Huashan Hospital, Fudan University, Shanghai 200040, P.R. China
| | - Zhenyang Li
- Department of General Surgery, Huashan Hospital, Fudan University, Shanghai 200040, P.R. China
| | - Jun Hong
- Department of General Surgery, Huashan Hospital, Fudan University, Shanghai 200040, P.R. China
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