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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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Chanthong S, Sathitsamitphong L, Natesirinilkul R, Charoenkwan P, Suwansirikul S, Choed-Amphai C. Treatment modalities of ALK-positive relapsed/refractory inflammatory myofibroblastic tumor of the brain and lungs in 7-year-old girl: case-based reviews. Childs Nerv Syst 2023; 39:331-342. [PMID: 36515740 DOI: 10.1007/s00381-022-05789-w] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/14/2022] [Accepted: 12/08/2022] [Indexed: 12/15/2022]
Abstract
PURPOSE Inflammatory myofibroblastic tumor (IMT) belongs to mesenchymal neoplasm of intermediate malignancy in WHO classification. Primary CNS disease or CNS metastases (CNS-IMT) occur in minority. We describe a case of relapsed/refractory IMT of lungs with multiple brain metastases in young child who achieved long-term complete response after alectinib. This systematic review also summarizes treatment modalities and outcome of children and adolescent with CNS-IMT. METHODS PRISMA 2020 guideline was applied to select an article from PubMed, Scopus, and Cochrane databases without time limits. This review focused on children and adolescent 0-24 years of age with CNS-IMT or inflammatory pseudotumor (CNS-IPT). The clinical characteristics and treatment outcome were explored. RESULTS A total of 51 patients in 49 publications were identified. Median age of patients with CNS-IMT/IPT was 15-year-old and 60.8% were male. The most common location of tumor was cerebral cortex (54.9%). Complete resection of CNS-IMT/IPT was performed in 27 cases with 100% complete response and 18.5% recurrence. Nearly half of patients who received partial resection without adjuvant therapy experienced progressive disease, while the contrast group totally achieved partial response. Overall responses in 7 patients treating with ALK inhibitors were 57.1% durable complete response and 42.9% transient partial response. CONCLUSION First-line treatment of CNS-IMT/IPT is complete resection. Patients who received partial tumor removal might have benefit from adjuvant therapy. ALK inhibitors reveal a promising result in unresectable CNS-IMT/IPT. Our case has shown a success in treating relapsed and refractory CNS-IMT as well as the primary site using 2nd-generation ALK inhibitor.
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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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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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Grabenstetter A, D’Alfonso TM. Inflammatory, Reactive, and Infectious Conditions of the Breast. A COMPREHENSIVE GUIDE TO CORE NEEDLE BIOPSIES OF THE BREAST 2022:131-164. [DOI: 10.1007/978-3-031-05532-4_4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
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Wei L, Jiang G, Bai L, Li T, Ma X, Jiang L, Wang J, Li S. Inflammatory Myofibroblastoma of the Breast: A Case Report. Front Oncol 2021; 11:646336. [PMID: 34141609 PMCID: PMC8204013 DOI: 10.3389/fonc.2021.646336] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/26/2020] [Accepted: 04/19/2021] [Indexed: 12/31/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare tumor with low-grade malignant risk mainly occurring in soft tissues and lungs, and it is extremely rare in the breast. Meanwhile, imaging findings of the tumor often present with non-specific features that lead to misdiagnosis and delayed treatment. Here, we report a case of inflammatory myofibroblastic tumor in the breast with the imaging findings of mammography, magnetic resonance imaging (MRI), and pathologic findings to improve the understanding of the disease. The patient was treated by surgical operation, and was followed up for 44 months, no local recurrence and distant metastasis.
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Affiliation(s)
- Ling Wei
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
| | - Guoyuan Jiang
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
| | - Lala Bai
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
| | - Tingchao Li
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
| | - Xuejin Ma
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
| | - Lin Jiang
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
| | - Jie Wang
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
| | - Shiguang Li
- Department of Radiology, The Third Affiliated Hospital of Zunyi Medical University, The First People's Hospital of Zunyi, Zunyi, China
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Xu L, Ge R, Gao S. Imaging features and radiologic-pathologic correlations of inflammatory pseudotumor-like follicular dendritic cell sarcoma. BMC Med Imaging 2021; 21:52. [PMID: 33731032 PMCID: PMC7972340 DOI: 10.1186/s12880-021-00584-6] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/15/2020] [Accepted: 03/10/2021] [Indexed: 12/23/2022] Open
Abstract
Background Inflammatory pseudotumor-like follicular dendritic cell sarcoma (IPT-like FDCS) is a rare tumor. This study aimed to reveal the radiological characteristics of IPT-like FDCS by radiologic-pathologic correlation. Results We analyzed two cases of IPT-like FDCS in the liver, nine in the spleen, and two in both the liver and spleen concomitantly. IPT-like FDCS presented as well-defined iso- or hypodense masses on unenhanced computed tomography (CT) images in both the liver and spleen. Hyperintensities on T1-weighted images and hypointensities on T2-weighted images with hypointense rings were characteristic features in splenic cases. “Halo signs” were observed in two out of three liver tumors. Hepatic lesions showed significant enhancement, whereas splenic lesions showed only mild enhancement. Delayed annular enhancement was observed in both liver and spleen cases. On ultrasonographic examination, IPT-like FDCS presented as hypoechoic lesions with enhancement similar to that observed on CT. Hyaline fibrous pseudocapsules, which correlated with the hypointensities on T2-weighted images, were microscopically observed at the tumor edge. IPT-like FDCS was characterized by an abundance of small blood vessels and capillaries. Capillaries were also found in the fibrous capsule of some IPT-like FDCSs, which may explain the delayed annular enhancement. Conclusions The manifestations of IPT-like FDCS in the liver and spleen showed differences that warrant them to be approached differently during diagnosis. Characteristic radiological findings of IPT-like FDCS included different enhancement patterns between liver and spleen tumors and rim-like hypointensities on T2-weighted images, as well as annular enhancement on CT and magnetic resonance images. These imaging features correlated with tumor pathology.
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Affiliation(s)
- Lingbin Xu
- Department of Radiology, Ningbo Medical Center Lihuili Hospital, Ningbo University, 1111 Jiangnan Road, Ningbo, Zhejiang, 315000, People's Republic of China
| | - Rong Ge
- Department of Diagnosis, Ningbo Diagnostic Pathology Center, Ningbo, Zhejiang, 315021, People's Republic of China
| | - Shanshan Gao
- Department of Ultrasound, Hwa Mei Hospital, University of Chinese Academy of Sciences (Ningbo No. 2 Hospital), 41 Northwest Street, Ningbo, Zhejiang, 315010, People's Republic of China.
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Doroudinia A, Kaghazchi F, Mehrian P, Dorudinia A. Recurrent inflammatory myofibroblastic tumour of the lung: FDG PET/CT scan findings. BMJ Case Rep 2018; 2018:bcr-2018-224373. [PMID: 30007906 DOI: 10.1136/bcr-2018-224373] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/17/2022] Open
Abstract
Inflammatory myofibroblastic tumour (IMT) is a rare neoplasm, occurring most often in children and young adults. IMTs have intermediate biological behaviour with the chance of local invasion, recurrence and even distant metastasis. Wide range of clinical presentations makes the precise diagnosis of IMT more challenging. The best method for definitive diagnosis is tissue biopsy and newer imaging modalities including fleurodeoxyglucose (FDG) positron emission tomography (PET)/CT are useful tools in detection of disease recurrence or distant metastasis. Complete surgical resection is the best-known treatment for this tumour. Here we are presenting an IMT case in a 12-year-old girl in which her recurrent pulmonary IMT was diagnosed based on FDG PET/CT findings and referred for further salvage treatment. Overall imaging modalities are not specific, but PET/CT scan can be useful tool for evaluation of IMT regarding initial staging and restaging to assess treatment response and recurrence.
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Affiliation(s)
- Abtin Doroudinia
- Chronic Respiratory Diseases Research Center, National Research Institute of Tuberculosis and Lung Diseases (NRITLD), Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Fatemeh Kaghazchi
- Chronic Respiratory Diseases Research Center, National Research Institute of Tuberculosis and Lung Diseases (NRITLD), Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Payam Mehrian
- Department of Radiology, Telemedicine Research Center (TRC), National Research Institute of Tuberculosis and Lung Diseases (NRITLD), Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Atosa Dorudinia
- Department of Pathology, Tracheal Diseases Research Center (TDRC), National Research Institute of Tuberculosis and Lung Diseases (NRITLD), Masih Daneshvari Hospital. Shahid Beheshti University of Medical Sciences, Tehran, Iran
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