Inflammatory Myofibroblastic Tumor of the Lung: Unusual Imaging Findings of Three Cases

Ufuk, Furkan; Herek, Duygu; Karabulut, Nevzat

doi:10.12659/pjr.894902

Cited by 11 publications

(14 citation statements)

References 12 publications

(12 reference statements)

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“…It is composed of myofibroblastic spindle cells admixed with infiltrates of plasma cells, lymphocytes and eosinophils [1]. Most common site is the lung followed by other sites.…”

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confidence: 99%

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Clinicopathological Study of 18 Cases of Inflammatory Myofibroblastic Tumors with Reference to ALK-1 Expression: 5-Year Experience in a Tertiary Care Center

Telugu¹,

Prabhu

Kalappurayil

et al. 2017

J Pathol Transl Med

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BackgroundInflammatory myofibroblastic tumor is a histopathologically distinctive neoplasm of children and young adults. According to World Health Organization (WHO) classification, inflammatory myofibroblastic tumor is an intermediate-grade tumor, with potential for recurrence and rare metastasis. There are no definite histopathologic, molecular, or cytogenetic features to predict malignant transformation, recurrence, or metastasis.MethodsA 5-year retrospective study of histopathologically diagnosed inflammatory myofibroblastic tumors of various anatomic sites was conducted to correlate anaplastic lymphoma kinase-1 (ALK-1) expression with histological atypia, multicentric origin of tumor, recurrence, and metastasis. Clinical details of all the cases were noted from the clinical work station. Immunohistochemical stains for ALK-1 and other antibodies were performed. Statistical analysis was done using Fisher exact test.ResultsA total of 18 cases of inflammatory myofibroblastic tumors were found during the study period, of which 14 were classical. The female-male ratio was 1:1 and the mean age was 23.8 years. Histologically atypical (four cases) and multifocal tumors (three cases, multicentric in origin) were noted. Recurrence was noted in 30% of ALK-1 positive and 37.5% of ALK-1 negative cases, whereas metastasis to the lung, liver, and pelvic bone was noted in the ALK-1 positive group only.ConclusionsOverall, ALK-1 protein was expressed in 55.6% of inflammatory myofibroblastic tumors. There was no statistically significant correlation between ALK-1 expression, tumor type, recurrence and metastasis. However, ALK-1 immunohistochemistry is a useful diagnostic aid in the appropriate clinical and histomorphologic context.

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“…It is composed of myofibroblastic spindle cells admixed with infiltrates of plasma cells, lymphocytes and eosinophils [1]. Most common site is the lung followed by other sites.…”

mentioning

confidence: 99%

“…Inflammatory myofibroblastic tumor is a histopathologically distinctive neoplasm that has been described in various anatomic sites of children and young adults. It is composed of myofibroblastic spindle cells admixed with infiltrates of plasma cells, lymphocytes and eosinophils [ 1 ]. Most common site is the lung followed by other sites.…”

mentioning

confidence: 99%

Clinicopathological Study of 18 Cases of Inflammatory Myofibroblastic Tumors with Reference to ALK-1 Expression: 5-Year Experience in a Tertiary Care Center

Telugu¹,

Prabhu

Kalappurayil

et al. 2017

J Pathol Transl Med

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“…IMT is a rare neoplasm which belongs to a subtype of soft tissue sarcoma, with an overall prevalence of approximately 0.04%-0.7% (13,14) It may occur at any age but it is more common in children and adolescents, constituting less than 1% of adult lung tumors. (15,16) Therapeutic options for patients with unresectable and/or advanced IMT are limited in particular for ALK negative cases. (17) Other treatments besides surgery include radiation and chemotherapy.…”

Section: Discussionmentioning

confidence: 99%

ALK-Negative Lung Inflammatory Myofibroblastic Tumor in a Young Adult: a Case Report and Literature Review of Molecular Alterations

Debonis

Bongiovanni

Pieri

et al. 2021

Preprint

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Background: Inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor and is prevalent among children and adolescents. Surgery is the most important therapeutic approach for IMT and complete resection is recommended. Although 50% of IMTs present anaplastic lymphoma kinase (ALK) rearrangements, an efficacy has been shown by the use of Crizotinib . However the genetic landscape of this tumor is not fully understood and the therapeutic options are limited in particular for the remaining percentage of negative ALK tumors. Case presentation: In our case, we detail the clinical history of 18-year-old female patient diagnosed with pulmonary IMT negative for ALK, subjected to surgery and subsequently to follow-up. The initial pathology report oriented for a salivary gland lung cancer. Afterwards due to a second look by another pathologist an ALK negative IMT of the lung was diagnosed. We also perform a literature review based on IMT and other kinase fusions found in addition to ALK such as ROS proto-oncogene 1 (ROS1), rearranged during transfection (RET), neurotrophic receptor tyrosine kinases (NTRKs) and platelet derived growth factor receptor (PDGFR beta). Conclusions: IMT is a very rare disease involving children and adolescents. Little is known about the clinical and molecular characteristics, pathological diagnosis, prognosis and optimal management strategy of IMT. Since there is no "standard of care" therapy for IMT, identifying feasible genomic alterations could redefine the management of patients with negative ALK disease.

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“…Inflammatory myofibroblastic tumour, previously under the umbrella of the inflammatory pseudotumour, is a distinct myofibroblastic neoplasm with infiltrates of plasma cells, lymphocytes and eosinophils. [ 9 ] IMT has an incidence of 0.04% to 1% among all pulmonary neoplasms[ 10 ] and occurs mostly in children and young adults. The underlying aetiology of IMT is not certain.…”

Section: Discussionmentioning

confidence: 99%

Inflammatory myofibroblastic tumours of the thorax: Radiologic and clinicopathological correlation

Irodi

Chacko

Prajapati

et al. 2020

Indian Journal of Radiology and Imaging

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Context and Aims: Inflammatory myofibroblastic tumour (IMT) is a rare mesenchymal neoplasm with intermediate malignant potential. The aim of this study is to describe and compare the clinical presentation, computed tomography (CT) findings and anaplastic lymphoma kinase -1 (ALK-1) expression of IMT of the thorax in children and adults. We also sought to study the tumour behaviour after treatment on the follow-up imaging. Materials and Method: This is a retrospective observational study of 22 histopathologically proven cases of IMT in the thorax. The clinical parameters, CT findings, biopsy results, treatment received and follow-up were recorded. Statistical analysis was performed using Fisher's exact test. Results: IMT of the thorax had diverse imaging appearances, presenting either as large invasive lung masses with or without calcifications or as smaller endobronchial lesions. Children commonly presented with long duration fever ( P = 0.02) and large invasive lung masses ( P = 0.026), whereas adults presented with long duration haemoptysis ( P = 0.001) and endobronchial lesions or smaller lung parenchymal lesions. Calcifications were more common in children ( P = 0.007). ALK-1 was positive in 40% of children and 18.2% of adults ( P = 0.547). Endobronchial lesions showed a trend for ALK-1 negativity. Patients with bronchoscopic excision had local recurrence and patients with surgical wedge resection had metastatic brain lesions as compared to those with lobectomy and pneumonectomy ( P = 0.0152). A patient with unresectable lung mass had malignant transformation to spindle cell sarcoma after 9.5 years. Conclusions: Thoracic IMT presents with some distinct clinical and CT findings in adults and children. The CT findings and management options have implications for prognosis. If resectable, lobectomy is a better option than wedge resection or bronchoscopic excision for preventing local recurrence and metastasis. IMT can undergo malignant transformation.

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Inflammatory Myofibroblastic Tumor of the Lung: Unusual Imaging Findings of Three Cases

Cited by 11 publications

References 12 publications

Clinicopathological Study of 18 Cases of Inflammatory Myofibroblastic Tumors with Reference to ALK-1 Expression: 5-Year Experience in a Tertiary Care Center

Clinicopathological Study of 18 Cases of Inflammatory Myofibroblastic Tumors with Reference to ALK-1 Expression: 5-Year Experience in a Tertiary Care Center

ALK-Negative Lung Inflammatory Myofibroblastic Tumor in a Young Adult: a Case Report and Literature Review of Molecular Alterations

Inflammatory myofibroblastic tumours of the thorax: Radiologic and clinicopathological correlation

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