Pulmonary Inflammatory Myofibroblastic Tumor: A Case Report

Abstract

An inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor that occurs predominantly in children and young adults. Etiology remains unclear. But based on the frequent detection of chromosomic alterations, especially near the anaplastic lymphoma kinase (ALK) gene, IMT is now considered to be a true neoplasm. In addition, the possible aggressive behavior, and the ability to metastasize suggest at least an intermediate malignant potential. Surgery remains the treatment of choice, but the use of chemotherapy, nonsteroidal anti-inflammatory drugs, immunotherapy, and targeted therapy are reported. We describe a case of a pulmonary IMT in a 6-year-old boy with an incidental finding of a lesion in the right upper lobe. A video-assisted thoracoscopic right upper lobectomy with lymph node resection was performed. Microscopic examination confirmed the diagnosis of IMT with the nodule showing spindle cells in a background of plasma cells. ALK immunohistochemical expression was negative.

Keywords

inflammatory myofibroblastic tumor - histopathology - anaplastic lymphoma kinase - pediatric surgery

Publikationsverlauf

Eingereicht: 03. Dezember 2023

Angenommen: 17. September 2024

Accepted Manuscript online:
01. Oktober 2024

Artikel online veröffentlicht:
04. November 2024

• References

• 1 Weldon CB, Shamberger RC. Pediatric pulmonary tumors: primary and metastatic. Semin Pediatr Surg 2008; 17 (01) 17-29
• 2 Pettinato G, Manivel JC, De Rosa N, Dehner LP. Inflammatory myofibroblastic tumor (plasma cell granuloma). Clinicopathologic study of 20 cases with immunohistochemical and ultrastructural observations. Am J Clin Pathol 1990; 94 (05) 538-546
• 3 Cerfolio RJ, Allen MS, Nascimento AG. et al. Inflammatory pseudotumors of the lung. Ann Thorac Surg 1999; 67 (04) 933-936
• 4 Dehner LP. The enigmatic inflammatory pseudotumours: the current state of our understanding, or misunderstanding. J Pathol 2000; 192 (03) 277-279
• 5 Griffin CA, Hawkins AL, Dvorak C, Henkle C, Ellingham T, Perlman EJ. Recurrent involvement of 2p23 in inflammatory myofibroblastic tumors. Cancer Res 1999; 59 (12) 2776-2780
• 6 Bhagat P, Bal A, Das A, Singh N, Singh H. Pulmonary inflammatory myofibroblastic tumor and IgG4-related inflammatory pseudotumor: a diagnostic dilemma. Virchows Arch 2013; 463 (06) 743-747
• 7 Fletcher CDM, Unni KK, Mertens F. Pathology and Genetics of Tumours of Soft Tissue and Bone WHO Classification of Tumours. 3rd ed. Lyon:: The International Agency for Research on Cancer;; 2002
• 8 Mir MH, Dar W, Aejaz Aziz S, Mohamad G, Wani B. Clinico-radiological and pathological characteristics of inflammatory myofibroblastic tumors in children: a retrospective study. Indian J Med Paediatr Oncol 2017; 38 (03) 261-265
• 9 Cuch B, Opasała U, Nachulewicz P, Szumiło J. Inflammatory myofibroblastic tumour in a 5-year-old child - a case report and review of the literature. Wideochir Inne Tech Malo Inwazyjne 2014; 9 (04) 658-661
• 10 Camela F, Gallucci M, di Palmo E. et al. Pulmonary inflammatory myofibroblastic tumor in childern: a case report and brief review of literature. Front Pediatr 2018; 6: 35
• 11 Stoll LM, Li QK. Cytology of fine-needle aspiration of inflammatory myofibroblastic tumor. Diagn Cytopathol 2011; 39 (09) 663-672
• 12 Naime S, Bandarkar A, Nino G, Perez G. Pulmonary inflammatory myofibroblastic tumour misdiagnosed as a round pneumonia. BMJ Case Rep 2018; 2018: 1-2
• 13 Borak S, Siegal GP, Reddy V, Jhala N, Jhala D. Metastatic inflammatory myofibroblastic tumor identified by EUS-FNA in mediastinal lymph nodes with ancillary FISH studies for ALK rearrangement. Diagn Cytopathol 2012; 40 (Suppl. 02) E118-E125
• 14 Coffin CM, Hornick JL, Fletcher CDM. Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive cases. Am J Surg Pathol 2007; 31 (04) 509-520
• 15 Tothova Z, Wagner AJ. Anaplastic lymphoma kinase-directed therapy in inflammatory myofibroblastic tumors. Curr Opin Oncol 2012; 24 (04) 409-413
• 16 Wachter F, Al-Ibraheemi A, Trissal MC. et al. Molecular characterization of inflammatory tumors facilitates initiation of effective therapy. Pediatrics 2021; 148 (06) 1-6
• 17 Chavez C, Hoffman MA. Complete remission of ALK-negative plasma cell granuloma (inflammatory myofibroblastic tumor) of the lung induced by celecoxib: a case report and review of the literature. Oncol Lett 2013; 5 (05) 1672-1676
• 18 McDermott M. Inflammatory myofibroblastic tumour. Semin Diagn Pathol 2016; 33: 358-366
• 19 Kinoshita Y, Tajiri T, Ieiri S. et al. A case of an inflammatory myofibroblastic tumor in the lung which expressed TPM3-ALK gene fusion. Pediatr Surg Int 2007; 23 (06) 595-599