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Year : 2018  |  Volume : 14  |  Issue : 8  |  Page : 257-259

Uterine metastases originating from a pulmonary inflammatory myofibroblastic tumor

1 Department of Gynecology, The Fourth Hospital of Hebei Medical University, Shijiazhuang, China
2 Department of Pathology, The Second Hospital of Hebei Medical University, Shijiazhuang, China

Date of Web Publication26-Mar-2018

Correspondence Address:
Lei Lou
Department of Pathology, The Second Hospital of Hebei Medical University, Shijiazhuang
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-1482.172129

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 > Abstract 

Pulmonary inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor, and is considered an inflammatory pseudotumor. Here, we report of a 37-year-old woman with severe cough and stridor who was diagnosed with primary IMT of the lungs, with adenocarcinoma in situ based on the pathology in December 2012. A year later, transabdominal ultrasonography demonstrated a solid mass in the uterine wall that was initially diagnosed as a leiomyoma. However, postoperative histological examination and immunohistochemical staining revealed it as a uterine metastasis of the lung IMT. One year thereafter, the patient died of the lung tumor.

Keywords: Inflammatory myofibroblastic tumor, pulmonary, uterine metastases

How to cite this article:
Zhang J, Li Y, Lou L. Uterine metastases originating from a pulmonary inflammatory myofibroblastic tumor. J Can Res Ther 2018;14, Suppl S1:257-9

How to cite this URL:
Zhang J, Li Y, Lou L. Uterine metastases originating from a pulmonary inflammatory myofibroblastic tumor. J Can Res Ther [serial online] 2018 [cited 2021 Dec 4];14:257-9. Available from: https://www.cancerjournal.net/text.asp?2018/14/8/257/172129

 > Introduction Top

Inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor with low malignant potential.[1] It occurs in the soft tissues, including the lungs, most frequently in children and young adults.[2] Histologically, IMT is composed of variable admixture of bland, spindle-shaped myofibroblast-like cells, and has an inflammatory component of lymphocytes, eosinophils, plasma cells, and macrophages. It is mainly characterized by regional infiltrative growth, vascular invasion, and local recurrence, but rarely with metastasis. Extrapulmonary metastasis of IMT is particularly rare.[3] Here, we describe the case of a patient with extrapulmonary metastasis of IMT to the uterus.

 > Case Report Top

A 37-year-old female patient presented with left chest pain, cough, expectoration, and bloody phlegm of 3-months duration. The expectoration sample was pathologically examined, revealing spindle cell proliferation [Figure 1]. Electronic bronchoscopy showed smooth mucous in the left middle lobar bronchus whereas the left inferior lobar bronchus was completely obstructed by myxoid tissues and was not clearly visible. Chest computed tomography (CT) imaging showed a solid mass with central lucency in the posterior segment of the left upper pulmonary lobe, associated with upper lobe atelectasis, which pushed the mediastinum to the left side [Figure 2]. Hilar lymphadenopathy was absent. Results of sputum microscopy, culture, and cytological examination were negative.
Figure 1: Expectoration tissue containing a mixture of spindle cells and infiltrating inflammatory cells containing lymphocytes and plasma cells (H and E, ×100)

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Figure 2: Computed tomography scan showing an irregular mass in the left lung field

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For diagnostic and therapeutic purposes, the patient underwent a left pneumonectomy. Macroscopically, the tumor was 10 cm × 6 cm × 4 cm in size, firm, whitish, and myxoid [Figure 3]a (i)]. The mass consisted of spindle cells, with prominent inflammatory cell infiltration, and myxoid stroma with adenocarcinoma [Figure 3]a (ii)]. Immunohistochemistry indicated that the tumor was positive for anaplastic lymphoma kinase (ALK), smooth muscle actin, and vimentin, but negative for S-100 [Figure 3]b, suggesting that the tumor was an IMT. Moreover, the tumor was also confirmed as a multifocal adenocarcinoma in situ of the lung.
Figure 3: (a) Macroscopic appearance after surgical ablation (i); histological examination showing the presence of adenocarcinoma of lung and myofibroblastic spindle cells associated with inflammatory lymphocytes and plasma cells (H and E, ×40) (ii). (b) Immunohistochemical study showing positivity for anaplastic lymphoma kinase, smooth muscle actin, and vimentin, as well as negativity for S-100 (original magnification, ×100)

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One year later, the patient was admitted to hospital for general fatigue and abdominal pain. Laboratory tests revealed iron-deficiency anemia. Transabdominal ultrasonography revealed a solid mass in the uterine wall; the ovaries were not visualized, and ascites was absent [Figure 4]a. The patient was diagnosed with a leiomyoma and underwent a total simple hysterectomy. During surgery, a 6-cm mass was found attached to the uterine corpus [Figure 4]b. Postoperative histological examination revealed it as an IMT of pulmonary origin. Furthermore, immunohistochemistry indicated that the tumor was positive for ALK [Figure 4]c, smooth muscle actin, and vimentin. Histology and immunohistochemistry thus confirmed the diagnosis of this mass as a uterine metastasis of the pulmonary IMT. The patient's postoperative course was uneventful, and she was discharged from hospital 2 weeks later. One year subsequently, she died of lung cancer.
Figure 4: Transabdominal ultrasonography demonstrated a solid uterine mass. (a) Pathologic findings of resected specimens from the patient. (b) Grossly, the solitary mass was white-gray and rubbery. (c) Immunohistochemical staining showing positive reactivity for anaplastic lymphoma kinase (original magnification, ×100)

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 > Discussion Top

IMT is a rare mass-forming lesion characterized by fibroblastic or myofibroblastic spindle cell proliferation associated with varying degrees of inflammatory cell infiltrations.[4] Although inflammatory pseudotumors are regarded as inflammatory or reactive lesions rather than as neoplasms, they also feature local invasion or recurrence, distant metastases, and cytogenetic clonal changes.[5],[6]

It is exceedingly uncommon for lung IMT with adenocarcinoma in situ to be associated with uterine metastasis, as these rare metastases usually originate from endometrial carcinoma; in addition, extrapulmonary metastasis of lung IMT is extremely rare. The diagnosis of IMTs is difficult because they lack characteristic clinical manifestations and present with extremely diverse symptoms. Most patients are asymptomatic, and the tumor is typically discovered incidentally on chest radiographs.[7],[8] To date, there is no specific physical or laboratory examination that can be used to identify this conditions;[3],[9],[10] although ultrasonography, magnetic resonance imaging, and CT scans are able to provide some evidence, they are not specific enough to differentiate this type of lesion from other neoplasms.[11] Thus, differential diagnosis between IMT and other neoplasms mainly depends on pathological and immunohistochemical findings.[12],[13]

Microscopically, the tumor contains a myofibroblastic, slightly atypical spindle cell component, which is more or less fasciculated in a hyaline stroma and is associated with many inflammatory lymphocytes/plasma cells. IMTs stain positively for vimentin and smooth muscle actin. Immunohistochemical cytoplasmic staining for ALK is detectable in about half the cases.[14] The macroscopic appearance of IMT is that of a slow-growing, nonencapsulated, frequently single, multilobular mass, with a yellow-to-brownish surface. It may demonstrate necrotic − hemorrhagic changes and calcifications.[15] Local recurrence is attributed to incomplete resection of the primary lesion. Metastasis of the tumor to the mediastinum or the brain has been reported even many years after complete resection whereas association with other malignancies has been reported sporadically.[16]

When uterine IMT is identified, it must first be differentiated from leiomyoma. The differential diagnosis also includes nontumor lesions such as abscesses or tuberculous granulomas, as well as benign or malignant tumor. Lymphoma or desmoid fibromatosis, angiomyofibroblastoma, fibrosarcoma, leiomyoma, and malignant fibrous histiocytoma must also be eliminated.[17]

Despite being benign, IMT is considered a low-grade tumor owing to malignant features such as local invasiveness, recurrence, or malignant transformation. The prognosis of IMT after complete surgical resection has generally been considered favorable.[12],[18] However, careful follow-up is necessary, as certain patients with inflammatory pseudotumors of the liver have been reported to have succumbed to the diseases.[19],[20]

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Conflicts of interest

There are no conflicts of interest.

 > References Top

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Coffin CM, Patel A, Perkins S, Elenitoba-Johnson KS, Perlman E, Griffin CA. ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol 2001;14:569-76.  Back to cited text no. 3
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Matsubara O, Tan-Liu NS, Kenney RM, Mark EJ. Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fibrous histiocytoma or to plasma cell granuloma in 32 cases. Hum Pathol 1988;19:807-14.  Back to cited text no. 9
Anthony PP. Inflammatory pseudotumour (plasma cell granuloma) of lung, liver and other organs. Histopathology 1993;23:501-3.  Back to cited text no. 10
Ma ZH, Tian XF, Ma J, Zhao YF. Inflammatory pseudotumor of the spleen: A case report and review of published cases. Oncol Lett 2013;5:1955-57.  Back to cited text no. 11
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Ishida T, Oka T, Nishino T, Tateishi M, Mitsudomi T, Sugimachi K. Inflammatory pseudotumor of the lung in adults: Radiographic and clinicopathological analysis. Ann Thorac Surg 1989;48:90-5.  Back to cited text no. 13
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Gleason BC, Hornick JL. Inflammatory myofibroblastic tumours: Where are we now? J Clin Pathol 2008;61:428-37.  Back to cited text no. 15
Matsubayashi H, Mizoue T, Mizuguchi Y, Shinohara Y, Magami Y, Horibe T, et al. A case of hemangioma accompanied by inflammatory pseudotumor of the spleen. J Clin Gastroenterol 2000;31:258-61.  Back to cited text no. 16
Bakhos R, Wojcik EM, Olson MC. Transthoracic fine-needle aspiration cytology of inflammatory pseudotumor, fibrohistiocytic type: A case report with immunohistochemical studies. Diagn Cytopathol 1998;19:216-20.  Back to cited text no. 17
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Tsutsumi N, Kawanaka H, Yamaguchi S, Sakai M, Momosaki S, Endo K, et al. Huge inflammatory pseudotumor of the spleen with postoperative portal vein thrombosis: Report of a case. Surg Today 2012;42:382-5.  Back to cited text no. 19
Horiuchi R, Uchida T, Kojima T, Shikata T. Inflammatory pseudotumor of the liver. Clinicopathologic study and review of the literature. Cancer 1990;65:1583-90.  Back to cited text no. 20


  [Figure 1], [Figure 2], [Figure 3], [Figure 4]


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