Abstract
Inflammatory myofibroblastic tumor (IMT) of the larynx is an unusual lesion, particularly in the pediatric age group. Laryngeal IMTs in children follow a benign clinical course with reports of only rare recurrences and no metastases. Although anaplastic lymphoma kinase (ALK) has been associated with IMTs, there is only one pediatric laryngeal IMT reported to be ALK-positive with immunohistochemical staining. Here, we present a case of a 10-year-old boy with a laryngeal IMT that recurred four times and was misdiagnosed as recurrent respiratory papillomatosis after the initial three operations. ALK positivity was demonstrated by both immunohistochemical staining and fluorescence in situ hybridization. To the best of our knowledge, this case report is the first to describe a laryngeal IMT that recurred multiple times and was confirmed to be ALK-positive at the molecular level.
Highlights
Inflammatory myofibroblastic tumor (IMT) is an uncommon lesion that usually involves the lungs and mostly affects young adults [1,2]
Anaplastic lymphoma kinase (ALK) gene translocation can be seen in about 50% of IMTs in young patients [2,12], there is only one case of childhood laryngeal IMT that reported anaplastic lymphoma kinase (ALK)-1 positivity by immunohistochemical staining [4]
ALK positivity was demonstrated by both immunohistochemical staining and fluorescence in situ hybridization (FISH)
Summary
Inflammatory myofibroblastic tumor (IMT) is an uncommon lesion that usually involves the lungs and mostly affects young adults [1,2]. The laryngeal presentation is extremely rare; about 30 cases have been described in the literature in English and only eight of these involved children [4,5,6,7,8,9,10,11] Among these eight IMTs, there is only one case of recurrence after resection [7], demonstrating the benign clinical behavior of this tumor. We present a case of a laryngeal IMT in a 10-yearold boy It recurred four times and was misdiagnosed as recurrent respiratory papillomatosis (RRP) after the initial three operations. The lesions displayed an exophytic, polypoid, and papillary appearance under low power (Figure 2A) with a hyperplastic squamous epithelial lining In this squamous lining, there appeared to be koilocyte-like cells. On the basis of the histopathological and immunohistochemical features and the result of the FISH test, a diagnosis of IMT was rendered
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