{"title":"皮肤及粘膜部位炎性肌成纤维细胞瘤:3例临床病理及分子分析,并着重于鉴别诊断。","authors":"Danting Xiong, Xiaona Yin, Yongli Gan, Wenjuan Gan, Xiao Cheng, Ming Zhao","doi":"10.1016/j.anndiagpath.2025.152569","DOIUrl":null,"url":null,"abstract":"<p><p>This study presents three molecularly confirmed cases of ALK-rearranged inflammatory myofibroblastic tumors (IMTs) occurring in superficial locations, demonstrating their rarity, clinicopathologic heterogeneity and diagnostic complexity. The series comprised tumors involving oropharyngeal mucosa, dermal/subcutaneous tissue of the forearm, and tongue mucosa. Histopathological evaluation revealed characteristic proliferations of spindle-to-ovoid cells arranged in fascicular patterns within variably collagenous to myxoid stroma, accompanied by chronic inflammatory infiltrates. Notable morphologic variations included: (1) rhabdomyoblastic differentiation evidenced by rhabdoid morphology and desmin, MyoD1 and myogenin co-expression in one case, histologically overlapping with inflammatory rhabdomyoblastic tumor; and (2) histiocytoid morphology featuring microvesicular cytoplasm in another case, resembling non-neural granular cell tumor. All cases exhibited strong diffuse cytoplasmic ALK immunoreactivity. Molecular profiling identified DCTN1(exon26)::ALK(exon20) fusions in two cases and TIMP3(exon1)::ALK(exon19) fusion in the lingual lesion, the latter corroborating established associations between TIMP3::ALK fusions and head/neck mucosal sites. With follow-up periods of 4-30 months post complete resection, all patients remained disease-free. These findings expand the recognized morphologic spectrum of cutaneous and superficial mucosal ALK-rearranged IMTs while underscoring the indispensable role of integrated histopathologic and molecular pathologic evaluation in differentiating these neoplasms from their histologic mimics, such as inflammatory rhabdomyoblastic tumor, non-neural granular cell tumor, and epithelioid fibrous histiocytoma/superficial ALK-rearranged myxoid spindle cell neoplasm.</p>","PeriodicalId":50768,"journal":{"name":"Annals of Diagnostic Pathology","volume":"80 ","pages":"152569"},"PeriodicalIF":1.4000,"publicationDate":"2025-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Inflammatory myofibroblastic tumors of the skin and mucosal sites: A clinicopathological and molecular analysis of 3 cases with emphasis on differential diagnosis.\",\"authors\":\"Danting Xiong, Xiaona Yin, Yongli Gan, Wenjuan Gan, Xiao Cheng, Ming Zhao\",\"doi\":\"10.1016/j.anndiagpath.2025.152569\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>This study presents three molecularly confirmed cases of ALK-rearranged inflammatory myofibroblastic tumors (IMTs) occurring in superficial locations, demonstrating their rarity, clinicopathologic heterogeneity and diagnostic complexity. The series comprised tumors involving oropharyngeal mucosa, dermal/subcutaneous tissue of the forearm, and tongue mucosa. Histopathological evaluation revealed characteristic proliferations of spindle-to-ovoid cells arranged in fascicular patterns within variably collagenous to myxoid stroma, accompanied by chronic inflammatory infiltrates. Notable morphologic variations included: (1) rhabdomyoblastic differentiation evidenced by rhabdoid morphology and desmin, MyoD1 and myogenin co-expression in one case, histologically overlapping with inflammatory rhabdomyoblastic tumor; and (2) histiocytoid morphology featuring microvesicular cytoplasm in another case, resembling non-neural granular cell tumor. All cases exhibited strong diffuse cytoplasmic ALK immunoreactivity. Molecular profiling identified DCTN1(exon26)::ALK(exon20) fusions in two cases and TIMP3(exon1)::ALK(exon19) fusion in the lingual lesion, the latter corroborating established associations between TIMP3::ALK fusions and head/neck mucosal sites. With follow-up periods of 4-30 months post complete resection, all patients remained disease-free. These findings expand the recognized morphologic spectrum of cutaneous and superficial mucosal ALK-rearranged IMTs while underscoring the indispensable role of integrated histopathologic and molecular pathologic evaluation in differentiating these neoplasms from their histologic mimics, such as inflammatory rhabdomyoblastic tumor, non-neural granular cell tumor, and epithelioid fibrous histiocytoma/superficial ALK-rearranged myxoid spindle cell neoplasm.</p>\",\"PeriodicalId\":50768,\"journal\":{\"name\":\"Annals of Diagnostic Pathology\",\"volume\":\"80 \",\"pages\":\"152569\"},\"PeriodicalIF\":1.4000,\"publicationDate\":\"2025-10-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Annals of Diagnostic Pathology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1016/j.anndiagpath.2025.152569\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"PATHOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Annals of Diagnostic Pathology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1016/j.anndiagpath.2025.152569","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"PATHOLOGY","Score":null,"Total":0}
Inflammatory myofibroblastic tumors of the skin and mucosal sites: A clinicopathological and molecular analysis of 3 cases with emphasis on differential diagnosis.
This study presents three molecularly confirmed cases of ALK-rearranged inflammatory myofibroblastic tumors (IMTs) occurring in superficial locations, demonstrating their rarity, clinicopathologic heterogeneity and diagnostic complexity. The series comprised tumors involving oropharyngeal mucosa, dermal/subcutaneous tissue of the forearm, and tongue mucosa. Histopathological evaluation revealed characteristic proliferations of spindle-to-ovoid cells arranged in fascicular patterns within variably collagenous to myxoid stroma, accompanied by chronic inflammatory infiltrates. Notable morphologic variations included: (1) rhabdomyoblastic differentiation evidenced by rhabdoid morphology and desmin, MyoD1 and myogenin co-expression in one case, histologically overlapping with inflammatory rhabdomyoblastic tumor; and (2) histiocytoid morphology featuring microvesicular cytoplasm in another case, resembling non-neural granular cell tumor. All cases exhibited strong diffuse cytoplasmic ALK immunoreactivity. Molecular profiling identified DCTN1(exon26)::ALK(exon20) fusions in two cases and TIMP3(exon1)::ALK(exon19) fusion in the lingual lesion, the latter corroborating established associations between TIMP3::ALK fusions and head/neck mucosal sites. With follow-up periods of 4-30 months post complete resection, all patients remained disease-free. These findings expand the recognized morphologic spectrum of cutaneous and superficial mucosal ALK-rearranged IMTs while underscoring the indispensable role of integrated histopathologic and molecular pathologic evaluation in differentiating these neoplasms from their histologic mimics, such as inflammatory rhabdomyoblastic tumor, non-neural granular cell tumor, and epithelioid fibrous histiocytoma/superficial ALK-rearranged myxoid spindle cell neoplasm.
期刊介绍:
A peer-reviewed journal devoted to the publication of articles dealing with traditional morphologic studies using standard diagnostic techniques and stressing clinicopathological correlations and scientific observation of relevance to the daily practice of pathology. Special features include pathologic-radiologic correlations and pathologic-cytologic correlations.