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Intraosseous inflammatory myofibroblastic tumor of the mandible with a novel ATIC-ALK fusion mutation: a case report.
Diagn Pathol 2016; 11(1):132DP

Abstract

BACKGROUND

Inflammatory myofibroblastic tumor (IMT) is a rare low-grade malignant neoplasm with a predilection for children and young adults, and typically arises in the lung, abdominopelvic region, and retroperitoneum. IMTs in the maxillofacial region are extreme rare. Approximately 50% of IMT harbor rearrangements of the anaplastic lymphoma kinase (ALK) gene at 2p23 with various fusion partners.

CASE PRESENTATION

We herein report a case of intraosseous IMT of the mandible with a novel ATIC-ALK fusion. Tooth 43 did not erupt after the loss of tooth 83 in an 11-year-old girl with no previous history of trauma. Panoramic tomography showed a unilocular radiolucent lesion in the right anterior mandible resorbing the root of tooth 42 and the medial side of the root of tooth 44. Computed tomography revealed a well- circumscribed 3-cm osteolytic lesion of the right anterior mandible eroding the buccal cortical plate. The entire lesion was curetted out. A histopathological examination revealed the proliferation of plump spindle cells with a storiform architecture and lymphocytes scattered around spindle cells. The spindle cells showed diffuse cytoplasmic staining for ALK by immunohistochemistry. A fluorescence in situ hybridization analysis revealed the translocation of a part of the ALK gene locus at chromosome 2p23. A rapid amplification of cDNA ends analysis confirmed the rearrangement of ALK and identified ATIC as a partner of this ALK fusion mutant.

CONCLUSION

To the best of our knowledge, this is the first case of intraosseous IMT of the mandible with a novel ATIC-ALK fusion. We also herein reviewed similar tumors reported in the literature.

Authors+Show Affiliations

Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan. ytateish@yokohama-cu.ac.jp.Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan.Department of Pathology, Japanese Red-Cross Musashino Hospital, Tokyo, Japan.Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan.Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan.Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan.Department of Oral and Maxillofacial Surgery, Yokohama City University Graduate School of Medicine, Yokohama, Japan.Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

27846861

Citation

Tateishi, Yoko, et al. "Intraosseous Inflammatory Myofibroblastic Tumor of the Mandible With a Novel ATIC-ALK Fusion Mutation: a Case Report." Diagnostic Pathology, vol. 11, no. 1, 2016, p. 132.
Tateishi Y, Okudela K, Kawai S, et al. Intraosseous inflammatory myofibroblastic tumor of the mandible with a novel ATIC-ALK fusion mutation: a case report. Diagn Pathol. 2016;11(1):132.
Tateishi, Y., Okudela, K., Kawai, S., Suzuki, T., Umeda, S., Matsumura, M., ... Ohashi, K. (2016). Intraosseous inflammatory myofibroblastic tumor of the mandible with a novel ATIC-ALK fusion mutation: a case report. Diagnostic Pathology, 11(1), p. 132.
Tateishi Y, et al. Intraosseous Inflammatory Myofibroblastic Tumor of the Mandible With a Novel ATIC-ALK Fusion Mutation: a Case Report. Diagn Pathol. 2016 Nov 15;11(1):132. PubMed PMID: 27846861.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Intraosseous inflammatory myofibroblastic tumor of the mandible with a novel ATIC-ALK fusion mutation: a case report. AU - Tateishi,Yoko, AU - Okudela,Koji, AU - Kawai,Shigeo, AU - Suzuki,Takehisa, AU - Umeda,Shigeaki, AU - Matsumura,Mai, AU - Kioi,Mitomu, AU - Ohashi,Kenichi, Y1 - 2016/11/15/ PY - 2016/02/12/received PY - 2016/11/09/accepted PY - 2016/11/17/entrez PY - 2016/11/17/pubmed PY - 2017/2/18/medline KW - 5′ rapid amplification of cDNA ends KW - ATIC-ALK fusion KW - Anaplastic lymphoma kinase gene KW - Fluorescence in situ hybridization KW - Inflammatory myofibroblastic tumor KW - Mandible KW - RT-PCR SP - 132 EP - 132 JF - Diagnostic pathology JO - Diagn Pathol VL - 11 IS - 1 N2 - BACKGROUND: Inflammatory myofibroblastic tumor (IMT) is a rare low-grade malignant neoplasm with a predilection for children and young adults, and typically arises in the lung, abdominopelvic region, and retroperitoneum. IMTs in the maxillofacial region are extreme rare. Approximately 50% of IMT harbor rearrangements of the anaplastic lymphoma kinase (ALK) gene at 2p23 with various fusion partners. CASE PRESENTATION: We herein report a case of intraosseous IMT of the mandible with a novel ATIC-ALK fusion. Tooth 43 did not erupt after the loss of tooth 83 in an 11-year-old girl with no previous history of trauma. Panoramic tomography showed a unilocular radiolucent lesion in the right anterior mandible resorbing the root of tooth 42 and the medial side of the root of tooth 44. Computed tomography revealed a well- circumscribed 3-cm osteolytic lesion of the right anterior mandible eroding the buccal cortical plate. The entire lesion was curetted out. A histopathological examination revealed the proliferation of plump spindle cells with a storiform architecture and lymphocytes scattered around spindle cells. The spindle cells showed diffuse cytoplasmic staining for ALK by immunohistochemistry. A fluorescence in situ hybridization analysis revealed the translocation of a part of the ALK gene locus at chromosome 2p23. A rapid amplification of cDNA ends analysis confirmed the rearrangement of ALK and identified ATIC as a partner of this ALK fusion mutant. CONCLUSION: To the best of our knowledge, this is the first case of intraosseous IMT of the mandible with a novel ATIC-ALK fusion. We also herein reviewed similar tumors reported in the literature. SN - 1746-1596 UR - https://www.unboundmedicine.com/medline/citation/27846861/Intraosseous_inflammatory_myofibroblastic_tumor_of_the_mandible_with_a_novel_ATIC_ALK_fusion_mutation:_a_case_report_ L2 - https://diagnosticpathology.biomedcentral.com/articles/10.1186/s13000-016-0586-z DB - PRIME DP - Unbound Medicine ER -