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A novel LRRFIP1-ALK fusion in inflammatory myofibroblastic tumor of hip and response to crizotinib

Investigational New Drugs volume 39, pages 278–282 (2021)Cite this article

Summary

An inflammatory myofibroblastic tumor (IMT) is a rare invasive soft tissue mass with intramuscular penetration that is primarily treated via a surgical procedure. However, with unclear boundaries and a high rate of relapse, there is no standard treatment for recurrence or unresectable tumors. It is noteworthy that approximately half of IMTs harbor genetic rearrangements of the anaplastic lymphoma kinase (ALK). ALK inhibitors have been used successfully in the treatment of IMTs with a variety of ALK fusions. Here, we present a case of a 15-year-old patient with IMT around the hip. Next-generation sequencing (NGS) revealed an LRRFIP1-ALK fusion, which has not yet been reported in the literature. Crizotinib, an ALK inhibitor, was effective in the treatment of this patient, indicating that ALK inhibitors may be effective for IMT with LRRFIP1-ALK fusions. This report expands the list of gene fusions in IMTs and highlights a new target for treatment.

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Acknowledgements

The authors thank Fajun Yang MD and Lin Hao MD for their contributions to the data collection of this research.

Funding

This research was supported by National Natural Science Foundation of China (51973021), Beijing JST Research Funding (ZR-201902) and CSCO-Youth Innovation Research Funding (Y-young2019-070).

Author information

Author notes

  1. Weifeng Liu and Qianqian Duan contributed equally to this work.

Affiliations

  1. Deptartment of Orthopaedic Oncology Surgery, Beijing Ji Shui Tan Hospital, Peking University, Beijing, 100035, People’s Republic of China

    Weifeng Liu, Yongkun Yang, Zhen Huang & Xiaohui Niu

  2. Fourth Clinical College of Peking University, Beijing, People’s Republic of China

    Weifeng Liu

  3. State Key Laboratory of Translational Medicine and Innovative Drug Development, Jiangsu Simcere Diagnostics Co., Ltd, Nanjing, People’s Republic of China

    Qianqian Duan & Hao Guo

  4. Deptartment of pathology, Beijing Ji Shui Tan Hospital, Peking University, Beijing, People’s Republic of China

    Lihua Gong

Authors
  1. Weifeng Liu
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  2. Qianqian Duan
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  3. Lihua Gong
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  4. Yongkun Yang
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  5. Zhen Huang
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  6. Hao Guo
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  7. Xiaohui Niu
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Contributions

Conception/Design: Weifeng Liu, Qianqian Duan.

Provision of study material or patients: Weifeng Liu, Lihua Gong, Yongkun Yang, Zhen Huang, Xiaohui Niu.

Collection of data: Weifeng Liu, Qianqian Duan, Lihua Gong, Yongkun Yang, Zhen Huang, Hao Guo, Xiaohui Niu.

Data analysis and interpretation: Weifeng Liu, Hao Guo, Qianqian Duan.

Manuscript writing: Weifeng Liu, Qianqian Duan.

Final approval of manuscript: Weifeng Liu, Qianqian Duan, Lihua Gong, Yongkun Yang, Zhen Huang, Hao Guo, Xiaohui Niu.

Corresponding author

Correspondence to Xiaohui Niu.

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Conflict of interest

None of the authors have conflicts of interest to disclose.

Ethics approval

This study was approved by the ethics committee of Beijing Jishuitan Hospital, Peking University (No: 202003-11).

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Informed consent was obtained from the patient for the publication of this case report.

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Cite this article

Liu, W., Duan, Q., Gong, L. et al. A novel LRRFIP1-ALK fusion in inflammatory myofibroblastic tumor of hip and response to crizotinib. Invest New Drugs 39, 278–282 (2021). https://doi.org/10.1007/s10637-020-00984-5

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  • DOI: https://doi.org/10.1007/s10637-020-00984-5

Keywords

  • LRRFIP1-ALK fusion
  • Inflammatory myofibroblastic tumor
  • Novel target
  • Crizotinib