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Investigational New Drugs

, Volume 37, Issue 4, pp 791–795 | Cite as

Durable response to the ALK inhibitor alectinib in inflammatory myofibroblastic tumor of the head and neck with a novel SQSTM1–ALK fusion: a case report

  • Kazunori Honda
  • Shigenori KadowakiEmail author
  • Kyoko Kato
  • Nobuhiro Hanai
  • Yasuhisa Hasegawa
  • Yasushi Yatabe
  • Kei Muro
SHORT REPORT

Summary

An inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal neoplasm that typically develops in the lungs and seldom in the head and neck region. It is often related to the anaplastic lymphoma kinase (ALK) fusion gene. Crizotinib, a first-generation ALK inhibitor, has been shown to have a notable response in patients with ALK-positive IMT. Here, we report the first case of a 46-year-old man with IMT harboring a novel SQSTM1–ALK fusion gene who demonstrated marked response to alectinib. The patient presented a right neck mass (5-cm diameter) that progressively enlarged and expanded to the upper mediastinum. ALK-rearranged IMT was diagnosed after complete tumor resection. Spindle cells displayed diffuse cytoplasmic staining for ALK on immunohistochemistry. A fluorescence in situ hybridization analysis revealed the translocation of a part of the ALK gene locus at chromosome 2p23. FoundationOne CDx™ assay identified an SQSTM1-ALK gene fusion. After a year, right cervical, subclavian, and mediastinal lymph node metastases, considered unresectable, developed. Notably, the patient exhibited a marked response to alectinib treatment and has sustained for 17 months following systemic therapy initiation without significant adverse events. This report highlights the possibility of alectinib being a reasonable option for advanced IMT with the SQSTM1-ALK fusion.

Keywords

Alectinib Inflammatory myofibroblastic tumor Head and neck ALK gene rearrangement SQSTM1-ALK gene fusion 

Notes

Acknowledgements

The authors would like to thank Enago (www.enago.jp) for the English language review.

Compliance with ethical standards

Conflict of interest

All authors have no conflicts of interest to disclose.

Ethical approval

This study does not contain any studies with human participants or animals performed by any of the authors.

Informed consent

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

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© Springer Science+Business Media, LLC, part of Springer Nature 2019

Authors and Affiliations

  1. 1.Department of Clinical OncologyAichi Cancer Center HospitalNagoyaJapan
  2. 2.Department of Head and Neck SurgeryAichi Cancer Center HospitalNagoyaJapan
  3. 3.Department of Pathology and Molecular DiagnosticsAichi Cancer Center HospitalNagoyaJapan

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