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BMJ Case Reports 2018; doi:10.1136/bcr-2017-221553
  • Rare disease
  • CASE REPORT

Treatment-refractory ALK-positive inflammatory myofibroblastic tumour of the oral cavity

  1. Danielle N Margalit4
  1. 1School of Medicine, Johns Hopkins University, Baltimore, Maryland, USA
  2. 2Department of Pathology, Brigham and Women’s Hospital, Boston, Massachusetts, USA
  3. 3Department of Medical Oncology, Dana-Farber Cancer Institute, Boston, Massachusetts, USA
  4. 4Department of Radiation Oncology, Dana-Farber Cancer Institute, Brigham and Women’s Hospital, Boston, Massachusetts, USA
  1. Correspondence to Dr Danielle N Margalit, dmargalit{at}lroc.harvard.edu
  • Accepted 28 March 2018
  • Published 11 April 2018

Summary

We present a challenging case of a previously healthy 23-year-old man who developed an inflammatory myofibroblastic tumour of the hard palate, harbouring a rearrangement of the anaplastic lymphoma kinase (ALK) locus. Despite surgical intervention, radiotherapy and ALK-inhibition therapy, the tumour recurred locally and metastasised to regional lymph nodes, and the patient passed away roughly 9 months after diagnosis from local progression. The rapid progression of this patient’s disease and its resistance to treatment demonstrate the potentially aggressive clinical course of inflammatory myofibroblastic tumours. ALK-inhibition therapy was unsuccessful in this ALK-positive tumour, highlighting the need for further investigation of markers predictive of disease progression and treatment response.

Footnotes

  • Contributors Study conception and design (DNM, DMM, AWL) and drafting (DNM, AWL, DMM, DRD). All authors participated in the analysis/interpretation of the case report, in revising the manuscript and read and approved the final manuscript.

  • Funding This research received no specific grant from any funding agency in the public,commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent Parental/guardian consent obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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