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

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...

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Bibliographic Details
Published inBMJ case reports Vol. 2018; p. bcr-2017-221553
Main Authors LaVigne, Anna W, Meredith, David M, D’Adamo, David R, Margalit, Danielle N
Format Journal Article
LanguageEnglish
Published England BMJ Publishing Group LTD 11.04.2018
BMJ Publishing Group
SeriesCase Report
Subjects
Anaplastic Lymphoma Kinase
Biopsy
Cancer therapies
Case reports
Chromosomes
Crizotinib
Fatal Outcome
Granuloma, Plasma Cell - diagnostic imaging
Granuloma, Plasma Cell - pathology
Granuloma, Plasma Cell - therapy
Head & neck cancer
Humans
Larynx
Lymph Nodes - pathology
Lymphoma
Male
Mouth Diseases - diagnostic imaging
Mouth Diseases - enzymology
Mouth Diseases - pathology
Mouth Diseases - therapy
Palate, Hard
Pathology
Protein Kinase Inhibitors - therapeutic use
Pyrazoles - therapeutic use
Pyridines - therapeutic use
Radiation therapy
Rare Disease
Receptor Protein-Tyrosine Kinases - antagonists & inhibitors
Recurrence
Tomography, X-Ray Computed
Tumors
Young Adult
radiotherapy
genetics
head and neck cancer
cancer intervention
oral and maxillofacial surgery
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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.
Bibliography:ObjectType-Case Study-2
SourceType-Scholarly Journals-1
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ISSN:1757-790X
1757-790X
DOI:10.1136/bcr-2017-221553