Aggressive Metastatic Inflammatory Myofibroblastic Tumor After Allogeneic Stem Cell Transplant With Fatal Pulmonary Toxicity From Crizotinib.
Inflammatory myofibroblastic tumors (IMTs) are rare tumors with an intermediate spectrum of biological behavior. IMTs are uncommon secondary malignancies after hematopoietic stem cell transplant. The presence of anaplastic lymphoma kinase rearrangements in 50% of IMTs has led to therapeutic trials with crizotinib, although limited experience remains with crizotinib use in children. We describe the first reported case of a highly aggressive and metastatic IMT (secondary malignancy) in an 8-year-old girl following umbilical cord blood transplant. Although tumor response was demonstrated with anaplastic lymphoma kinase inhibition, she later developed fatal pulmonary toxicity from diffuse alveolar damage, a feature felt most likely to be due to crizotinib.
*Pediatric Hematology-Oncology-BMT, CancerCare Manitoba ‡Department of Pathology, University of Manitoba, Winnipeg, MB, Canada †Department of Pediatrics, King Fahd Hospital of the University and Medical College, University of Dammam, Dammam, Saudi Arabia.
Hwazen
H
Shash
Camelia
C
Stefanovici
Susan
S
Phillips
Geoffrey D E
GD
Cuvelier
D000077548:Anaplastic Lymphoma Kinase; D036101:Cord Blood Stem Cell Transplantation / Q000009:adverse effects*; D000077547:Crizotinib; D017809:Fatal Outcome; D005260:Female; D006801:Humans; D007223:Infant; D055370:Lung Injury / Q000139:chemically induced*; D009379:Neoplasms, Muscle Tissue / Q000150:complications / Q000188:drug therapy* / Q000209:etiology*; D016609:Neoplasms, Second Primary; D047428:Protein Kinase Inhibitors; D011720:Pyrazoles / Q000008:administration & dosage / Q000633:toxicity*; D011725:Pyridines / Q000008:administration & dosage / Q000633:toxicity*; D020794:Receptor Protein-Tyrosine Kinases / Q000037:antagonists & inhibitors
D047428:Protein Kinase Inhibitors; D011720:Pyrazoles; D011725:Pyridines; D000077547:Crizotinib; C000626173:ALK protein, human; D000077548:Anaplastic Lymphoma Kinase; D020794:Receptor Protein-Tyrosine Kinases