ETV6-FLT3-positive myeloid/lymphoid neoplasm with eosinophilia presenting in an infant: an entity distinct from JMML.


Journal

Blood advances
ISSN: 2473-9537
Titre abrégé: Blood Adv
Pays: United States
ID NLM: 101698425

Informations de publication

Date de publication:
13 04 2021
Historique:
received: 04 11 2020
accepted: 16 02 2021
entrez: 1 4 2021
pubmed: 2 4 2021
medline: 1 6 2021
Statut: ppublish

Résumé

Myeloid/lymphoid neoplasm with eosinophilia (MLN-Eo) is a World Health Organization (WHO) established category of hematologic malignancies primarily arising in adults. We discuss an 8-month-old infant who presented with clinical features similar to those of juvenile myelomonocytic leukemia (JMML) but who was diagnosed with MLN-Eo driven by an ETV6-FLT3 fusion. Results of patient-derived leukemia ex vivo studies demonstrated increased sensitivity to type I FLT3 inhibitors as compared with type II inhibitors. Treatment with the type I inhibitor gilteritinib resulted in complete immunophenotypic and cytogenetic remission. This patient subsequently underwent a hematopoietic stem cell transplant and remains in complete remission 1 year later. This is the youngest patient reported with an ETV6-FLT3 fusion and adds to the mounting reports of FLT3-rearranged MLN-Eo, supporting its addition to the WHO classification. Furthermore, this case highlights the clinical utility of ex vivo drug testing of targeted therapies.

Identifiants

pubmed: 33792628
pii: S2473-9529(21)00233-0
doi: 10.1182/bloodadvances.2020003699
pmc: PMC8045514
doi:

Substances chimiques

FLT3 protein, human EC 2.7.10.1
fms-Like Tyrosine Kinase 3 EC 2.7.10.1

Types de publication

Journal Article Research Support, N.I.H., Extramural

Langues

eng

Sous-ensembles de citation

IM

Pagination

1899-1902

Subventions

Organisme : NCI NIH HHS
ID : P30 CA008748
Pays : United States

Informations de copyright

© 2021 by The American Society of Hematology.

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Auteurs

Barbara Spitzer (B)

MSK Kids, Memorial Sloan Kettering Cancer Center, New York, NY.
Department of Pediatrics, Weill Cornell Medicine, New York, NY.

Filemon S Dela Cruz (FS)

MSK Kids, Memorial Sloan Kettering Cancer Center, New York, NY.

Glorymar D Ibanez Sanchez (GD)

Sloan Kettering Institute.

Yanming Zhang (Y)

Hematopathology Service, Department of Pathology, and.

Wenbin Xiao (W)

Hematopathology Service, Department of Pathology, and.

Ryma Benayed (R)

Hematopathology Service, Department of Pathology, and.

Alina Markova (A)

Dermatology Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY; and.
Department of Dermatology, Weill Cornell Medicine, New York, NY.

M Irene Rodriguez-Sanchez (MI)

MSK Kids, Memorial Sloan Kettering Cancer Center, New York, NY.

Nancy Bouvier (N)

MSK Kids, Memorial Sloan Kettering Cancer Center, New York, NY.

Mikhail Roshal (M)

Hematopathology Service, Department of Pathology, and.

Andrew L Kung (AL)

MSK Kids, Memorial Sloan Kettering Cancer Center, New York, NY.

Neerav Shukla (N)

MSK Kids, Memorial Sloan Kettering Cancer Center, New York, NY.

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Classifications MeSH