Amplified EPOR/JAK2 Genes Define a Unique Subtype of Acute Erythroid Leukemia.


Journal

Blood cancer discovery
ISSN: 2643-3249
Titre abrégé: Blood Cancer Discov
Pays: United States
ID NLM: 101764786

Informations de publication

Date de publication:
06 09 2022
Historique:
received: 18 10 2021
revised: 05 05 2022
accepted: 12 07 2022
pubmed: 16 7 2022
medline: 9 9 2022
entrez: 15 7 2022
Statut: ppublish

Résumé

Acute erythroid leukemia (AEL) is a unique subtype of acute myeloid leukemia characterized by prominent erythroid proliferation whose molecular basis is poorly understood. To elucidate the underlying mechanism of erythroid proliferation, we analyzed 121 AEL using whole-genome, whole-exome, and/or targeted-capture sequencing, together with transcriptome analysis of 21 AEL samples. Combining publicly available sequencing data, we found a high frequency of gains and amplifications involving EPOR/JAK2 in TP53-mutated cases, particularly those having >80% erythroblasts designated as pure erythroid leukemia (10/13). These cases were frequently accompanied by gains and amplifications of ERG/ETS2 and associated with a very poor prognosis, even compared with other TP53-mutated AEL. In addition to activation of the STAT5 pathway, a common feature across all AEL cases, these AEL cases exhibited enhanced cell proliferation and heme metabolism and often showed high sensitivity to ruxolitinib in vitro and in xenograft models, highlighting a potential role of JAK2 inhibition in therapeutics of AEL. This study reveals the major role of gains, amplifications, and mutations of EPOR and JAK2 in the pathogenesis of pure erythroleukemia. Their frequent response to ruxolitinib in patient-derived xenograft and cell culture models highlights a possible therapeutic role of JAK2 inhibition for erythroleukemia with EPOR/JAK2-involving lesions. This article is highlighted in the In This Issue feature, p. 369.

Identifiants

pubmed: 35839275
pii: 707014
doi: 10.1158/2643-3230.BCD-21-0192
pmc: PMC9894574
doi:

Substances chimiques

Receptors, Erythropoietin 0
JAK2 protein, human EC 2.7.10.2
Janus Kinase 2 EC 2.7.10.2

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

410-427

Informations de copyright

©2022 The Authors; Published by the American Association for Cancer Research.

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Auteurs

June Takeda (J)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Kenichi Yoshida (K)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Masahiro M Nakagawa (MM)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Yasuhito Nannya (Y)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.
Division of Hematopoietic Disease Control, The Institute of Medical Science, The University of Tokyo, Tokyo, Japan.

Akinori Yoda (A)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Ryunosuke Saiki (R)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Yotaro Ochi (Y)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Lanying Zhao (L)

Institute for the Advanced Study of Human Biology (WPI ASHBi), Kyoto University, Kyoto, Japan.

Rurika Okuda (R)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Xingxing Qi (X)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Takuto Mori (T)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Ayana Kon (A)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Kenichi Chiba (K)

Division of Genome Analysis Platform Development, National Cancer Center Research Institute, Tokyo, Japan.

Hiroko Tanaka (H)

M&D Data Science Center, Tokyo Medical and Dental University, Tokyo, Japan.

Yuichi Shiraishi (Y)

Division of Genome Analysis Platform Development, National Cancer Center Research Institute, Tokyo, Japan.

Ming-Chung Kuo (MC)

Division of Hematology-Oncology, Department of Internal Medicine, Chang Gung Memorial Hospital-Linkou, Chang Gung University, Taoyuan, Taiwan.

Cassandra M Kerr (CM)

Department of Translational Hematology and Oncology Research, Taussig Cancer Institute, Cleveland Clinic, Cleveland, Ohio.

Yasunobu Nagata (Y)

Department of Translational Hematology and Oncology Research, Taussig Cancer Institute, Cleveland Clinic, Cleveland, Ohio.

Daisuke Morishita (D)

Chordia Therapeutics Inc., Kanagawa, Japan.

Nobuhiro Hiramoto (N)

Department of Hematology, Kobe City Medical Center General Hospital, Kobe, Japan.

Akira Hangaishi (A)

Department of Hematology, NTT Medical Centre Tokyo, Tokyo, Japan.

Hideyuki Nakazawa (H)

Department of Hematology, Shinshu University Hospital, Matsumoto, Japan.

Ken Ishiyama (K)

Department of Hematology, Kanazawa University, Kanazawa, Japan.

Satoru Miyano (S)

M&D Data Science Center, Tokyo Medical and Dental University, Tokyo, Japan.

Shigeru Chiba (S)

Department of Hematology, Faculty of Medicine, University of Tsukuba, Tsukuba, Japan.

Yasushi Miyazaki (Y)

Department of Hematology, Atomic Bomb Disease Institute, Nagasaki University, Nagasaki, Japan.
Japan Adult Leukemia Study Group, Japan.

Toshiyuki Kitano (T)

Department of Hematology, Kitano Hospital, Tazuke Kofukai Medical Research Institute, Osaka, Japan.

Kensuke Usuki (K)

Department of Hematology, NTT Medical Centre Tokyo, Tokyo, Japan.

Nobuo Sezaki (N)

Department of Hematology, Chugoku Central Hospital, Hiroshima, Japan.

Hisashi Tsurumi (H)

Department of Hematology, Gifu University, Gifu, Japan.

Shuichi Miyawaki (S)

Division of Hematology, Tokyo Metropolitan Ohtsuka Hospital, Tokyo, Japan.

Jaroslaw P Maciejewski (JP)

Department of Translational Hematology and Oncology Research, Taussig Cancer Institute, Cleveland Clinic, Cleveland, Ohio.

Takayuki Ishikawa (T)

Department of Hematology, Kobe City Medical Center General Hospital, Kobe, Japan.

Kazuma Ohyashiki (K)

Department of Hematology, Tokyo Medical University, Tokyo, Japan.

Arnold Ganser (A)

Department of Hematology, Hemostasis, Oncology, and Stem Cell Transplantation, Hannover Medical School, Hannover, Germany.

Michael Heuser (M)

Department of Hematology, Hemostasis, Oncology, and Stem Cell Transplantation, Hannover Medical School, Hannover, Germany.

Felicitas Thol (F)

Department of Hematology, Hemostasis, Oncology, and Stem Cell Transplantation, Hannover Medical School, Hannover, Germany.

Lee-Yung Shih (LY)

Division of Hematology-Oncology, Department of Internal Medicine, Chang Gung Memorial Hospital-Linkou, Chang Gung University, Taoyuan, Taiwan.

Akifumi Takaori-Kondo (A)

Department of Hematology/Oncology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Hideki Makishima (H)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Seishi Ogawa (S)

Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan.
Institute for the Advanced Study of Human Biology (WPI ASHBi), Kyoto University, Kyoto, Japan.
Center for Hematology and Regenerative Medicine, Karolinska Institutet, Stockholm, Sweden.

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