A case of recurrent histiocytic sarcoma with MAP2K1 pathogenic variant treated with the MEK inhibitor trametinib

Tadashi Kumamoto; Yuki Aoki; Tomoko Sonoda; Miho Yamanishi; Ayumu Arakawa; Masanaka Sugiyama; Nami Shirakawa; Sae Ishimaru; Yoshimasa Saito; Akiko Maeshima; Miho Maeda; Chitose Ogawa

doi:10.1007/s12185-018-2553-9

A case of recurrent histiocytic sarcoma with MAP2K1 pathogenic variant treated with the MEK inhibitor trametinib

Tadashi Kumamoto
, Yuki Aoki
, Tomoko Sonoda
, Miho Yamanishi
, Ayumu Arakawa
, Masanaka Sugiyama
, Nami Shirakawa
, Sae Ishimaru
, Yoshimasa Saito
, Akiko Maeshima
, Miho Maeda
, Chitose Ogawa

Research output: Contribution to journal › Article › peer-review

17 Citations (Scopus)

Abstract

Histiocytic sarcoma in advanced clinical stages is typically an aggressive neoplasm, with poor response to conventional chemotherapy. An 18-year-old male with refractory histiocytic sarcoma that had transformed from Rosai–Dorfman disease was admitted to our hospital. A pathogenic variant of MAP2K1 was detected by next-generation sequencing of tumor specimens. Affected regions showed excellent responses to the MEK inhibitor trametinib. It has been reported that RAS/MEK/ERK pathway is activated in many cases of histiocytic sarcoma. MEK inhibition may represent a useful treatment option in histiocytic sarcoma.

Original language	English
Pages (from-to)	228-232
Number of pages	5
Journal	International Journal of Hematology
Volume	109
Issue number	2
DOIs	https://doi.org/10.1007/s12185-018-2553-9
Publication status	Published - 20 Feb 2019
Externally published	Yes

Keywords

Histiocytic sarcoma
MAP2K1
MEK inhibitor
Rosai–Dorfman disease
Trametinib

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10.1007/s12185-018-2553-9

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Kumamoto, T., Aoki, Y., Sonoda, T., Yamanishi, M., Arakawa, A., Sugiyama, M., Shirakawa, N., Ishimaru, S., Saito, Y., Maeshima, A., Maeda, M., & Ogawa, C. (2019). A case of recurrent histiocytic sarcoma with MAP2K1 pathogenic variant treated with the MEK inhibitor trametinib. International Journal of Hematology, 109(2), 228-232. https://doi.org/10.1007/s12185-018-2553-9

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title = "A case of recurrent histiocytic sarcoma with MAP2K1 pathogenic variant treated with the MEK inhibitor trametinib",

abstract = "Histiocytic sarcoma in advanced clinical stages is typically an aggressive neoplasm, with poor response to conventional chemotherapy. An 18-year-old male with refractory histiocytic sarcoma that had transformed from Rosai–Dorfman disease was admitted to our hospital. A pathogenic variant of MAP2K1 was detected by next-generation sequencing of tumor specimens. Affected regions showed excellent responses to the MEK inhibitor trametinib. It has been reported that RAS/MEK/ERK pathway is activated in many cases of histiocytic sarcoma. MEK inhibition may represent a useful treatment option in histiocytic sarcoma.",

keywords = "Histiocytic sarcoma, MAP2K1, MEK inhibitor, Rosai–Dorfman disease, Trametinib",

author = "Tadashi Kumamoto and Yuki Aoki and Tomoko Sonoda and Miho Yamanishi and Ayumu Arakawa and Masanaka Sugiyama and Nami Shirakawa and Sae Ishimaru and Yoshimasa Saito and Akiko Maeshima and Miho Maeda and Chitose Ogawa",

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doi = "10.1007/s12185-018-2553-9",

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Kumamoto, T, Aoki, Y, Sonoda, T, Yamanishi, M, Arakawa, A, Sugiyama, M, Shirakawa, N, Ishimaru, S, Saito, Y, Maeshima, A, Maeda, M & Ogawa, C 2019, 'A case of recurrent histiocytic sarcoma with MAP2K1 pathogenic variant treated with the MEK inhibitor trametinib', International Journal of Hematology, vol. 109, no. 2, pp. 228-232. https://doi.org/10.1007/s12185-018-2553-9

TY - JOUR

T1 - A case of recurrent histiocytic sarcoma with MAP2K1 pathogenic variant treated with the MEK inhibitor trametinib

AU - Kumamoto, Tadashi

AU - Aoki, Yuki

AU - Sonoda, Tomoko

AU - Yamanishi, Miho

AU - Arakawa, Ayumu

AU - Sugiyama, Masanaka

AU - Shirakawa, Nami

AU - Ishimaru, Sae

AU - Saito, Yoshimasa

AU - Maeshima, Akiko

AU - Maeda, Miho

AU - Ogawa, Chitose

PY - 2019/2/20

Y1 - 2019/2/20

N2 - Histiocytic sarcoma in advanced clinical stages is typically an aggressive neoplasm, with poor response to conventional chemotherapy. An 18-year-old male with refractory histiocytic sarcoma that had transformed from Rosai–Dorfman disease was admitted to our hospital. A pathogenic variant of MAP2K1 was detected by next-generation sequencing of tumor specimens. Affected regions showed excellent responses to the MEK inhibitor trametinib. It has been reported that RAS/MEK/ERK pathway is activated in many cases of histiocytic sarcoma. MEK inhibition may represent a useful treatment option in histiocytic sarcoma.

AB - Histiocytic sarcoma in advanced clinical stages is typically an aggressive neoplasm, with poor response to conventional chemotherapy. An 18-year-old male with refractory histiocytic sarcoma that had transformed from Rosai–Dorfman disease was admitted to our hospital. A pathogenic variant of MAP2K1 was detected by next-generation sequencing of tumor specimens. Affected regions showed excellent responses to the MEK inhibitor trametinib. It has been reported that RAS/MEK/ERK pathway is activated in many cases of histiocytic sarcoma. MEK inhibition may represent a useful treatment option in histiocytic sarcoma.

KW - Histiocytic sarcoma

KW - MAP2K1

KW - MEK inhibitor

KW - Rosai–Dorfman disease

KW - Trametinib

UR - https://www.scopus.com/pages/publications/85055680091

U2 - 10.1007/s12185-018-2553-9

DO - 10.1007/s12185-018-2553-9

M3 - Article

C2 - 30361829

AN - SCOPUS:85055680091

SN - 0925-5710

VL - 109

SP - 228

EP - 232

JO - International Journal of Hematology

JF - International Journal of Hematology

IS - 2

ER -

A case of recurrent histiocytic sarcoma with MAP2K1 pathogenic variant treated with the MEK inhibitor trametinib

Abstract

Keywords

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