Paraneoplastic syndrome of angiomatoid fibrous histiocytoma may be caused by EWSR1-CREB1 fusion-induced excessive interleukin-6 production

Masaharu Akiyama; Masayoshi Yamaoka; Yoko Mikami-Terao; Kentaro Yokoi; Takashi Inoue; Tomomasa Hiramatsu; Shuichi Ashizuka; Jyoji Yoshizawa; Hiroaki Katagi; Masahiro Ikegami; Hiroyuki Ida; Atsuko Nakazawa; Hajime Okita; Kenji Matsumoto

doi:10.1097/MPH.0000000000000390

Paraneoplastic syndrome of angiomatoid fibrous histiocytoma may be caused by EWSR1-CREB1 fusion-induced excessive interleukin-6 production

Masaharu Akiyama, Masayoshi Yamaoka, Yoko Mikami-Terao, Kentaro Yokoi, Takashi Inoue, Tomomasa Hiramatsu, Shuichi Ashizuka, Jyoji Yoshizawa, Hiroaki Katagi, Masahiro Ikegami, Hiroyuki Ida, Atsuko Nakazawa, Hajime Okita, Kenji Matsumoto

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

12 Citations (Scopus)

Abstract

We describe a 7-year-old girl with angiomatoid fibrous histiocytoma (AFH) presenting severe inflammatory symptoms. The cytokine/chemokine profile of serum samples before and after surgery demonstrated that interleukin (IL)-6 had decreased by the greatest percentage. The AFH cells were immunopathologically positive for IL-6 and Tyr705-phosphorylation of signal transducer and activator of transcription 3. The EWSR1-CREB1 fusion gene detected in the tumor leads to continuous activation of CREB1 and IL-6 production, because the promoter region of IL-6 has a CREB binding site. Thus, IL-6 plays pivotal roles in both paraneoplastic syndrome and the oncogenesis of AFH.

Original language	English
Pages (from-to)	554-559
Number of pages	6
Journal	Journal of Pediatric Hematology/Oncology
Volume	37
Issue number	7
DOIs	https://doi.org/10.1097/MPH.0000000000000390
Publication status	Published - 2015 Oct 1
Externally published	Yes

Keywords

Angiomatoid fibrous histiocytoma
EWSR1-CREB1 fusion gene
Interleukin-6
Paraneoplastic syndrome

ASJC Scopus subject areas

Pediatrics, Perinatology, and Child Health
Hematology
Oncology

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10.1097/MPH.0000000000000390

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Akiyama, M., Yamaoka, M., Mikami-Terao, Y., Yokoi, K., Inoue, T., Hiramatsu, T., Ashizuka, S., Yoshizawa, J., Katagi, H., Ikegami, M., Ida, H., Nakazawa, A., Okita, H., & Matsumoto, K. (2015). Paraneoplastic syndrome of angiomatoid fibrous histiocytoma may be caused by EWSR1-CREB1 fusion-induced excessive interleukin-6 production. Journal of Pediatric Hematology/Oncology, 37(7), 554-559. https://doi.org/10.1097/MPH.0000000000000390

Akiyama, M, Yamaoka, M, Mikami-Terao, Y, Yokoi, K, Inoue, T, Hiramatsu, T, Ashizuka, S, Yoshizawa, J, Katagi, H, Ikegami, M, Ida, H, Nakazawa, A, Okita, H & Matsumoto, K 2015, 'Paraneoplastic syndrome of angiomatoid fibrous histiocytoma may be caused by EWSR1-CREB1 fusion-induced excessive interleukin-6 production', Journal of Pediatric Hematology/Oncology, vol. 37, no. 7, pp. 554-559. https://doi.org/10.1097/MPH.0000000000000390

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title = "Paraneoplastic syndrome of angiomatoid fibrous histiocytoma may be caused by EWSR1-CREB1 fusion-induced excessive interleukin-6 production",

abstract = "We describe a 7-year-old girl with angiomatoid fibrous histiocytoma (AFH) presenting severe inflammatory symptoms. The cytokine/chemokine profile of serum samples before and after surgery demonstrated that interleukin (IL)-6 had decreased by the greatest percentage. The AFH cells were immunopathologically positive for IL-6 and Tyr705-phosphorylation of signal transducer and activator of transcription 3. The EWSR1-CREB1 fusion gene detected in the tumor leads to continuous activation of CREB1 and IL-6 production, because the promoter region of IL-6 has a CREB binding site. Thus, IL-6 plays pivotal roles in both paraneoplastic syndrome and the oncogenesis of AFH.",

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author = "Masaharu Akiyama and Masayoshi Yamaoka and Yoko Mikami-Terao and Kentaro Yokoi and Takashi Inoue and Tomomasa Hiramatsu and Shuichi Ashizuka and Jyoji Yoshizawa and Hiroaki Katagi and Masahiro Ikegami and Hiroyuki Ida and Atsuko Nakazawa and Hajime Okita and Kenji Matsumoto",

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doi = "10.1097/MPH.0000000000000390",

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AU - Akiyama, Masaharu

AU - Yamaoka, Masayoshi

AU - Mikami-Terao, Yoko

AU - Yokoi, Kentaro

AU - Inoue, Takashi

AU - Hiramatsu, Tomomasa

AU - Ashizuka, Shuichi

AU - Yoshizawa, Jyoji

AU - Katagi, Hiroaki

AU - Ikegami, Masahiro

AU - Ida, Hiroyuki

AU - Nakazawa, Atsuko

AU - Okita, Hajime

AU - Matsumoto, Kenji

PY - 2015/10/1

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AB - We describe a 7-year-old girl with angiomatoid fibrous histiocytoma (AFH) presenting severe inflammatory symptoms. The cytokine/chemokine profile of serum samples before and after surgery demonstrated that interleukin (IL)-6 had decreased by the greatest percentage. The AFH cells were immunopathologically positive for IL-6 and Tyr705-phosphorylation of signal transducer and activator of transcription 3. The EWSR1-CREB1 fusion gene detected in the tumor leads to continuous activation of CREB1 and IL-6 production, because the promoter region of IL-6 has a CREB binding site. Thus, IL-6 plays pivotal roles in both paraneoplastic syndrome and the oncogenesis of AFH.

KW - Angiomatoid fibrous histiocytoma

KW - EWSR1-CREB1 fusion gene

KW - Interleukin-6

KW - Paraneoplastic syndrome

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Paraneoplastic syndrome of angiomatoid fibrous histiocytoma may be caused by EWSR1-CREB1 fusion-induced excessive interleukin-6 production

Abstract

Keywords

ASJC Scopus subject areas

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