Clinicopathological review of solitary fibrous tumors: dedifferentiation is a major cause of patient death

Yuichi Yamada, Kenichi Kohashi, Izumi Kinoshita, Hidetaka Yamamoto, Takeshi Iwasaki, Masato Yoshimoto, Shin Ishihara, Yu Toda, Yoshihiro Itou, Yutaka Koga, Mikiko Hashisako, Yui Nozaki, Daisuke Kiyozawa, Daichi Kitahara, Takeshi Inoue, Munenori Mukai, Yumi Honda, Gouji Toyokawa, Kenji Tsuchihashi, Yoshifumi MatsushitaFumiyoshi Fushimi, Kenichi Taguchi, Sadafumi Tamiya, Yumi Oshiro, Masutaka Furue, Yasuharu Nakashima, Satoshi Suzuki, Toru Iwaki, Yoshinao Oda

研究成果: ジャーナルへの寄稿記事

抄録

Solitary fibrous tumor (SFT) is a soft-tissue neoplasm of intermediate malignant potential, presenting a wide histopathological spectrum. Poorer prognosis of hemangiopericytoma of the central nervous system (CNS), hypoglycemic SFT, and dedifferentiation are well-known characters of SFT, but their clinical significance were not demonstrated enough by large-sized study. Here, the clinicopathological features of SFTs are reviewed and the relationship between genetics and clinicopathological features is examined using 145 SFT cases. All cases were STAT6 IHC-positive and/or NAB2-STAT6 fusion gene-positive. Tumor location was classified into three categories: 30 pleuropulmonary, 96 non-pleuropulmonary/non-central nervous system (CNS), and 18 CNS tumors. The tumor developed recurrence in 21 of 93 available cases (22.5%), metastasis in 11 of 93 (11.8%), and tumor death in 9 of 93 (9.6%). Hypoglycemia occurred in 2 primary tumors and 1 metastatic tumor among 63 reviewable cases, and dedifferentiation occurred in 10 cases (6.8%) including 6 primary tumors, 2 recurrent tumors, and 2 metastatic tumors. Recurrence was positively associated with CNS location (p = 0.0109) and hypoglycemia (p = 0.001); metastasis was positively associated with CNS location (p = 0.0231), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001), while metastasis was negatively correlated with pleural location (p = 0.0471). Tumor death was positively associated with male sex (p = 0.0154), larger size (p = 0.0455), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001). Multivariate analysis revealed independent statistical significance of dedifferentiation for overall survival (p = 0.0467). Exon variant of the fusion gene had no statistical correlation with clinical outcome. In conclusion, dedifferentiation is a major prognostic factor of SFT, and specific location such as cerebromeningeal and intra-abdominal site and hypoglycemia also had a high risk for unfavorable prognosis.

元の言語英語
ページ(範囲)467-477
ページ数11
ジャーナルVirchows Archiv
475
発行部数4
DOI
出版物ステータス出版済み - 10 1 2019

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Solitary Fibrous Tumors
Cause of Death
Hypoglycemia
Neoplasms
Central Nervous System
Gene Fusion
Neoplasm Metastasis
Soft Tissue Neoplasms
Hemangiopericytoma
Recurrence
Central Nervous System Neoplasms
Hypoglycemic Agents
Nervous System
Exons
Multivariate Analysis

All Science Journal Classification (ASJC) codes

  • Pathology and Forensic Medicine
  • Molecular Biology
  • Cell Biology

これを引用

Clinicopathological review of solitary fibrous tumors : dedifferentiation is a major cause of patient death. / Yamada, Yuichi; Kohashi, Kenichi; Kinoshita, Izumi; Yamamoto, Hidetaka; Iwasaki, Takeshi; Yoshimoto, Masato; Ishihara, Shin; Toda, Yu; Itou, Yoshihiro; Koga, Yutaka; Hashisako, Mikiko; Nozaki, Yui; Kiyozawa, Daisuke; Kitahara, Daichi; Inoue, Takeshi; Mukai, Munenori; Honda, Yumi; Toyokawa, Gouji; Tsuchihashi, Kenji; Matsushita, Yoshifumi; Fushimi, Fumiyoshi; Taguchi, Kenichi; Tamiya, Sadafumi; Oshiro, Yumi; Furue, Masutaka; Nakashima, Yasuharu; Suzuki, Satoshi; Iwaki, Toru; Oda, Yoshinao.

:: Virchows Archiv, 巻 475, 番号 4, 01.10.2019, p. 467-477.

研究成果: ジャーナルへの寄稿記事

Yamada, Y, Kohashi, K, Kinoshita, I, Yamamoto, H, Iwasaki, T, Yoshimoto, M, Ishihara, S, Toda, Y, Itou, Y, Koga, Y, Hashisako, M, Nozaki, Y, Kiyozawa, D, Kitahara, D, Inoue, T, Mukai, M, Honda, Y, Toyokawa, G, Tsuchihashi, K, Matsushita, Y, Fushimi, F, Taguchi, K, Tamiya, S, Oshiro, Y, Furue, M, Nakashima, Y, Suzuki, S, Iwaki, T & Oda, Y 2019, 'Clinicopathological review of solitary fibrous tumors: dedifferentiation is a major cause of patient death', Virchows Archiv, 巻. 475, 番号 4, pp. 467-477. https://doi.org/10.1007/s00428-019-02622-9
Yamada, Yuichi ; Kohashi, Kenichi ; Kinoshita, Izumi ; Yamamoto, Hidetaka ; Iwasaki, Takeshi ; Yoshimoto, Masato ; Ishihara, Shin ; Toda, Yu ; Itou, Yoshihiro ; Koga, Yutaka ; Hashisako, Mikiko ; Nozaki, Yui ; Kiyozawa, Daisuke ; Kitahara, Daichi ; Inoue, Takeshi ; Mukai, Munenori ; Honda, Yumi ; Toyokawa, Gouji ; Tsuchihashi, Kenji ; Matsushita, Yoshifumi ; Fushimi, Fumiyoshi ; Taguchi, Kenichi ; Tamiya, Sadafumi ; Oshiro, Yumi ; Furue, Masutaka ; Nakashima, Yasuharu ; Suzuki, Satoshi ; Iwaki, Toru ; Oda, Yoshinao. / Clinicopathological review of solitary fibrous tumors : dedifferentiation is a major cause of patient death. :: Virchows Archiv. 2019 ; 巻 475, 番号 4. pp. 467-477.
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title = "Clinicopathological review of solitary fibrous tumors: dedifferentiation is a major cause of patient death",
abstract = "Solitary fibrous tumor (SFT) is a soft-tissue neoplasm of intermediate malignant potential, presenting a wide histopathological spectrum. Poorer prognosis of hemangiopericytoma of the central nervous system (CNS), hypoglycemic SFT, and dedifferentiation are well-known characters of SFT, but their clinical significance were not demonstrated enough by large-sized study. Here, the clinicopathological features of SFTs are reviewed and the relationship between genetics and clinicopathological features is examined using 145 SFT cases. All cases were STAT6 IHC-positive and/or NAB2-STAT6 fusion gene-positive. Tumor location was classified into three categories: 30 pleuropulmonary, 96 non-pleuropulmonary/non-central nervous system (CNS), and 18 CNS tumors. The tumor developed recurrence in 21 of 93 available cases (22.5{\%}), metastasis in 11 of 93 (11.8{\%}), and tumor death in 9 of 93 (9.6{\%}). Hypoglycemia occurred in 2 primary tumors and 1 metastatic tumor among 63 reviewable cases, and dedifferentiation occurred in 10 cases (6.8{\%}) including 6 primary tumors, 2 recurrent tumors, and 2 metastatic tumors. Recurrence was positively associated with CNS location (p = 0.0109) and hypoglycemia (p = 0.001); metastasis was positively associated with CNS location (p = 0.0231), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001), while metastasis was negatively correlated with pleural location (p = 0.0471). Tumor death was positively associated with male sex (p = 0.0154), larger size (p = 0.0455), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001). Multivariate analysis revealed independent statistical significance of dedifferentiation for overall survival (p = 0.0467). Exon variant of the fusion gene had no statistical correlation with clinical outcome. In conclusion, dedifferentiation is a major prognostic factor of SFT, and specific location such as cerebromeningeal and intra-abdominal site and hypoglycemia also had a high risk for unfavorable prognosis.",
author = "Yuichi Yamada and Kenichi Kohashi and Izumi Kinoshita and Hidetaka Yamamoto and Takeshi Iwasaki and Masato Yoshimoto and Shin Ishihara and Yu Toda and Yoshihiro Itou and Yutaka Koga and Mikiko Hashisako and Yui Nozaki and Daisuke Kiyozawa and Daichi Kitahara and Takeshi Inoue and Munenori Mukai and Yumi Honda and Gouji Toyokawa and Kenji Tsuchihashi and Yoshifumi Matsushita and Fumiyoshi Fushimi and Kenichi Taguchi and Sadafumi Tamiya and Yumi Oshiro and Masutaka Furue and Yasuharu Nakashima and Satoshi Suzuki and Toru Iwaki and Yoshinao Oda",
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pages = "467--477",
journal = "Virchows Archiv",
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TY - JOUR

T1 - Clinicopathological review of solitary fibrous tumors

T2 - dedifferentiation is a major cause of patient death

AU - Yamada, Yuichi

AU - Kohashi, Kenichi

AU - Kinoshita, Izumi

AU - Yamamoto, Hidetaka

AU - Iwasaki, Takeshi

AU - Yoshimoto, Masato

AU - Ishihara, Shin

AU - Toda, Yu

AU - Itou, Yoshihiro

AU - Koga, Yutaka

AU - Hashisako, Mikiko

AU - Nozaki, Yui

AU - Kiyozawa, Daisuke

AU - Kitahara, Daichi

AU - Inoue, Takeshi

AU - Mukai, Munenori

AU - Honda, Yumi

AU - Toyokawa, Gouji

AU - Tsuchihashi, Kenji

AU - Matsushita, Yoshifumi

AU - Fushimi, Fumiyoshi

AU - Taguchi, Kenichi

AU - Tamiya, Sadafumi

AU - Oshiro, Yumi

AU - Furue, Masutaka

AU - Nakashima, Yasuharu

AU - Suzuki, Satoshi

AU - Iwaki, Toru

AU - Oda, Yoshinao

PY - 2019/10/1

Y1 - 2019/10/1

N2 - Solitary fibrous tumor (SFT) is a soft-tissue neoplasm of intermediate malignant potential, presenting a wide histopathological spectrum. Poorer prognosis of hemangiopericytoma of the central nervous system (CNS), hypoglycemic SFT, and dedifferentiation are well-known characters of SFT, but their clinical significance were not demonstrated enough by large-sized study. Here, the clinicopathological features of SFTs are reviewed and the relationship between genetics and clinicopathological features is examined using 145 SFT cases. All cases were STAT6 IHC-positive and/or NAB2-STAT6 fusion gene-positive. Tumor location was classified into three categories: 30 pleuropulmonary, 96 non-pleuropulmonary/non-central nervous system (CNS), and 18 CNS tumors. The tumor developed recurrence in 21 of 93 available cases (22.5%), metastasis in 11 of 93 (11.8%), and tumor death in 9 of 93 (9.6%). Hypoglycemia occurred in 2 primary tumors and 1 metastatic tumor among 63 reviewable cases, and dedifferentiation occurred in 10 cases (6.8%) including 6 primary tumors, 2 recurrent tumors, and 2 metastatic tumors. Recurrence was positively associated with CNS location (p = 0.0109) and hypoglycemia (p = 0.001); metastasis was positively associated with CNS location (p = 0.0231), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001), while metastasis was negatively correlated with pleural location (p = 0.0471). Tumor death was positively associated with male sex (p = 0.0154), larger size (p = 0.0455), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001). Multivariate analysis revealed independent statistical significance of dedifferentiation for overall survival (p = 0.0467). Exon variant of the fusion gene had no statistical correlation with clinical outcome. In conclusion, dedifferentiation is a major prognostic factor of SFT, and specific location such as cerebromeningeal and intra-abdominal site and hypoglycemia also had a high risk for unfavorable prognosis.

AB - Solitary fibrous tumor (SFT) is a soft-tissue neoplasm of intermediate malignant potential, presenting a wide histopathological spectrum. Poorer prognosis of hemangiopericytoma of the central nervous system (CNS), hypoglycemic SFT, and dedifferentiation are well-known characters of SFT, but their clinical significance were not demonstrated enough by large-sized study. Here, the clinicopathological features of SFTs are reviewed and the relationship between genetics and clinicopathological features is examined using 145 SFT cases. All cases were STAT6 IHC-positive and/or NAB2-STAT6 fusion gene-positive. Tumor location was classified into three categories: 30 pleuropulmonary, 96 non-pleuropulmonary/non-central nervous system (CNS), and 18 CNS tumors. The tumor developed recurrence in 21 of 93 available cases (22.5%), metastasis in 11 of 93 (11.8%), and tumor death in 9 of 93 (9.6%). Hypoglycemia occurred in 2 primary tumors and 1 metastatic tumor among 63 reviewable cases, and dedifferentiation occurred in 10 cases (6.8%) including 6 primary tumors, 2 recurrent tumors, and 2 metastatic tumors. Recurrence was positively associated with CNS location (p = 0.0109) and hypoglycemia (p = 0.001); metastasis was positively associated with CNS location (p = 0.0231), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001), while metastasis was negatively correlated with pleural location (p = 0.0471). Tumor death was positively associated with male sex (p = 0.0154), larger size (p = 0.0455), hypoglycemia (p < 0.0001), and dedifferentiation (p < 0.0001). Multivariate analysis revealed independent statistical significance of dedifferentiation for overall survival (p = 0.0467). Exon variant of the fusion gene had no statistical correlation with clinical outcome. In conclusion, dedifferentiation is a major prognostic factor of SFT, and specific location such as cerebromeningeal and intra-abdominal site and hypoglycemia also had a high risk for unfavorable prognosis.

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