Clinical Outcome of Low-Grade Myofibroblastic Sarcoma in Japan: A Multicenter Study from the Japanese Musculoskeletal Oncology Group

被引：7

作者：

Kito, Munehisa ^{[1
]}

Ae, Keisuke ^{[2
]}

Okamoto, Masanori ^{[1
]}

Endo, Makoto ^{[3
]}

Ikuta, Kunihiro ^{[4
]}

Takeuchi, Akihiko ^{[5
]}

Yasuda, Naohiro ^{[6
,7
]}

Yasuda, Taketoshi ^{[8
]}

Imura, Yoshinori ^{[9
]}

Morii, Takeshi ^{[10
]}

Kikuta, Kazutaka ^{[11
]}

Kawamoto, Teruya ^{[12
]}

Nezu, Yutaka ^{[13
]}

Baba, Ichiro ^{[14
]}

Ohshika, Shusa ^{[15
]}

Uehara, Takeshi ^{[16
]}

Ueda, Takafumi ^{[17
]}

Takahashi, Jun ^{[1
]}

Kawano, Hirotaka ^{[18
]}

机构：

[1] Shinshu Univ, Dept Orthopaed Surg, Sch Med, 3-1-1 Asahi, Matsumoto 3908621, Japan

[2] Canc Inst Hosp, Japanese Fdn Canc Res, Dept Orthopaed Surg, 3-8-31 Ariake,Koto Ku, Tokyo 1358550, Japan

[3] Kyushu Univ, Grad Sch Med Sci, Dept Orthopaed Surg, 3-1-1 Maidashi,Higashi Ku, Fukuoka 8128582, Japan

[4] Nagoya Univ, Dept Orthopaed Surg, Grad Sch Med, 65 Tsurumai,Showa Ku, Nagoya 4668560, Japan

[5] Kanazawa Univ, Grad Sch Med Sci, Dept Orthopaed Surg, 13-1 Takaramachi, Kanazawa 9208641, Japan

[6] Osaka Univ, Dept Orthopaed Surg, Grad Sch Med, 2-2 Yamadaoka, Suita 5650871, Japan

[7] Natl Hosp Org Osaka Natl Hosp, Dept Orthopaed Surg, 2-1-14 Houenzaka,Chuo Ku, Osaka 5400006, Japan

[8] Univ Toyama, Dept Orthopaed Surg, 2630 Sugitani, Toyama 9300194, Japan

[9] Osaka Int Canc Inst, Dept Orthopaed Surg, 3-1-69 Otemae,Chuo Ku, Osaka 5400008, Japan

[10] Kyorin Univ, Dept Orthopaed Surg, Fac Med, 6-20-2 Shinkawa, Tokyo 1818621, Japan

[11] Tochigi Canc Ctr, Dept Musculoskeletal Oncol & Orthopaed Surg, 4-9-13 Yonan, Utsunomiya 3200834, Japan

[12] Kobe Univ, Dept Orthopaed Surg, Grad Sch Med, 7-5-1 Kusunoki Cho,Chuo Ku, Kobe 6500017, Japan

[13] Natl Canc Ctr, Dept Musculoskeletal Oncol & Rehabil, 5-1-1 Tsukigi,Chuo Ku, Tokyo 1040045, Japan

[14] Osaka Med & Pharmaceut Univ, Dept Orthopaed Surg, 2-7 Daigakumachi, Takatsuki 5698686, Japan

[15] Hirosaki Univ, Dept Orthopaed Surg, Grad Sch Med, 5 Zaifu Cho, Takatsuki 0368562, Japan

[16] Shinshu Univ, Dept Lab Med, Sch Med, 3-1-1 Asahi, Matsumoto 3908621, Japan

[17] Kodama Hosp, Dept Orthopaed Surg, 1-3-2 Gotenyama, Takarazuka 6650841, Japan

[18] Teikyo Univ, Dept Orthopaed Surg, Sch Med, 2-11-1 Kaga,Itabashi Ku, Tokyo 1730806, Japan

来源：

CANCERS | 2023年 / 15卷 / 08期

基金：

日本学术振兴会;

关键词：

low-grade myofibroblastic sarcoma; rare sarcoma; wide excision; radiotherapy; local relapse; prognosis; SOFT-TISSUE SARCOMA; POPULATION; SYSTEMS; TUMORS; BONE; HEAD;

D O I：

10.3390/cancers15082314

中图分类号：

R73 [肿瘤学];

学科分类号：

100214 ;

摘要：

Simple Summary Low-grade myofibroblastic sarcoma (LGMS) is one of the rarest sarcomas. We aimed to clarify the clinical outcomes of patients with LGMS. Twenty-two patients underwent surgical treatment for the primary tumor and two underwent radical radiotherapy (RT). The best overall response in the two patients who underwent radical RT was one complete response and one partial response. Local relapse-free survival was 91.3% at 2 years and 75.4% at 5 years. Relapsed tumors were treated with surgery in two cases and radical RT in three cases. None of the patients experienced a second local relapse. Disease-specific survival was 100% at 5 years. Wide excision is recommended due to its tendency to local relapse. However, RT was considered a viable option in unresectable cases or in cases where surgery may cause significant functional impairment. This retrospective multicenter study aimed to analyze the clinical features and prognosis of 24 patients diagnosed with LGMS between 2002 and 2019 in the Japanese sarcoma network. Twenty-two cases were surgically treated and two cases were treated with radical radiotherapy (RT). The pathological margin was R0 in 14 cases, R1 in 7 cases, and R2 in 1 case. The best overall response in the two patients who underwent radical RT was one complete response and one partial response. Local relapse occurred in 20.8% of patients. Local relapse-free survival (LRFS) was 91.3% at 2 years and 75.4% at 5 years. In univariate analysis, tumors of 5 cm or more were significantly more likely to cause local relapse (p < 0.01). In terms of the treatment of relapsed tumors, surgery was performed in two cases and radical RT was performed in three cases. None of the patients experienced a second local relapse. Disease-specific survival was 100% at 5 years. A wide excision aimed at the microscopically R0 margin is considered the standard treatment for LGMS. However, RT may be a viable option in unresectable cases or in cases where surgery is expected to cause significant functional impairment.

引用

页数：12

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