Survival outcomes including salvage therapy of adult head and neck para-meningeal rhabdomyosarcoma : a multicenter retrospective study from Japan
© 2023. The Author(s)..
BACKGROUND: Rhabdomyosarcoma is the most common soft tissue sarcoma in children, but rare in adults. Para-meningeal rhabdomyosarcoma in head and neck (PM-HNRMS) is less applicable for surgery due to the anatomic reason. PM-HNRMS has a poor prognosis in children. However, its clinical outcomes remain unclear in adults due to the rarity. Further, there is almost no detailed data about salvage therapy.
METHODS: We retrospectively examined the adult patients with PM-HNRMS treated at institutions belonging to the Kyushu Medical Oncology Group from 2009 to 2022. We evaluated the overall survival (OS) and progression-free survival (PFS) of the patients who received a first-line therapy. We also reviewed the clinical outcomes of patients who progressed against a first-line therapy and received salvage therapy.
RESULTS: Total 11 patients of PM-HNRMS received a first-line therapy. The characteristics were as follows: median age: 38 years (range 25 - 63 years), histology (alveolar/spindle): 10/1, and risk group (intermediate/high): 7/4. As a first-line therapy, VAC and ARST0431-based regimen was performed in 10 and 1 patients, respectively. During a first-line therapy, definitive radiation for all lesions were performed in seven patients. The median PFS was 14.2 months (95%CI: 6.0 - 25.8 months): 17.1 months (95%CI: 6.0 - not reached (NR)) for patients with stage I-III and 8.5 months (95%CI: 5.2 - 25.8 months) for patients with stage IV. The 1-year and 3-year PFS rates were 54.5% and 11.3% for all patients. Median OS in all patients was 40.8 months (95%CI: 12.1 months-NR): 40.8 months (95%CI: 12.1 - NR) for patients with stage I-III and NR for patients with stage IV. The 5-year OS rate was 48.5% for all patients. Among seven patients who received salvage therapy, three are still alive, two of whom remain disease-free for over 4 years after completion of the last therapy. Those two patients received multi-modal therapy including local therapy for all detected lesions.
CONCLUSION: The cure rate of adult PM-HNRMS is low in spite of a first-line therapy in this study. Salvage therapy might prolong the survival in patients who received the multi-modal therapy including local therapy for all detected lesions.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2023 |
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| Erschienen: |
2023 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:23 |
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| Enthalten in: |
BMC cancer - 23(2023), 1 vom: 31. Okt., Seite 1046 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
Tsuchihashi, Kenji [VerfasserIn] |
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| Links: |
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| Themen: |
Adult |
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| Anmerkungen: |
Date Completed 22.01.2024 Date Revised 22.01.2024 published: Electronic Citation Status MEDLINE |
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| doi: |
10.1186/s12885-023-11528-4 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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NLM363960759 |
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| 100 | 1 | |a Tsuchihashi, Kenji |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Survival outcomes including salvage therapy of adult head and neck para-meningeal rhabdomyosarcoma |b a multicenter retrospective study from Japan |
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| 500 | |a Date Revised 22.01.2024 | ||
| 500 | |a published: Electronic | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a © 2023. The Author(s). | ||
| 520 | |a BACKGROUND: Rhabdomyosarcoma is the most common soft tissue sarcoma in children, but rare in adults. Para-meningeal rhabdomyosarcoma in head and neck (PM-HNRMS) is less applicable for surgery due to the anatomic reason. PM-HNRMS has a poor prognosis in children. However, its clinical outcomes remain unclear in adults due to the rarity. Further, there is almost no detailed data about salvage therapy | ||
| 520 | |a METHODS: We retrospectively examined the adult patients with PM-HNRMS treated at institutions belonging to the Kyushu Medical Oncology Group from 2009 to 2022. We evaluated the overall survival (OS) and progression-free survival (PFS) of the patients who received a first-line therapy. We also reviewed the clinical outcomes of patients who progressed against a first-line therapy and received salvage therapy | ||
| 520 | |a RESULTS: Total 11 patients of PM-HNRMS received a first-line therapy. The characteristics were as follows: median age: 38 years (range 25 - 63 years), histology (alveolar/spindle): 10/1, and risk group (intermediate/high): 7/4. As a first-line therapy, VAC and ARST0431-based regimen was performed in 10 and 1 patients, respectively. During a first-line therapy, definitive radiation for all lesions were performed in seven patients. The median PFS was 14.2 months (95%CI: 6.0 - 25.8 months): 17.1 months (95%CI: 6.0 - not reached (NR)) for patients with stage I-III and 8.5 months (95%CI: 5.2 - 25.8 months) for patients with stage IV. The 1-year and 3-year PFS rates were 54.5% and 11.3% for all patients. Median OS in all patients was 40.8 months (95%CI: 12.1 months-NR): 40.8 months (95%CI: 12.1 - NR) for patients with stage I-III and NR for patients with stage IV. The 5-year OS rate was 48.5% for all patients. Among seven patients who received salvage therapy, three are still alive, two of whom remain disease-free for over 4 years after completion of the last therapy. Those two patients received multi-modal therapy including local therapy for all detected lesions | ||
| 520 | |a CONCLUSION: The cure rate of adult PM-HNRMS is low in spite of a first-line therapy in this study. Salvage therapy might prolong the survival in patients who received the multi-modal therapy including local therapy for all detected lesions | ||
| 650 | 4 | |a Multicenter Study | |
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Adult | |
| 650 | 4 | |a Head and neck | |
| 650 | 4 | |a Para-meningeal | |
| 650 | 4 | |a Rhabdomyosarcoma | |
| 650 | 4 | |a Salvage | |
| 700 | 1 | |a Ito, Mamoru |e verfasserin |4 aut | |
| 700 | 1 | |a Arita, Shuji |e verfasserin |4 aut | |
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| 700 | 1 | |a Ueno, Shohei |e verfasserin |4 aut | |
| 700 | 1 | |a Taguchi, Ryosuke |e verfasserin |4 aut | |
| 700 | 1 | |a Yoshihiro, Tomoyasu |e verfasserin |4 aut | |
| 700 | 1 | |a Doi, Yasuhiro |e verfasserin |4 aut | |
| 700 | 1 | |a Arimizu, Kohei |e verfasserin |4 aut | |
| 700 | 1 | |a Ohmura, Hirofumi |e verfasserin |4 aut | |
| 700 | 1 | |a Kajitani, Tatsuhiro |e verfasserin |4 aut | |
| 700 | 1 | |a Nio, Kenta |e verfasserin |4 aut | |
| 700 | 1 | |a Nakano, Michitaka |e verfasserin |4 aut | |
| 700 | 1 | |a Oshima, Kotoe |e verfasserin |4 aut | |
| 700 | 1 | |a Tamura, Shingo |e verfasserin |4 aut | |
| 700 | 1 | |a Shirakawa, Tsuyoshi |e verfasserin |4 aut | |
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| 700 | 1 | |a Uchino, Keita |e verfasserin |4 aut | |
| 700 | 1 | |a Hanamura, Fumiyasu |e verfasserin |4 aut | |
| 700 | 1 | |a Okumura, Yuta |e verfasserin |4 aut | |
| 700 | 1 | |a Komoda, Masato |e verfasserin |4 aut | |
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