Forty-eight cases of leiomyosarcoma of bone in Japan: A multicenter study from the Japanese musculoskeletal oncology group

Mori, Tomoaki; Nakayama, Robert; Hiraga, Hiroaki; Tomita, Masato; Fukase, Naomasa; Kobayashi, Eisuke; Kawai, Akira; Ueda, Takafumi; Morioka, Hideo

doi:10.1002/jso.24322

Cited by 25 publications

(43 citation statements)

References 22 publications

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“…1 The long bones, particularly distal femur and proximal tibia, account for 70% of cases, whereas the craniofacial bones make up about 20%. [2][3][4][5][6][7] Imaging studies typically demonstrate an osteolytic, radiolucent intramedullary mass with cortical destruction and soft tissue invasion ( Figure 1). 1,3,4 Fifteen percent of cases are associated with prior radiation.…”

Section: Clinical Featuresmentioning

confidence: 99%

“…Similar to its uterine and soft tissue counterparts, LMSB consistently expresses smooth muscle actin (SMA) ( Figure 13) and muscle-specific actin in more than 95% and 93% of cases, respectively. 3,5 However, desmin is positive in only 50% of cases ( Figure 14) and thus should not be used as the only primary screening antibody. 3,5 It is worth noting that both cytokeratin and S100 protein expression have been reported.…”

Section: Clinical Featuresmentioning

confidence: 99%

“…3,5 However, desmin is positive in only 50% of cases ( Figure 14) and thus should not be used as the only primary screening antibody. 3,5 It is worth noting that both cytokeratin and S100 protein expression have been reported. Cytokeratin can be focally positive in more than 30% of cases ( Figure 15), especially in the epithelioid variant.…”

Section: Clinical Featuresmentioning

confidence: 99%

“…Unlike primary osteosarcoma, which is generally chemosensitive, multiple studies have reported poor response of LMSB to chemotherapy, as well as minimal impact on overall survival benefit. 5,6 Unfortunately, even fewer data are available in the management of metastatic LMSB. Chemotherapy, albeit suboptimal, is often used in conjunction with patientcentered and goal-oriented management in this setting.…”

Section: Prognosis and Treatmentmentioning

confidence: 99%

“…1 The 5-year overall survival of LMSB ranges from 62% to 78%, and the 5-year disease-free survival ranges from 45% to 82%. [4][5][6] However, stratification based on histologic grade reveals notable shifts in prognostic outcomes. Several studies [3][4][5][6] have supported the finding that high-stage tumors are associated with poorer outcomes.…”

Section: Prognosis and Treatmentmentioning

confidence: 99%

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Primary Leiomyosarcoma of Bone: Review and Update

Wang

Lucas

2019

Archives of Pathology &Amp; Laboratory Medicine

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Context.— Leiomyosarcoma of bone is a rare primary osseous sarcoma characterized by smooth muscle differentiation and absence of malignant osteoid formation. Leiomyosarcoma of bone is diagnostically challenging; this can be improved with greater awareness of this entity and the ability to differentiate it from its histologic mimics. Because of its rarity, only a small number of studies are available in the literature. These factors contribute to our limited understanding of its pathology, prognosis, and treatment. Objective.— To review the clinicopathologic features of leiomyosarcoma of bone and present the most up-to-date understanding of its behavior and management in accordance with the current literature. Data Sources.— Review of pertinent literature on the major features, current knowledge thereof, and the authors' experience in the diagnosis and management of leiomyosarcoma of bone. Conclusions.— Leiomyosarcoma of bone is a rare but well-recognized primary osseous sarcoma that may arise de novo or in association with radiation. Although it is diagnostically challenging, awareness of this rare sarcoma and knowledge of its key histomorphologic and immunohistochemical features allow for accurate diagnosis.

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Section: Clinical Featuresmentioning

confidence: 99%

Section: Clinical Featuresmentioning

confidence: 99%

Section: Clinical Featuresmentioning

confidence: 99%

Section: Prognosis and Treatmentmentioning

confidence: 99%

Section: Prognosis and Treatmentmentioning

confidence: 99%

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Primary Leiomyosarcoma of Bone: Review and Update

Wang

Lucas

2019

Archives of Pathology &Amp; Laboratory Medicine

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Rare bone sarcomas: A retrospective analysis of 145 adult patients from the French Sarcoma Group

Boudou‐Rouquette

Martin

Kempf

et al. 2021

Intl Journal of Cancer

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The benefit of chemotherapy (CT) in rare bone sarcomas is not documented in prospective studies. Our retrospective study from the French sarcoma network for bone tumors ResOs was performed in adult patients (pts) from 1976 to 2014, with histologically verified diagnosis of leiomyosarcomas (LMS), undifferentiated pleomorphic sarcoma (UPS) or radiation-associated sarcomas of bone. The median follow-up was 4.7 years (95% CI: 3.7-6.5). Clinical features, treatment modalities and outcomes were recorded and analyzed from 145 pts (median age 53 years [range 20-87]). Site of disease was extremities (66%) or axial skeleton (34%), 111 (77%) presented with localized and potentially resectable disease. The most common histological subtypes were UPS (58%) and LMS (33%); 58% were high-grade tumors. Surgery was performed in 127 pts. In the 111 localized pts, 28 pts (25%) underwent upfront surgery or exclusive radiotherapy (RT; >50 Gy) without CT, whereas 83 pts (75%) received either neoadjuvant (n = 26) or adjuvant CT (n = 13) or both (n = 44). Neoadjuvant and adjuvant CT was mostly doxorubicin-based (95%/86%) and cisplatin-based (67%/63%). R0 resection was achieved in 59 pts, and a good histological response in 15 patients (25%). Adjuvant RT was performed in 24 (22%) pts. For the whole cohort (n = 145), the 5-year overall survival (OS) rate was 53% [42; 62]. In univariate analysis, age ≤ 60 was associated with a longer disease-free survival (DFS) (P = .0436). Neoadjuvant and adjuvant CT tended to be associated with better DFS (P = .056) with no significant impact on OS in this retrospective series.

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Diagnoses, treatment, and oncologic outcomes in patients with calcaneal malignances: Case series, systematic literature review, and pooled cohort analysis

Newman

Rein

Theyskens

et al. 2020

Journal of Surgical Oncology

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Background and Objectives: Malignant tumors of the calcaneus are rare but pose a treatment challenge. Aims: (1) describe the demographics of calcaneal malignancies in a large cohort; (2) describe survival after amputation versus limb-salvage surgery for high-grade tumors. Methods: Study group: a "pooled" cohort of patients with primary calcaneal malignancies treated at two cancer centers (1984−2015) and systematic literature review. Kaplan−Meier analyses described survival across treatment and diagnostic groups; proportional hazards modeling assessed mortality after amputation versus limb salvage. Results: A total of 131 patients (11 treated at our centers and 120 patients from 53 published studies) with a median 36-month follow-up were included. Diagnoses included Ewing sarcoma (41%), osteosarcoma (30%), and chondrosarcoma (17%); 5-year survival rates were 43%, 73% (70%, high grade only), and 84% (60%, high grade only), respectively. Treatment involved amputation in 52%, limb salvage in 27%, and no surgery in 21%. There was no difference in mortality following limb salvage surgery (vs. amputation) for high-grade tumors (HR 0.38; 95% CI 0.14−1.05), after adjusting for Ewing sarcoma diagnosis (HR 5.15; 95% CI 1.55−17.14), metastatic disease at diagnosis (HR 3.88; 95% CI 1.29−11.64), and age (per-year HR 1.04; 95% CI 1.02−1.07). Conclusions: Limb salvage is oncologically-feasible for calcaneal malignancies.

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Forty-eight cases of leiomyosarcoma of bone in Japan: A multicenter study from the Japanese musculoskeletal oncology group

Cited by 25 publications

References 22 publications

Primary Leiomyosarcoma of Bone: Review and Update

Primary Leiomyosarcoma of Bone: Review and Update

Rare bone sarcomas: A retrospective analysis of 145 adult patients from the French Sarcoma Group

Diagnoses, treatment, and oncologic outcomes in patients with calcaneal malignances: Case series, systematic literature review, and pooled cohort analysis

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