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1.
Clin Nucl Med ; 49(7): 662-663, 2024 Jul 01.
Artículo en Inglés | MEDLINE | ID: mdl-38758532

RESUMEN

ABSTRACT: A 16-year-old girl presented with left chest pain. Radiography and CT revealed localized abnormal calcification in the left sixth rib and sixth thoracic vertebra. Bone scintigraphy confirmed abnormal uptake of 99m Tc in the same area. An open biopsy of the sixth rib was performed, leading to the diagnosis of melorheostosis. This case showed uniformly thickened calcification throughout the rib, unlike the typical "dripping candle wax" radiography finding associated with melorheostosis. This case implies the importance of open biopsy for diagnostic confirmation in cases with atypical imaging features.


Asunto(s)
Melorreostosis , Costillas , Tomografía Computarizada por Rayos X , Humanos , Melorreostosis/diagnóstico por imagen , Femenino , Costillas/diagnóstico por imagen , Costillas/patología , Adolescente
2.
Cancer Diagn Progn ; 4(1): 71-76, 2024.
Artículo en Inglés | MEDLINE | ID: mdl-38173666

RESUMEN

Background/Aim: High complication rates during the perioperative management of sarcomas around the pelvis have been reported; however, few include the detailed clinical course or complications in the late postoperative period. Radiotherapy is a multidisciplinary strategy for treating sarcomas. However, irradiated bone and soft tissues show a permanent loss of repair and immunocompetence. We present a case of pleomorphic rhabdomyosarcoma of the thigh that resulted in acetabular collapse induced by radiation and intestinal perforation during long-term follow-up. Additionally, we discuss the risk factors for late complications and pelvic reconstruction methods. Case Report: A 75-year-old man presented with a 1-month history of a recurring fever. Ten years prior, he was diagnosed with pleomorphic rhabdomyosarcoma of the right thigh and underwent a wide resection and bipolar hip arthroplasty, followed by chemotherapy and radiotherapy. Radiographs showed central dislocation of the bipolar head. Computed tomography revealed free air in the hip joint and thickening of the colon wall. Colonoscopy revealed displacement of the bipolar head into the colon wall. Colon resection and hip disarticulation were performed, as the bipolar head was contaminated with intestinal contents. Currently, he is able to walk stably with a walker, and there is no evidence of recurrence or metastasis of the tumor. Conclusion: Irradiation of the periacetabular bone may induce resorptive destruction, resulting in future structural failure. Hardware should not be used for periacetabular bone reconstruction; the risk of pelvic organ damage should be considered when the acetabular collapse becomes deteriorated. Therefore, other reliable and permanent reconstruction options are required.

3.
Spine (Phila Pa 1976) ; 49(6): 390-397, 2024 Mar 15.
Artículo en Inglés | MEDLINE | ID: mdl-38084012

RESUMEN

STUDY DESIGN: Retrospective diagnostic study. OBJECTIVE: To automatically detect osteolytic bone metastasis lesions in the thoracolumbar region using conventional computed tomography (CT) scans, we developed a new deep learning (DL)-based computer-aided detection model. SUMMARY OF BACKGROUND DATA: Radiographic detection of bone metastasis is often difficult, even for orthopedic surgeons and diagnostic radiologists, with a consequent risk for pathologic fracture or spinal cord injury. If we can improve detection rates, we will be able to prevent the deterioration of patients' quality of life at the end stage of cancer. MATERIALS AND METHODS: This study included CT scans acquired at Tokyo Medical and Dental University (TMDU) Hospital between 2016 and 2022. A total of 263 positive CT scans that included at least one osteolytic bone metastasis lesion in the thoracolumbar spine and 172 negative CT scans without bone metastasis were collected for the datasets to train and validate the DL algorithm. As a test data set, 20 positive and 20 negative CT scans were separately collected from the training and validation datasets. To evaluate the performance of the established artificial intelligence (AI) model, sensitivity, precision, F1-score, and specificity were calculated. The clinical utility of our AI model was also evaluated through observer studies involving six orthopaedic surgeons and six radiologists. RESULTS: Our AI model showed a sensitivity, precision, and F1-score of 0.78, 0.68, and 0.72 (per slice) and 0.75, 0.36, and 0.48 (per lesion), respectively. The observer studies revealed that our AI model had comparable sensitivity to orthopaedic or radiology experts and improved the sensitivity and F1-score of residents. CONCLUSION: We developed a novel DL-based AI model for detecting osteolytic bone metastases in the thoracolumbar spine. Although further improvement in accuracy is needed, the current AI model may be applied to current clinical practice. LEVEL OF EVIDENCE: Level III.


Asunto(s)
Aprendizaje Profundo , Neoplasias de la Columna Vertebral , Humanos , Inteligencia Artificial , Neoplasias de la Columna Vertebral/diagnóstico por imagen , Estudios Retrospectivos , Calidad de Vida , Tomografía Computarizada por Rayos X/métodos , Algoritmos
4.
JBMR Plus ; 7(7): e10749, 2023 Jul.
Artículo en Inglés | MEDLINE | ID: mdl-37457876

RESUMEN

Patients on bone-modifying agents (BMAs) for bone metastases are at risk of atypical femoral fractures (AFFs), which can lead to a sudden deterioration in performance status. In this study, we sought to determine the prevalence of radiographic precursory signs of AFF in patients on oncologic BMAs. Forty-two patients (23 men, 19 women; mean age 68.8 ± 10.0 years) on oncologic BMAs (zoledronate for >3 years and/or denosumab for >1 year) and without clinical symptoms were enrolled between 2019 and 2021. All patients were receiving denosumab at enrollment and 5 had previously used zoledronate. The mean duration of BMA use was 31.2 ± 18.5 months. Radiographs of both femurs were screened for precursory signs of AFF (e.g., thickening of the lateral cortex). The patients were divided into two groups according to thickening status and compared by duration of BMA use. They were also divided into three groups by duration of BMA use (12-23 months, n = 18; 24-59 months, n = 19; ≥60 months, n = 5), and the prevalence of apparent thickenings was examined. As a result, 18 patients (42.9%) showed minute local or diffuse thickening and 10 (23.8%) showed apparent local thickening. The duration of BMA use was significantly longer in patients with apparent thickening than in those without (47.3 ± 23.6 months [n = 10] versus 26.2 ± 13.5 months [n = 32]; p < 0.05). The prevalence of apparent thickening increased with increasing duration of BMA use (12-23 months, 5.6%; 24-59 months, 31.6%; ≥60 months, 60.0%). In conclusion, radiographic precursory signs of AFF are common in patients on oncologic BMAs. Radiographic screening for AFF could be relevant in patients who have been on long-term oncologic BMAs, even if asymptomatic. © 2023 The Authors. JBMR Plus published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research.

6.
Clin Cancer Res ; 28(12): 2633-2645, 2022 06 13.
Artículo en Inglés | MEDLINE | ID: mdl-35381070

RESUMEN

PURPOSE: Osteosarcoma, the most common bone malignancy in children, has a poor prognosis, especially when the tumor metastasizes to the lungs. Therefore, novel therapeutic strategies targeting both proliferation and metastasis of osteosarcoma are required. Podoplanin (PDPN) is expressed by various tumors and is associated with tumor-induced platelet activation via its interaction with C-type lectin-like receptor 2 (CLEC-2) on platelets. We previously found that PDPN contributed to osteosarcoma growth and metastasis through platelet activation; thus, in this study, we developed an anti-PDPN humanized antibody and evaluated its effect on osteosarcoma growth and metastasis. EXPERIMENTAL DESIGN: Nine osteosarcoma cell lines and two osteosarcoma patient-derived cells were collected, and we evaluated the efficacy of the anti-DPN-neutralizing antibody PG4D2 and the humanized anti-PDPN antibody AP201, which had IgG4 framework region. The antitumor and antimetastasis effect of PG4D2 and AP201 were examined in vitro and in vivo. In addition, growth signaling by the interaction between PDPN and CLEC-2 was analyzed using phospho-RTK (receptor tyrosine kinase) array, growth assay, or immunoblot analysis under the supression of RTKs by knockout and inhibitor treatment. RESULTS: We observed that PG4D2 treatment significantly suppressed tumor growth and pulmonary metastasis in osteosarcoma xenograft models highly expressing PDPN. The contribution of PDGFR activation by activated platelet releasates to osteosarcoma cell proliferation was confirmed, and the humanized antibody, AP201, suppressed in vivo osteosarcoma growth and metastasis without significant adverse events. CONCLUSIONS: Targeting PDPN with a neutralizing antibody against PDPN-CLEC-2 without antibody-dependent cell-mediated cytotoxicity and complement-dependent cytotoxicity is a novel therapeutic strategy for PDPN-positive osteosarcoma.


Asunto(s)
Neoplasias Óseas , Lectinas Tipo C , Neoplasias Pulmonares , Glicoproteínas de Membrana , Osteosarcoma , Anticuerpos Neutralizantes , Neoplasias Óseas/tratamiento farmacológico , Línea Celular Tumoral , Humanos , Neoplasias Pulmonares/metabolismo , Osteosarcoma/tratamiento farmacológico
7.
Virchows Arch ; 480(6): 1269-1275, 2022 Jun.
Artículo en Inglés | MEDLINE | ID: mdl-34635937

RESUMEN

Epithelioid malignant peripheral nerve sheath tumor (MPNST) is a rare subtype of MPNST composed of epithelioid cells with abundant cytoplasm. Currently, strong and diffuse immunostaining for S100 protein and SOX10 is generally regarded as a characteristic feature of epithelioid MPNST. However, malignant tumors with epithelioid morphology that arise from a peripheral nerve or a pre-existing benign nerve sheath tumor should be regarded as epithelioid MPNSTs when they do not show characteristic features that definitively lead to other specific diagnoses. Here, we describe 3 cases of epithelioid MPNST in the peripheral nerve or schwannoma that was negative for S100 protein and SOX10 expression. Instead, these tumors were positive for EMA, GLUT1, claudin 1, and cytokeratin to varying degrees, while all of them retained SMARCB1 and H3K27me3 by immunohistochemistry. EMA, GLUT1, and claudin 1 are known markers of perineurial cell differentiation; thus, they could possibly represent epithelioid MPNST with perineurial cell differentiation.


Asunto(s)
Neurofibrosarcoma , Biomarcadores de Tumor , Diferenciación Celular , Claudina-1 , Transportador de Glucosa de Tipo 1 , Humanos , Neurofibrosarcoma/patología , Nervios Periféricos , Proteínas S100 , Factores de Transcripción SOXE
8.
Cancers (Basel) ; 13(24)2021 Dec 14.
Artículo en Inglés | MEDLINE | ID: mdl-34944888

RESUMEN

Pazopanib with trabectedin and eribulin is widely used to treat soft-tissue sarcoma (STS). We have shown that baseline neutrophil-to-lymphocyte ratio (NLR) may predict the efficacy and patient prognosis of eribulin. Changes in NLR, but not baseline NLR, can predict patient prognosis of trabectedin. However, prognostic factors of pazopanib for STS have not been identified. We present a retrospective analysis of 141 patients treated with pazopanib for recurrent or metastatic non-round cell STS. Univariate and multivariate analyses were performed to determine the predictive factors of durable clinical benefit (DCB), overall survival (OS), and progression-free survival. L-sarcoma histology (odds ratio [OR] = 0.31, 95% CI = 0.12-0.79; p = 0.014) and pre-treatment NLR < 3.0 (OR = 2.03, 95% CI = 1.02-6.67; p = 0.045) were independent predictive factors of DCB. Pre-treatment NLR < 3.0 (hazard ratio [HR] = 0.55, 95% CI = 0.36-0.84; p = 0.0057), liposarcoma histology (HR = 1.78, 95% CI = 1.09-2.91; p = 0.022), primary extremity site (HR = 0.48, 95% CI = 0.31-0.75; p = 0.0010), ECOG PS ≥ 1 (HR = 1.62, 95% CI = 1.08-2.42; p = 0.019), and CRP < 0.3 (HR = 0.52, 95% CI = 0.33-0.82; p = 0.0050) were independent predictive factors of OS. These findings indicate that baseline NLR predicts the efficacy and patient prognosis of pazopanib for STS.

9.
Oncogene ; 40(36): 5548-5558, 2021 09.
Artículo en Inglés | MEDLINE | ID: mdl-34302117

RESUMEN

Osteosarcoma is the most common primary malignant bone cancer, with high rates of pulmonary metastasis. Osteosarcoma patients with pulmonary metastasis have worse prognosis than those with localized disease, leading to dramatically reduced survival rates. Therefore, understanding the biological characteristics of metastatic osteosarcoma and the molecular mechanisms of invasion and metastasis of osteosarcoma cells will lead to the development of innovative therapeutic intervention for advanced osteosarcoma. Here, we identified that osteosarcoma cells commonly exhibit high platelet activation-inducing characteristics, and molecules released from activated platelets promote the invasiveness of osteosarcoma cells. Given that heat-denatured platelet releasate maintained the ability to promote osteosarcoma invasion, we focused on heat-tolerant molecules, such as lipid mediators in the platelet releasate. Osteosarcoma-induced platelet activation leads to abundant lysophosphatidic acid (LPA) release. Exposure to LPA or platelet releasate induced morphological changes and increased invasiveness of osteosarcoma cells. By analyzing publicly available transcriptome datasets and our in-house osteosarcoma patient-derived xenograft tumors, we found that LPA receptor 1 (LPAR1) is notably upregulated in osteosarcoma. LPAR1 gene KO in osteosarcoma cells abolished the platelet-mediated osteosarcoma invasion in vitro and the formation of early pulmonary metastatic foci in experimental pulmonary metastasis models. Of note, the pharmacological inhibition of LPAR1 by the orally available LPAR1 antagonist, ONO-7300243, prevented pulmonary metastasis of osteosarcoma in the mouse models. These results indicate that the LPA-LPAR1 axis is essential for the osteosarcoma invasion and metastasis, and targeting LPAR1 would be a promising therapeutic intervention for advanced osteosarcoma.


Asunto(s)
Lisofosfolípidos , Osteosarcoma , Plaquetas , Humanos , Neoplasias Pulmonares , Activación Transcripcional
10.
Indian J Orthop ; 55(4): 892-897, 2021 Aug.
Artículo en Inglés | MEDLINE | ID: mdl-34194644

RESUMEN

PURPOSE: Intramuscular myxoma (IM) is a rare benign myxoid tumor that may be challenging to differentiate from sarcoma in small amounts of biopsied material. Although IM appears to be well-circumscribed macroscopically, it infiltrates the adjacent edematous muscle microscopically. The recommended treatment is resection, but there is controversy with regard to the appropriate surgical margin. This study aimed to clarify which surgical procedure that should be applied when the preoperative diagnosis is IM and how to manage treatment if the postoperative diagnosis turns out to be a sarcoma. METHODS: We retrospectively examined 55 IM patients treated from January 1982 to December 2014. Patient characteristics, tumor location, tumor size, radiograph, preoperative and postoperative pathological reports, surgical techniques, treatment outcome, and complications were reviewed. The patients were followed up on for at least 5 years. All patients were confirmed not to have Mazabraud syndrome. RESULTS: In the 55 IM patients examined, the mean patient age was 48 years and most were female. The most common tumor locations were in the muscles of the thighs (47%) and buttocks (20%). The mean tumor diameter was 5 cm. Wide resection and marginal resection were performed in 24 and 31 patients, respectively. The mean follow-up duration was 19 years. No local recurrence, malignant transformation, or complications were observed. CONCLUSIONS: Marginal resection is suitable in patients whose preoperative diagnosis is IM, as it is able to prevent local recurrence and allows for the preservation of muscle and muscle fascia. If the postoperative diagnosis turns out to be myxoid sarcoma, minimum surgical contamination makes additional wide resection less invasive.

11.
Anticancer Res ; 41(1): 527-532, 2021 Jan.
Artículo en Inglés | MEDLINE | ID: mdl-33419852

RESUMEN

BACKGROUND: Eribulin is widely used for the treatment of breast cancer and soft-tissue sarcoma (STS). Previous studies identified the pre-treatment absolute lymphocyte count, baseline neutrophil-to-lymphocyte ratio (NLR) and C-reactive protein concentration as potential prognostic markers in patients with breast cancer treated with eribulin. However, prognostic factors for eribulin treatment in patients with STS have not been identified. PATIENTS AND METHODS: This was a retrospective analysis of data collected prospectively from 53 patients who were treated with eribulin for recurrent or metastatic STS between March 2016 and August 2019. Univariate and multivariate analyses were performed to determine the predictive factors of durable clinical benefit, progression-free survival, and overall survival. RESULTS: L-Sarcoma histology [hazard ratio (HR)=28.20, 95% confidence intervaI (CI)=1.67-476.00; p=0.021] and pre-treatment NLR <3.0 (HR=9.96, 95% CI=1.28-77.7; p=0.028) were independent factors predictive of durable clinical benefit. In addition, pre-treatment NLR <3.0 (HR=0.34, 95% CI=0.16-0.74; p=0.0059) and male sex (HR=0.23, 95% CI=0.10-0.52; p<0.001) were independent factors predictive of better progression-free survival. CONCLUSION: This retrospective study found that baseline NLR predicts the efficacy of eribulin for STS.


Asunto(s)
Antineoplásicos/uso terapéutico , Furanos/uso terapéutico , Cetonas/uso terapéutico , Recuento de Leucocitos , Linfocitos , Neutrófilos , Sarcoma/sangre , Sarcoma/tratamiento farmacológico , Anciano , Anciano de 80 o más Años , Antineoplásicos/administración & dosificación , Antineoplásicos/efectos adversos , Biomarcadores , Femenino , Furanos/administración & dosificación , Furanos/efectos adversos , Humanos , Estimación de Kaplan-Meier , Cetonas/administración & dosificación , Cetonas/efectos adversos , Masculino , Persona de Mediana Edad , Pronóstico , Retratamiento , Estudios Retrospectivos , Sarcoma/diagnóstico , Sarcoma/mortalidad , Resultado del Tratamiento
12.
Mol Clin Oncol ; 14(1): 13, 2021 Jan.
Artículo en Inglés | MEDLINE | ID: mdl-33282288

RESUMEN

Clinical evidence regarding eribulin treatment for patients with soft tissue sarcoma (STS) is limited to those with L-sarcoma (leiomyosarcoma and liposarcoma) who have completed at least two chemotherapies. Whether histological subtypes and treatment lines affect the efficacy and safety of eribulin for patients with STS has yet to be elucidated. The current study retrospectively reviewed patients with STS receiving eribulin at the Cancer Institute Hospital of JFCR and evaluated the prognostic factors affecting its efficacy and safety by histological diagnoses and treatment lines. A total of 41 patients with STS, including 26 with L-sarcoma, underwent eribulin treatment. Additionally, a total of and 14 patients, including 12 with L-sarcoma, received eribulin as a second-line treatment. The results revealed that patients with L-sarcoma demonstrated longer progression-free survival (PFS) rates compared with patients without L-sarcoma (4.5 vs. 2.3 months; P=0.005). Furthermore, differences in treatment line significantly affected PFS (4.5 months in second-line treatment vs. 2.4 months in later lines; P=0.037). A high number of patients with L-sarcoma received eribulin as a second-line treatment. Regarding safety, several adverse events were reported, such as neutropenia, which were more frequently observed in patients with L-sarcoma or other patients receiving eribulin as a second-line treatment. However, most adverse events were tolerable. The clinical efficacy of eribulin was increased in patients with L-sarcoma, which was similar to previous clinical trials. However, treatment lines could also affect its efficacy. When evaluating the clinical value of eribulin to STS, it is important to consider treatment lines.

13.
Cancer Diagn Progn ; 1(4): 303-308, 2021.
Artículo en Inglés | MEDLINE | ID: mdl-35403143

RESUMEN

Background/Aim: Trabectedin and eribulin are widely used for the treatment of soft-tissue sarcoma (STS). Previously it was shown that the baseline neutrophil-to-lymphocyte ratio (NLR) predicts the efficacy of eribulin for STS. However, prognostic factors for trabectedin on STS have not been identified to date. Patients and Methods: We conducted a retrospective study of data collected prospectively from 39 patients treated with trabectedin for recurrent or metastatic STS between October 2012 and December 2019. To determine the predictive factors of overall survival (OS) and progression-free survival (PFS), univariate and multivariate analyses were performed. Results: Age ≥40 (HR=0.33, 95% CI=0.15-0.71; p=0.0050) and changes in NLR (ΔNLR) <0.5 (HR=2.40, 95% CI-1.01-5.72; p=0.048) were independent factors predictive of longer OS. In addition, age ≥40 (HR=0.23, 95% CI=0.10-0.52; p<0.001) was an independent predictor of longer PFS. Conclusion: Changes in NLR and age ≥40 years were able to predict the efficacy of trabectedin for STS.

14.
Oxf Med Case Reports ; 2020(11): omaa076, 2020 Nov.
Artículo en Inglés | MEDLINE | ID: mdl-33269078

RESUMEN

A skip metastasis was defined as a solitary separate focus of osteosarcoma occurring synchronously with a primary osteosarcoma in the absence of anatomic extension. The progression of skip metastasis is considered less likely because the articular cartilage acts as a barrier, so there have been few reports on progression of the extremity bone tumor across a joint. In our case report, the acetabular osteosarcoma progressed to the femoral head through the ligament of the femoral head. From the findings of magnetic resonance imaging and resected specimen and tissue specimen, we considered that the tumor progressed between ligament and synovial tissue covering the ligament, and not passing through the inside of the ligament. This case suggested a possibility that the tumor might progress through the synovium around the ligament of femoral head in the cases of osteosarcoma arising from the proximal femur and acetabulum.

15.
J Orthop Traumatol ; 21(1): 13, 2020 Aug 31.
Artículo en Inglés | MEDLINE | ID: mdl-32865641

RESUMEN

BACKGROUND: Recently, the number of osteosarcomas in middle-aged and older patients has demonstrated an increasing trend; moreover, their results are comparatively worse than those of young patients. In Europe and the USA, the prognosis for osteosarcoma in middle-aged and older patients has improved with adjuvant chemotherapy. In Japan, however, the prognosis has remained poor. MATERIALS AND METHODS: We retrospectively analyzed the outcomes of osteosarcoma, especially in regards to preoperative chemotherapy, from January 1980 to July 2014. A total of 29 patients with high-grade osteosarcoma between the age of 40 and 65 years were included. We included patients without distant metastasis and with primary lesions that were deemed resectable. The mean age was 52.8 years (range 41-65 years), and the mean follow-up period was 103.2 months (range 5-314 months). RESULTS: Adjuvant chemotherapy was administered to 27 of 29 patients (93%), and 8 of 15 cases (53%) were able to undergo preoperative chemotherapy as planned, including CDDP. A major complication of chemotherapy was acute kidney injury due to CDDP (26%). The 5-year OS and 5-year EFS were 64.9% and 57.1%, respectively. After 2006, a policy to prioritize the resection of the primary lesion was implemented, and if the primary lesion was deemed resectable, preoperative chemotherapy was either not administered or administered for only a short duration. The 5-year OS after 2006 improved to 78.8%. CONCLUSIONS: This study shows that administration of high-dose intensity preoperative chemotherapy was difficult in middle-aged and older patients due to their high rate of acute kidney injury by CDDP. For cases of osteosarcoma in middle-aged and older patients, if the primary lesion is resectable, preoperative chemotherapy should be minimized to prioritize the resection of the primary lesion. It was considered that, with appropriate measures to prevent complications, adjuvant chemotherapy may lead to improved prognosis. LEVEL OF EVIDENCE: V.


Asunto(s)
Neoplasias Óseas/tratamiento farmacológico , Neoplasias Óseas/cirugía , Osteosarcoma/tratamiento farmacológico , Osteosarcoma/cirugía , Adulto , Factores de Edad , Anciano , Neoplasias Óseas/patología , Quimioterapia Adyuvante , Femenino , Humanos , Masculino , Persona de Mediana Edad , Clasificación del Tumor , Osteosarcoma/patología , Pronóstico , Estudios Retrospectivos , Tasa de Supervivencia
16.
Clin Case Rep ; 8(7): 1213-1216, 2020 Jul.
Artículo en Inglés | MEDLINE | ID: mdl-32695360

RESUMEN

Nodular fasciitis (NF) is a rapid-growth benign that is misdiagnosed as sarcoma and leads to overtreatment. The spontaneous regression of NF is a possible phenomenon. "Wait and see" ideal is one of the treatment strategies of NF.

17.
Anticancer Res ; 40(2): 1029-1034, 2020 Feb.
Artículo en Inglés | MEDLINE | ID: mdl-32014949

RESUMEN

BACKGROUND: The role of neoadjuvant chemotherapy for localized synovial sarcoma is still controversial. This study aimed to explore the influence of neoadjuvant chemotherapy combined with surgery in localized synovial sarcoma through analysis of our hospital's patient records. PATIENTS AND METHODS: A total of 122 patients diagnosed with synovial sarcoma were enrolled in this study from January 1980 to December 2016 at the Cancer Institute Hospital of The Japanese Foundation for Cancer Research. The impact of neoadjuvant chemotherapy on overall survival was assessed to show how clinicopathological factors (e.g. age, tumor size, treatment, dose intensity, pathological pattern and histological grading) influenced patient prognosis. RESULTS: Among 106 patients, 76 (71.7%) received neoadjuvant chemotherapy and 30 (28.3%) did not. The median follow-up was 39.2 (range=12-286) months. The 5-year and 10-year overall survival rates were 65.4% and 58.4% respectively. The tumor size and histological grade influenced the patient's overall survival (p<0.05). Among the patients with grade 2 tumor, tumor size did not influence prognosis. Neoadjuvant chemotherapy improved the overall survival of patients who had grade 3 tumors. CONCLUSION: Treatment with neoadjuvant chemotherapy proved beneficial for high-risk patients with grade 3 synovial sarcoma but was not effective for those with low-risk and grade 2 tumor. Tumor size and histological grade were important factors in patient prognosis but had no connection with pathological patterns.


Asunto(s)
Protocolos de Quimioterapia Combinada Antineoplásica/uso terapéutico , Sarcoma Sinovial/tratamiento farmacológico , Adulto , Protocolos de Quimioterapia Combinada Antineoplásica/efectos adversos , Quimioterapia Adyuvante , Terapia Combinada , Femenino , Humanos , Estimación de Kaplan-Meier , Masculino , Persona de Mediana Edad , Terapia Neoadyuvante , Clasificación del Tumor , Estadificación de Neoplasias , Sarcoma Sinovial/diagnóstico , Sarcoma Sinovial/mortalidad , Resultado del Tratamiento , Carga Tumoral , Adulto Joven
18.
Anticancer Res ; 40(2): 1035-1039, 2020 Feb.
Artículo en Inglés | MEDLINE | ID: mdl-32014950

RESUMEN

BACKGROUND: Extraskeletal myxoid chondrosarcoma (EMC) is a rare malignant soft-tissue tumor and often shows extracompartmental tumoral invasion. The aim of our study was to investigate the clinical features, especially extracompartmental tumoral invasion (ETI) of EMC. PATIENTS AND METHODS: A total of 35 operative patients diagnosed with EMC were enrolled in this study from January 1980 to March 2018 in the Cancer Institute Hospital of The Japanese Foundation for Cancer Research. The operative procedure was principally wide excision. Univariate analysis assessed how clinicopathological factors (e.g. age, gender, tumor site, tumor size, histopathological grade, surgical margin, metastasis before operation, barrier invasion, local recurrence, metastasis after operation) influenced patient prognosis. We assessed how clinicopathological factors influenced ETI of EMC. RESULTS: Among 35 patients, 10 patients showed ETI. The average follow-up was 5.57 (range=0.2-20 years). The 5- and 10-year overall survival was 91.3% and 71.2%, respectively. The 5- and 10-year overall survival of patients with M0 disease was 96.1% and 73.2%, respectively, while both were 75.0% for those with M1 disease, respectively. The patients with distant metastasis at first visit tended to have a poor prognosis (p=0.07). It is notable that all of the 10 patients with ETI had distant metastasis after surgery. CONCLUSION: Patients with distant metastasis at first visit tended to have a poor prognosis. ETI of EMC induced distant metastasis after surgery. Patients with ETI of EMC should, therefore, be carefully monitored over a prolonged period.


Asunto(s)
Condrosarcoma/patología , Neoplasias de los Tejidos Conjuntivo y Blando/patología , Adulto , Anciano , Condrosarcoma/diagnóstico por imagen , Condrosarcoma/etiología , Condrosarcoma/terapia , Femenino , Humanos , Estimación de Kaplan-Meier , Imagen por Resonancia Magnética , Masculino , Márgenes de Escisión , Persona de Mediana Edad , Clasificación del Tumor , Invasividad Neoplásica , Metástasis de la Neoplasia , Estadificación de Neoplasias , Neoplasias de los Tejidos Conjuntivo y Blando/diagnóstico por imagen , Neoplasias de los Tejidos Conjuntivo y Blando/etiología , Neoplasias de los Tejidos Conjuntivo y Blando/terapia , Pronóstico , Factores de Riesgo
19.
Pathol Int ; 69(12): 706-709, 2019 Dec.
Artículo en Inglés | MEDLINE | ID: mdl-31538390

RESUMEN

Nodular fasciitis (NF) is a self-limiting benign disease that is characterized by rapid proliferation of fibroblastic and myofibroblastic cells. The characteristic gene fusion containing the USP6 gene is a genetic hallmark of NF and MYH9-USP6 is the most frequent fusion, suggesting that NF is not a reactive condition but a neoplastic disease. Malignant transformation of NF has been reported rarely as a single case associated with the PPP6R3-USP6 fusion. Here we report a case of soft part tumor of which the histological feature was a typical NF but showed aggressive and non-regressing growth with local invasion. Targeted RNA sequencing and fluorescence in situ hybridization analysis identified PPP6R3-USP6 with gene amplification. These findings indicate that the present case is the second case of malignant NF, and we suggest potential malignant transformation in certain NF cases.


Asunto(s)
Fascitis/diagnóstico , Neoplasias de los Tejidos Conjuntivo y Blando/diagnóstico , Fosfoproteínas Fosfatasas/genética , Ubiquitina Tiolesterasa/genética , Adulto , Transformación Celular Neoplásica , Fascitis/genética , Fascitis/patología , Fusión Génica , Reordenamiento Génico , Humanos , Hibridación Fluorescente in Situ , Masculino , Miofibroblastos/patología , Neoplasias de los Tejidos Conjuntivo y Blando/genética , Neoplasias de los Tejidos Conjuntivo y Blando/patología
20.
J Clin Med ; 8(1)2019 Jan 08.
Artículo en Inglés | MEDLINE | ID: mdl-30626115

RESUMEN

The approved standard dose of pazopanib is 800 mg per day, but the appropriate dose of pazopanib to treat soft tissue sarcoma (STS) patients in real-world practice is controversial. Of 124 STS patients treated with pazopanib, we retrospectively analyzed the cases of STS patients who achieved progression-free survival at 12 weeks by pazopanib treatment as pazopanib responders, and we evaluated their relative dose intensity (RDI) in the initial 12 weeks (12W-RDI). We enrolled 78 STS patients in the analyses as pazopanib responders, and 54 patients of the 78 pazopanib responders (69%) were able to maintain 12W-RDI ≥80%. In landmark analyses, patients with 12W-RDI of 80% ≥80% had significantly longer progression-free survival compared to those with 12W-RDI <80% (30.7 weeks vs. 22.0 weeks, hazard ratio [HR]: 0.56 [95%CI: 0.33⁻0.94], p = 0.026). The most frequently observed reasons of treatment interruption and/or dose reduction of pazopanib during the initial 12 weeks were anorexia and liver function disorders. Liver toxicity was the adverse event most frequently observed in the 12W-RDI <80% patients throughout the treatment periods. Based on our results, it appears that maintaining as high a dose intensity as possible that is tolerable-at least during the initial 12 weeks-is likely to be the better option in pazopanib treatment for STS patients.

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