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1.
Zhonghua Er Ke Za Zhi ; 57(10): 767-773, 2019 Oct 02.
Artigo em Chinês | MEDLINE | ID: mdl-31594063

RESUMO

Objective: To summarize the clinical characteristics, treatment response and prognostic factors of rhabdomyosarcoma (RMS) in children. Methods: The clinical characteristics such as age at diagnosis, primary tumor site, tumor size, pathological type, clinical stage, and risk grouping of 213 RMS patients (140 males and 73 females) treated in Hematology Oncology Center of Beijing Children's Hospital, Capital Medical University, from May 2006 to June 2018 were analyzed retrospectively. The clinical characteristics, overall survival (OS), event free survival (EFS) and prognostic factors of children treated with the Beijing Children's Hospital-Rhabdomyosarcoma (BCH-RMS) regimen were analyzed. Survival data were analyzed by Kaplan-Meier survival analysis, and single factor analysis was performed by Log-Rank test. Results: The diagnostic age of 213 cases was 48.0 months (ranged 3.0-187.5 months), of which 136 cases (63.8%) were younger than 10 years old. The head and neck region was the most common primary site of tumor (30%, 64 cases), followed by the genitourinary tract (26.8%, 57 cases). Among pathological subtypes, embryonal RMS accounted for 71.4% (152 cases), while alveolar RMS and anaplastic RMS accounted for only 26.8% (57 cases) and 1.9% (4 cases), respectively. According to the Intergroup Rhabdomyosarcoma Study Group (IRS), IRS-Ⅲ and Ⅳ accounted for 85.0% (181 cases) of all RMS patients. In all patients, 9.4% (20 cases) patients were divided in to low-risk group, 52.1% (111 cases) patients in to intermediate -risk group, 25.8% (55 cases) patients in to high-risk group, and 12.7% (27 cases) patients in to the central nervous system invasion group, respectively. All patients with RMS received chemotherapy. The cycles of chemotherapy were 13.5 (ranged 5.0-18.0) for patients without event occurrence, while 14.2 (ranged 3.0-30.0) for patients with event occurrence. Among the 213 patients, 200 patients had surgical operation, of whom 103 patients underwent surgery before chemotherapy and 97 patients at the end of chemotherapy, 21 patients had secondary surgical resection. Radiotherapy was performed in 114 patients. The follow-up time was 23.0 months (ranged 0.5-151.0 months) . There were 98 patients with relapsed or progressed disease and 67 patients with death. The median time to progression was 10 months, of which 67 (68.4%) relapse occurred within 1 year and no recurrence occurred after follow-up for more than 5 years. The 3-year EFS and 5-year EFS were (52±4) % and (48±4) %, while the 3-year OS and 5-year OS were (65±4) % and (64±4) % by survival analysis. The 5-year OS of the low-risk, intermediate-risk, the high-risk were 100%, (74±5) %, (48±8) %, and the 2-year OS of the central nervous system invasion group was (36±11) % (χ(2)=33.52, P<0.01). The 5-year EFS of the low-risk, intermediate-risk, the high-risk were (93±6) %, (51±5) %, (36±7) % and the 2-year EFS of the central nervous system invasion group was (31±10) % (χ(2)=24.73, P<0.01) . Survival factor analysis suggested that the OS of children was correlated with age(χ(2)=4.16, P=0.038), tumor TNM stage (χ(2)=22.02, P=0.001), IRS group (χ(2)=4.49, P<0.01) and the risk group (χ(2)=33.52, P<0.01). Conclusions: This study showed that the median age of newly diagnosed RMS patients was 4 years. The head and neck and the genitourinary tract were the most common primary origin of RMS. The OS was low in single-center RMS children. The median time to recurrence was 10 months, and recurrence was rare 3 years later.


Assuntos
Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Neoplasias de Cabeça e Pescoço/diagnóstico , Neoplasias de Cabeça e Pescoço/terapia , Rabdomiossarcoma/diagnóstico , Rabdomiossarcoma/terapia , Neoplasias Urogenitais/diagnóstico , Neoplasias Urogenitais/terapia , Adolescente , Criança , Pré-Escolar , Terapia Combinada , Intervalo Livre de Doença , Feminino , Neoplasias de Cabeça e Pescoço/mortalidade , Neoplasias de Cabeça e Pescoço/patologia , Humanos , Masculino , Recidiva Local de Neoplasia/terapia , Prognóstico , Estudos Retrospectivos , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/patologia , Rabdomiossarcoma Alveolar , Rabdomiossarcoma Embrionário , Análise de Sobrevida , Neoplasias Urogenitais/mortalidade , Neoplasias Urogenitais/patologia
2.
Int J Radiat Oncol Biol Phys ; 105(5): 1043-1054, 2019 12 01.
Artigo em Inglês | MEDLINE | ID: mdl-31419513

RESUMO

PURPOSE: The standard of care of childhood parameningeal rhabdomyosarcoma (pRMS) is chemotherapy and local radiation therapy. Protons are increasingly being used to decrease late effects. The aim of the present study is to analyze the pattern of relapse and the correlation with dosimetric factors in pRMS treated with proton therapy. METHODS AND MATERIALS: This retrospective evaluation includes children treated in our institution for pRMS. Information on demographics, treatment, tumor characteristics, and toxicities and outcome was prospectively collected within the in-house registry. For patients presenting with local relapse, a fusion of the dosimetry with magnetic resonance imaging displaying site and geometry of recurrence was performed. RESULTS: Median follow-up time was 2.9 years (0.5-4.7). Forty-six patients were identified in our institution between July 2013 and November 2017. Main characteristics of patients were as follows: 56.5% male, median age 5.1 years (1.3-17.5), 39.1% alveolar histology, 26.1%, 52.2%, 8.7%, and 13% patients with subgroup risk classification D, E/F/G, H, or metastatic, respectively, median total prescribed dose 55.8 Gy (50.4-56.4). Estimated 2-year local control, metastasis-free survival, event-free survival, and overall survival were 83.8%, 87.8%, 76.9%, and 88.9%, respectively. No acute or late local toxicity exceeding grade 3 was observed. Risk-group was identified as prognostic factor for metastasis-free survival in univariate analysis but not in multivariate analysis (trend: P = .09). In this cohort, dosimetric factors did not correlate with outcome. Isolated local failure happened in 5 of the 11 relapses. Local relapses were matched with dosimetry for 6 patients: 4 of them occurred in the high dose volume and 2 in the intermediate or low dose volume. CONCLUSIONS: Proton therapy was effective and well feasible even in a critical cohort. Still, local relapse within the target volume of the radiation therapy remains an important issue in pRMS and new treatment strategies are needed.


Assuntos
Neoplasias Meníngeas/radioterapia , Recidiva Local de Neoplasia , Terapia com Prótons , Rabdomiossarcoma/radioterapia , Adolescente , Análise de Variância , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Criança , Pré-Escolar , Intervalo Livre de Doença , Estudos de Viabilidade , Feminino , Seguimentos , Humanos , Lactente , Imagem por Ressonância Magnética/métodos , Masculino , Neoplasias Meníngeas/diagnóstico por imagem , Neoplasias Meníngeas/mortalidade , Neoplasias Meníngeas/cirurgia , Recidiva Local de Neoplasia/diagnóstico por imagem , Recidiva Local de Neoplasia/mortalidade , Prognóstico , Terapia com Prótons/efeitos adversos , Lesões por Radiação/patologia , Dosagem Radioterapêutica , Planejamento da Radioterapia Assistida por Computador , Estudos Retrospectivos , Rabdomiossarcoma/diagnóstico por imagem , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/cirurgia , Rabdomiossarcoma Alveolar/diagnóstico por imagem , Rabdomiossarcoma Alveolar/mortalidade , Rabdomiossarcoma Alveolar/radioterapia , Rabdomiossarcoma Alveolar/cirurgia , Fatores de Tempo , Resultado do Tratamento
3.
J Cancer Res Clin Oncol ; 145(11): 2793-2802, 2019 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-31444550

RESUMO

BACKGROUND: The purpose of this study was to explore the demographics, multimodality therapeutic outcomes*** and prognostic factors in sinonasal rhabdomyosarcoma (SNRMS). METHODS: We conducted a retrospective analysis of 40 patients who underwent treatment of SNRMS from March 2007 to March 2018. The Kaplan-Meier method and the log-rank test were used to assess survival rates. The Cox regression model was used for multivariate survival analysis. RESULTS: In total, 25 males and 15 females were included in the study; the median age was 33 years (range, 2-67 years). All patients underwent surgical resection, and surgery prior to or after adjuvant therapy (chemotherapy and radiotherapy) was performed in 91.4% of the patients. The overall 1-, 3- and 5-year survival rates were 77.0%, 46.5% and 46.5%, respectively, during a mean follow-up time of 27.9 (range, 2-128) months in all patients. The log-rank test showed Intergroup Rhabdomyosarcoma Study (IRS) group and infiltration of the skull base influenced overall survival (p = 0.001; p = 0.022). Advanced IRS stage, lymph node metastasis and tumor size ≥ 5 cm were also associated with an unfavorable outcome on overall survival (p = 0.01; p = 0.035; p = 0.02). The results of multivariate regression analysis showed patients with IRS group I were associated with better prognosis outcome on overall survival. CONCLUSION: Patients with SNRMS have poor 5-year overall survival, and IRS group is the independent prognostic factor for overall survival.


Assuntos
Neoplasias dos Seios Paranasais/mortalidade , Rabdomiossarcoma/mortalidade , Adolescente , Adulto , Idoso , Criança , Pré-Escolar , Terapia Combinada , Feminino , Seguimentos , Humanos , Masculino , Pessoa de Meia-Idade , Neoplasias dos Seios Paranasais/terapia , Prognóstico , Estudos Retrospectivos , Rabdomiossarcoma/terapia , Taxa de Sobrevida , Adulto Jovem
4.
Pediatr Blood Cancer ; 66(11): e27932, 2019 11.
Artigo em Inglês | MEDLINE | ID: mdl-31385434

RESUMO

PURPOSE: The presence of pleural effusion or ascites at the time of diagnosis is generally considered a poor prognostic factor for children with rhabdomyosarcoma (RMS), and treatment is usually intensified despite the fact that there are no published studies to support this decision. We investigated the prognostic role of the presence of pleural effusion or ascites at diagnosis in patients with localized RMS consecutively enrolled in the Italian Soft Tissue Sarcoma Committee protocols over a 30-year period. METHODS: We reviewed the radiological reports at diagnosis of 150 children with supradiaphragmatic and infradiaphragmatic RMS, noting any presence of effusion and its extent (minimal, moderate, or massive). All patients received intensive chemotherapy, surgery, and standard or hyperfractionated radiotherapy. RESULTS: Effusion was identified in 32 children (21.3%), 14 with pleural effusion and 18 with ascites. As for its extent, 13 children presented with minimal, 12 with moderate, and 7 with massive effusion. The 5-year progression-free survival (PFS) rate was 49.8% (confidence interval [CI] 31.7-65.5) and 49.5% (CI 40-58.2) for patients with and without effusion, respectively (P = .5). When only patients with moderate or massive effusion were considered, however, their PFS was 36.8% (CI 16.5-57.5) versus 51.2% (CI 42.2-59.5) in patients with minimal or no effusion (P = .01). On the whole, patients with pleural effusion had a very poor outcome with a 5-year PFS of 35.7% (CI 13-59.4). CONCLUSIONS: The presence of moderate or massive effusion seems to be an unfavorable prognostic factor in children with RMS, and justifies their inclusion in experimental studies.


Assuntos
Ascite/etiologia , Derrame Pleural Maligno/etiologia , Rabdomiossarcoma/mortalidade , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Criança , Pré-Escolar , Ensaios Clínicos como Assunto/estatística & dados numéricos , Terapia Combinada , Gerenciamento Clínico , Feminino , Humanos , Lactente , Itália/epidemiologia , Estimativa de Kaplan-Meier , Masculino , Estudos Multicêntricos como Assunto/estatística & dados numéricos , Especificidade de Órgãos , Prognóstico , Intervalo Livre de Progressão , Estudos Retrospectivos , Rabdomiossarcoma/complicações , Rabdomiossarcoma/terapia , Resultado do Tratamento
5.
Gulf J Oncolog ; 1(30): 22-28, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-31242978

RESUMO

INTRODUCTION: Paediatric soft tissue sarcoma treatments and outcomes have improved significantly in the last few decades. However, a significant number of patients still succumb to the disease. In low-middle income countries there are dual problems of advanced disease at presentation and financial burden leading to poor compliance to therapy. Hence, we designed a low-cost oral metronomic chemotherapy protocol for these patients and studied the responses and toxicities to therapy in a tertiary referral hospital. PATIENTS AND METHODS: This is retrospective, single institutional, observational study. We retrospectively reviewed data of patients with relapsed, refractory or metastatic soft tissue sarcoma (STS) [ Ewing Sarcoma (ES); Rhabdomyosarcoma (RMS) or other STS] who were treated with the metronomic protocol of oral Tamoxifen, Etoposide and Cyclophosphamide (TEC) during the period April 1998 to September 2013, at the Tata Memorial Hospital, Parel, Mumbai. Patients with ES and RMS were primarily treated on our Institutional protocols. The patients included in the analysis were those who had relapsed after the primary protocols and then treated with metronomic TEC protocol; or those with primary refractory or metastatic disease (RMS, ES) and received metronomic TEC therapy. RESULTS: 49 patients were enrolled. Among the 49 patients, 32 were diagnosed ES, 13 RMS and 4 other STS. For the whole cohort response rates (RR) were 59% and clinical benefit rate (CBR) was 79%. Patients in the study were grouped into the following subgroups. Systemic recurrent/relapsed disease (N=24), metastatic disease at presentation (N=15) and local disease (refractory/recurrent) (N=10). None of the patients required blood or platelet support or admission for supportive care. The PFS for the above groups were 16.8 months, 12.5 months and 126.68 months respectively. This compares favorably with other historical cohorts in a similar setting. CONCLUSIONS: This study provides a preliminary evidence efficacy and tolerability of metronomic chemotherapy in poor risk ES and RMS. It also demonstrates that with this low-cost low risk treatment few patients could go into long term remissions despite high disease burden.


Assuntos
Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Resistencia a Medicamentos Antineoplásicos , Recidiva Local de Neoplasia/mortalidade , Rabdomiossarcoma/mortalidade , Terapia de Salvação , Sarcoma de Ewing/mortalidade , Sarcoma/mortalidade , Administração Metronômica , Adolescente , Adulto , Neoplasias Ósseas/tratamento farmacológico , Neoplasias Ósseas/mortalidade , Neoplasias Ósseas/patologia , Criança , Pré-Escolar , Ciclofosfamida/administração & dosagem , Etoposídeo/administração & dosagem , Feminino , Seguimentos , Humanos , Metástase Linfática , Masculino , Pessoa de Meia-Idade , Recidiva Local de Neoplasia/tratamento farmacológico , Recidiva Local de Neoplasia/patologia , Prognóstico , Estudos Retrospectivos , Rabdomiossarcoma/tratamento farmacológico , Rabdomiossarcoma/secundário , Sarcoma/tratamento farmacológico , Sarcoma/secundário , Sarcoma de Ewing/tratamento farmacológico , Sarcoma de Ewing/secundário , Taxa de Sobrevida , Tamoxifeno/administração & dosagem , Adulto Jovem
6.
Pediatr Blood Cancer ; 66(9): e27805, 2019 09.
Artigo em Inglês | MEDLINE | ID: mdl-31131973

RESUMO

We describe our experience in managing nine children and adolescents with rhabdomyosarcoma (RMS) and peritoneal involvement. The radiological pattern of peritoneal involvement was diverse from only ascites to solid peritoneal mass/omental caking. Treatment included systemic chemotherapy in all, surgery in three, and radiotherapy in eight. Two patients with presumed nonmalignant ascites, no solid peritoneal metastasis, nonalveolar histology, near-complete resection of residual disease, and radiotherapy survived long term. One patient has just completed treatment, and the remaining six relapsed/progressed at the time of reporting. Five of six patients died after a median of 5 (3-7) months from relapse despite second-line chemotherapy.


Assuntos
Neoplasias Peritoneais , Rabdomiossarcoma , Adolescente , Adulto , Criança , Pré-Escolar , Intervalo Livre de Doença , Feminino , Humanos , Masculino , Metástase Neoplásica , Neoplasia Residual , Neoplasias Peritoneais/diagnóstico , Neoplasias Peritoneais/mortalidade , Neoplasias Peritoneais/patologia , Neoplasias Peritoneais/terapia , Rabdomiossarcoma/diagnóstico por imagem , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/patologia , Rabdomiossarcoma/terapia , Taxa de Sobrevida , Reino Unido
7.
J Surg Res ; 241: 205-214, 2019 09.
Artigo em Inglês | MEDLINE | ID: mdl-31028942

RESUMO

BACKGROUND: The significance of lymph node sampling (LNS) on disease-specific survival (DSS) of extremity soft tissue sarcomas (STS) is unknown. We investigated the effect of LNS on DSS in child and adolescent extremity STS. MATERIALS AND METHODS: The Surveillance, Epidemiology, and End Results registry was queried for patients aged <20 y with extremity STS who underwent surgery. Patient demographics were collected and analyzed. RESULTS: A total of 1550 patients were included, with findings of 10-y DSS of 74% for all extremity STS and 49% for rhabdoymyosarcoma (RMS) (P < 0.005). LNS was associated with worse DSS in patients with extremity nonrhabdomyosacrcoma soft tissue sarcomas (79% versus 84%, P = 0.036). Conversely, LNS was associated with an improved DSS in patients with extremity RMS (64% versus 49%, P = 0.005). CONCLUSIONS: LNS is positively associated with an improved DSS in child and adolescent extremity RMS. Multivariate analysis found no correlation between DSS and LNS in child and adolescent extremity nonrhabdomyosarcoma soft tissue sarcomas.


Assuntos
Excisão de Linfonodo/estatística & dados numéricos , Metástase Linfática/patologia , Rabdomiossarcoma/cirurgia , Adolescente , Criança , Pré-Escolar , Extremidades , Feminino , Seguimentos , Humanos , Lactente , Recém-Nascido , Estimativa de Kaplan-Meier , Linfonodos/patologia , Linfonodos/cirurgia , Masculino , Estudos Retrospectivos , Rabdomiossarcoma/mortalidade , Programa de SEER/estatística & dados numéricos , Resultado do Tratamento , Adulto Jovem
8.
Pediatr Blood Cancer ; 66(7): e27725, 2019 07.
Artigo em Inglês | MEDLINE | ID: mdl-30920113

RESUMO

OBJECTIVES: To evaluate the impact of local therapies on the outcome of patients with liver-bile duct rhabdomyosarcoma (LBDRMS). METHODS: Data of 30 patients included in the EpSSG-RMS 2005 study were analyzed. RESULTS: The median age at diagnosis was 3 years (11 months-8 years). All patients had non-alveolar histology. Fifteen patients had a tumor > 5 cm and six had enlarged regional lymph nodes on imaging. Eight patients (27%) had primary surgery (1 R0). Six of them received external beam radiotherapy (EBRT). All are in first complete remission (CR1) except one (R1, EBRT+ , local relapse, death). Six patients (20%) received EBRT without surgery: one had local relapse and died. Sixteen patients (53%) underwent delayed surgery, with 12 achieving R0 margins, which were higher than those in the primary surgery group (P = 0.003). Three patients with R0 margins received EBRT; one had a metastatic relapse and died. Nine patients with R0 resection did not receive EBRT, three relapsed locally (two deaths). Four R1 patients received additional EBRT without relapses. Local relapse occurred in two among 19 patients with EBRT and three among 11 without EBRT (P = 0.326). At a median follow-up of 61 months (48-84 months), five patients died; all had a tumor size > 5 cm (P = 0.01). The five-year overall survival was 85% (95% CI, 65-94), and event-free survival was 76% (95% CI, 54-89). CONCLUSION: This analysis did not show any significant difference in outcome between irradiated and nonirradiated patients. Local relapse in LBDRMS is related to initial tumor size and is often fatal.


Assuntos
Neoplasias dos Ductos Biliares , Recidiva Local de Neoplasia , Rabdomiossarcoma , Neoplasias dos Ductos Biliares/diagnóstico por imagem , Neoplasias dos Ductos Biliares/mortalidade , Neoplasias dos Ductos Biliares/radioterapia , Neoplasias dos Ductos Biliares/cirurgia , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Masculino , Metástase Neoplásica , Recidiva Local de Neoplasia/diagnóstico , Recidiva Local de Neoplasia/mortalidade , Recidiva Local de Neoplasia/radioterapia , Recidiva Local de Neoplasia/cirurgia , Rabdomiossarcoma/diagnóstico por imagem , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/radioterapia , Rabdomiossarcoma/cirurgia
9.
Pediatr Blood Cancer ; 66(6): e27652, 2019 06.
Artigo em Inglês | MEDLINE | ID: mdl-30762282

RESUMO

BACKGROUND: Rhabdomyosarcoma (RMS) diagnosed during the first year of life is reported to have poor outcome. Little is known about treatment and outcome data of relapsed disease (RD). METHODS: Characteristics, treatment, and outcome of 155 patients ≤ 12 months registered within the Cooperative Weichteilsarkom Studiengruppe (CWS) between 1981 and 2016 were evaluated. RESULTS: Localized disease (LD) was diagnosed in 144 patients and metastatic disease (MD) in 11. The histological diagnosis was alveolar (RMA) (n = 38, 23/25 examined patients PAX7/3:FOXO1-positive), embryonal (RME) (n = 100), botryoid (n = 10), anaplastic (n = 1), and spindle-cell RMS (n = 6). Multimodal treatment including conventional (age-adjusted) chemotherapy (CHT) (n = 150), resection (n = 137), and radiotherapy (RT) (n = 37) was administered. Complete remission was achieved in 129 of 144 patients with LD. RD occurred in 51 infants at a median age of 1.7 years (range, 0.3-8.8). Sixty-three percent of patients with RMA suffered RD, in contrast to 28% of patients with RME. Relapse treatment consisted of conventional CHT (n = 48), resection (n = 28), and RT (n = 21). The pattern of relapse and best resection were significant prognostic factors for patients with RD (P = 0.000 and P = 0.002). Late effects occurred as secondary malignancies in 6%, long-term toxicity in 21%, and resection-related impairment in 33% of the 105 surviving patients. The 5-year event-free survival and overall survival for infants with initial LD were 51% and 69%, 14% and 14% for patients with initial MD and 39% and 41% for relapsed patients, respectively. CONCLUSION: Multimodal treatment including microscopically complete resection is strongly recommended to achieve a good prognosis in LD and RD of infants with RMS.


Assuntos
Recidiva Local de Neoplasia/mortalidade , Sistema de Registros/estatística & dados numéricos , Rabdomiossarcoma/mortalidade , Criança , Pré-Escolar , Ensaios Clínicos como Assunto , Terapia Combinada , Feminino , Seguimentos , Humanos , Lactente , Recém-Nascido , Masculino , Metástase Neoplásica , Recidiva Local de Neoplasia/diagnóstico , Recidiva Local de Neoplasia/terapia , Prognóstico , Estudos Prospectivos , Rabdomiossarcoma/diagnóstico , Rabdomiossarcoma/terapia , Taxa de Sobrevida
10.
J Clin Oncol ; 37(9): 723-730, 2019 03 20.
Artigo em Inglês | MEDLINE | ID: mdl-30702969

RESUMO

PURPOSE: To evaluate the clinical significance of indeterminate pulmonary nodules at diagnosis (defined as ≤ 4 pulmonary nodules < 5 mm or 1 nodule measuring ≥ 5 and < 10 mm) in patients with pediatric rhabdomyosarcoma (RMS). PATIENTS AND METHODS: We selected patients with supposed nonmetastatic RMS treated in large pediatric oncology centers in the United Kingdom, France, Italy, and the Netherlands, who were enrolled in the European Soft Tissue Sarcoma Study Group (E pSSG) RMS 2005 study. Patients included in the current study received a diagnosis between September 2005 and December 2013, and had chest computed tomography scans available for review that were done at time of diagnosis. Local radiologists were asked to review the chest computed tomography scans for the presence of pulmonary nodules and to record their findings on a standardized case report form. In the E pSSG RMS 2005 Study, patients with indeterminate pulmonary nodules were treated identically to patients without pulmonary nodules, enabling us to compare event-free survival and overall survival between groups by log-rank test. RESULTS: In total, 316 patients were included; 67 patients (21.2%) had indeterminate pulmonary nodules on imaging and 249 patients (78.8%) had no pulmonary nodules evident at diagnosis. Median follow-up for survivors (n = 258) was 75.1 months; respective 5-year event-free survival and overall survival rates (95% CI) were 77.0% (64.8% to 85.5%) and 82.0% (69.7% to 89.6%) for patients with indeterminate nodules and 73.2% (67.1% to 78.3%) and 80.8% (75.1% to 85.3%) for patients without nodules at diagnosis ( P = .68 and .76, respectively). CONCLUSION: Our study demonstrated that indeterminate pulmonary nodules at diagnosis do not affect outcome in patients with otherwise localized RMS. There is no need to biopsy or upstage patients with RMS who have indeterminate pulmonary nodules at diagnosis.


Assuntos
Neoplasias Pulmonares/secundário , Nódulos Pulmonares Múltiplos/secundário , Rabdomiossarcoma/secundário , Neoplasias de Tecidos Moles/patologia , Nódulo Pulmonar Solitário/secundário , Adolescente , Criança , Pré-Escolar , Europa (Continente) , Feminino , Humanos , Lactente , Recém-Nascido , Neoplasias Pulmonares/diagnóstico por imagem , Neoplasias Pulmonares/mortalidade , Neoplasias Pulmonares/terapia , Masculino , Nódulos Pulmonares Múltiplos/diagnóstico por imagem , Nódulos Pulmonares Múltiplos/mortalidade , Nódulos Pulmonares Múltiplos/terapia , Valor Preditivo dos Testes , Intervalo Livre de Progressão , Rabdomiossarcoma/diagnóstico por imagem , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/terapia , Fatores de Risco , Neoplasias de Tecidos Moles/diagnóstico por imagem , Neoplasias de Tecidos Moles/mortalidade , Neoplasias de Tecidos Moles/terapia , Nódulo Pulmonar Solitário/diagnóstico por imagem , Nódulo Pulmonar Solitário/mortalidade , Nódulo Pulmonar Solitário/terapia , Fatores de Tempo , Tomografia Computadorizada por Raios X , Carga Tumoral , Adulto Jovem
11.
Cancer ; 125(2): 290-297, 2019 01 15.
Artigo em Inglês | MEDLINE | ID: mdl-30351457

RESUMO

BACKGROUND: The outcome for patients with metastatic rhabdomyosarcoma (RMS) remains poor. A previous Children's Oncology Group (COG) study (ARST0431) for patients with metastatic RMS produced no improvement in outcome using multiple cytotoxic agents in a dose-intensive manner. The authors report results from the subsequent COG study (ARST08P1), which evaluated the feasibility and efficacy of adding cixutumumab (insulin-like growth factor-1 monoclonal antibody) or temozolomide to the ARST0431 intensive chemotherapy backbone. METHODS: Two nonrandomized pilot studies were conducted in patients with metastatic RMS, initially to determine feasibility, and both pilots were expanded to assess efficacy. All patients received 54 weeks of chemotherapy, including vincristine/irinotecan, interval-compressed vincristine/doxorubicin/cyclophosphamide alternating with ifosfamide/etoposide, and vincristine/dactinomycin/cyclophosphamide. In pilot 1, patients received intravenous cixutumumab (3, 6, or 9 mg/kg) once weekly throughout therapy. In pilot 2, patients received oral temozolomide (100 mg/m2 ) daily for 5 days with irinotecan. All patients received radiation to the primary tumor and to metastatic sites. RESULTS: One hundred sixty-eight eligible patients were enrolled (97 on pilot 1 and 71 on pilot 2). Most patients were aged ≥10 years (73%), with alveolar histology (70%), and had bone and/or bone marrow metastases (59%). Toxicities observed in each pilot were similar to those reported on ARST0431. With a median follow-up of 2.9 years, the 3-year event-free survival rate was 16% (95% confidence interval, 7%-25%) with cixutumumab and 18% (95% confidence interval, 2%-35%) with temozolomide. CONCLUSIONS: The addition of cixutumumab or temozolomide to intensive multiagent chemotherapy for metastatic RMS was safe and feasible. Neither agent improved outcome compared with the same chemotherapy that was used on ARST0431.


Assuntos
Protocolos de Quimioterapia Combinada Antineoplásica/efeitos adversos , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Rabdomiossarcoma/tratamento farmacológico , Adolescente , Anticorpos Monoclonais/administração & dosagem , Anticorpos Monoclonais Humanizados , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Recém-Nascido , Masculino , Projetos Piloto , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/patologia , Taxa de Sobrevida , Temozolomida/administração & dosagem , Resultado do Tratamento
12.
J Craniofac Surg ; 30(2): e113-e116, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-30550438

RESUMO

OBJECTIVE: The aim of the study was to explore an optimal surgery way for head and neck rhabdomyosarcoma (HNRMS) children who have failed primary chemotherapy. METHODS: A total of 51 HNRMS children who have failed primary chemotherapy were retrospectively analyzed from April 2005 to May 2017. Surgery was performed in 2 ways, widely radical resection (22 patients) and conservative resection (29 patients). Multivariate analysis was performed to identify the various variables related to overall survival (OS). RESULTS: The estimated 5-year OS was 53%. Embryonic RMS enjoyed a favorable outcome than those nonembryonic RMS (P = 0.03). Head and neck rhabdomyosarcoma children who received partial remission (PR) after primary chemotherapy enjoyed a better outcome than those only achieved stable disease (SD) (P = 0.006). A total of 22 children accepted widely radical resection, whereas 29 patients got conservative resection. Interestingly, the 2 groups did not have a statistical significance (P = 0.86). However, the latter group children have conserved more important organs, such as eyeball, facial nerves, and enough mandible or maxilla bones, and have enjoyed a better life quality. CONCLUSION: Primary chemotherapy is most important for HNRMS children, which influences the prognosis of HNRMS widely. Conservative resection is an optimal surgery way for HNRMS, bringing a better life quality for these children.


Assuntos
Dissecação , Neoplasias de Cabeça e Pescoço , Quimioterapia de Indução/métodos , Complicações Pós-Operatórias , Qualidade de Vida , Rabdomiossarcoma , Adolescente , Criança , Pré-Escolar , China/epidemiologia , Dissecação/efeitos adversos , Dissecação/métodos , Feminino , Neoplasias de Cabeça e Pescoço/tratamento farmacológico , Neoplasias de Cabeça e Pescoço/mortalidade , Neoplasias de Cabeça e Pescoço/patologia , Neoplasias de Cabeça e Pescoço/cirurgia , Humanos , Masculino , Análise Multivariada , Complicações Pós-Operatórias/epidemiologia , Complicações Pós-Operatórias/etiologia , Complicações Pós-Operatórias/psicologia , Prognóstico , Indução de Remissão/métodos , Estudos Retrospectivos , Rabdomiossarcoma/tratamento farmacológico , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/patologia , Rabdomiossarcoma/cirurgia , Análise de Sobrevida
13.
Int J Radiat Oncol Biol Phys ; 103(1): 19-27, 2019 01 01.
Artigo em Inglês | MEDLINE | ID: mdl-30138647

RESUMO

PURPOSE: For infants with localized rhabdomyosarcoma who were enrolled on Children's Oncology Group ARST0331 and ARST0531, local therapy guidelines were provided, but adherence was not mandated because of toxicity concerns. We examined adherence to protocol for local therapy guidelines, treatment variations, and outcomes for these patients. METHODS: Children aged ≤24 months who were enrolled on ARST0331 and ARST0531 were evaluated. Data were verified through radiologic, surgical, pathologic, and clinical records. Local therapy was assessed for adherence to protocol guidelines, with variations termed "individualized local therapy." The subdistribution hazards model assessed local failure, the Kaplan-Meier product limit method assessed event-free survival (EFS) and overall survival, and the log-rank test was used to evaluate prognostic impact. RESULTS: The median age of the patients was 14 months, and 124 patients were eligible. Common primary sites were genitourinary (40%), parameningeal (14%), and the extremities (11%). Most patients had unresected disease (group 3, 64%) and embryonal histology (73%). Fifty-eight percent of patients received radiation therapy at a median of week 12 (weeks 1-45). The median radiation dose was 48.6 Gy (30.6-54 Gy). Forty-three percent of patients received individualized local therapy (outside protocol guidelines), typically omission or delay of radiation therapy. Delayed primary excision was performed in 28% at a median of week 14 (weeks 7-34). With a median follow-up of 5.6 years, the 5-year local failure, EFS, and overall survival rates were 24%, 68%, and 82%, respectively. Local failure was significantly higher (35%) in patients receiving individualized local therapy than in patients who received protocol-specified local therapy (16%; P = .02). The site of failure was local in 64% of patients, local and distant in 5%, and distant only in 23%. EFS was significantly higher among patients who were aged 12 to 24 months, had tumors ≤5 cm, had group 1/2 disease, and underwent protocol-specified therapy. CONCLUSIONS: Local recurrence was the predominant pattern of failure and was more common in those receiving individualized local therapy. De-escalation of effective therapies because of concerns about treatment toxicity should be considered cautiously.


Assuntos
Rabdomiossarcoma/radioterapia , Feminino , Fidelidade a Diretrizes , Humanos , Lactente , Recém-Nascido , Masculino , Dosagem Radioterapêutica , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/patologia , Rabdomiossarcoma/cirurgia
14.
Tumori ; 105(2): 138-143, 2019 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-30131003

RESUMO

INTRODUCTION: From 2002 to 2011, the Italian Soft Tissue Sarcoma Committee explored a combination of topotecan and carboplatin as a second-line strategy for children with resistant or relapsing rhabdomyosarcoma. METHODS: Patients received two blocks of topotecan 2 mg/m2 on days 1, 2, and 3, and carboplatin 250 mg/m2 on days 4 and 5, followed by alternating blocks of topotecan-cyclophosphamide and carboplatin-etoposide for a total of six courses with 3-week intervals. Tumor response was assessed after two cycles, and local control was implemented when feasible. RESULTS: A total of 38 patients were included in this study: 18/38 had alveolar rhabdomyosarcoma (RMS), 10/38 had metastatic disease at diagnosis, 8/38 had tumor progression during first-line chemotherapy, 21/38 had locoregional relapses, and 9/38 had distant relapses. Thirty-two patients could be assessed for tumor response to topotecan-carboplatin, and 9 (28%) showed a complete or partial response. Twenty-four patients experienced grade IV hematologic toxicity, while transient grade 1 tubulopathy, grade 3 mucositis, transient grade 2 nephrotoxicity, and a grade 2 decline in cardiac function occurred in one patient each. The 5-year overall and progression-free survival rates were 17% and 14%, respectively. CONCLUSION: the prognosis for children with resistant or relapsing RMS remains unsatisfactory. The topotecan-carboplatin regimen was well-tolerated. Though in case of late relapse the response rate was similar to those reported for other regimes, the result achieved remains unsatisfactory. New approaches, possibly including target agents, seem more attractive for future studies.


Assuntos
Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Carboplatina/administração & dosagem , Rabdomiossarcoma/tratamento farmacológico , Topotecan/administração & dosagem , Adolescente , Criança , Pré-Escolar , Resistencia a Medicamentos Antineoplásicos/efeitos dos fármacos , Feminino , Humanos , Lactente , Estimativa de Kaplan-Meier , Masculino , Recidiva Local de Neoplasia/tratamento farmacológico , Rabdomiossarcoma/mortalidade , Adulto Jovem
15.
J Formos Med Assoc ; 118(1 Pt 2): 332-340, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-29903549

RESUMO

BACKGROUND: Taiwan Pediatric Oncology Group (TPOG) initiated two consecutive protocols for treating pediatric patients with rhabdomyosarcoma since 1995. However, the results have not been analyzed and reported yet. The aim of this study is to investigate the treatment results of these two protocols in our hospital and to assess whether the results are comparable to other large-scaled studies. METHODS: Treatment of pediatric patients with rhabdomyosarcoma according to TPOG protocols at National Taiwan University Hospital began in 1995. Between 1995 and 2006, patients were treated by TPOG RMS 95 protocol, which was based on IRS-III/IV. After 2007, patients were treated by TPOG RMS 2007 protocol which was adapted from IRS-V study. The clinical data of patients were obtained retrospectively by reviewing medical records. The date of the latest follow-up was December 31, 2016. RESULTS: Thirty-seven patients were enrolled in this study. The 5-year overall survival (OS) and event-free survival rates of them were 54.7 ± 8.8% and 48.5 ± 8.6%, respectively. The 5-year OS rates for patients treated by TPOG RMS 95 and TPOG RMS 2007 protocols were 55.0 ± 11.1% and 55.9 ± 14.0%, respectively. Age at diagnosis of less than ten years old and receiving operation with gross total or subtotal tumor resection were identified as independent prognostic factors that predicted better outcomes in the multivariate analysis. CONCLUSION: The clinical outcomes of pediatric patients with rhabdomyosarcoma in Taiwan improved dramatically after incorporating two consecutive protocols from TPOG. In addition, the treatment results of these two protocols were comparable to large-scale studies of other countries.


Assuntos
Terapia Combinada/métodos , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/terapia , Adolescente , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Recém-Nascido , Masculino , Análise Multivariada , Prognóstico , Estudos Retrospectivos , Rabdomiossarcoma/patologia , Análise de Sobrevida , Taiwan/epidemiologia
16.
J Pediatr Hematol Oncol ; 41(2): 152-154, 2019 03.
Artigo em Inglês | MEDLINE | ID: mdl-29668538

RESUMO

The incidence of central nervous system (CNS) involvement in patients with rhabdomyosarcoma (RMS) is low, and the outcome is dismal. We present a single institution analysis of CNS involvement of pediatric RMS. In 59 patients, the prevalence of CNS involvement was 11.9% (7 patients), higher than prior reports. Of the 6 deaths from disease, all had rapid progression, with a median survival of 14 days. The higher incidence could be secondary to treatment modifications or more sensitive detection. These findings are useful for decisions at the time of CNS involvement and could lead to modifications for future RMS clinical trials.


Assuntos
Neoplasias do Sistema Nervoso Central , Rabdomiossarcoma , Adolescente , Adulto , Neoplasias do Sistema Nervoso Central/mortalidade , Neoplasias do Sistema Nervoso Central/terapia , Criança , Pré-Escolar , Intervalo Livre de Doença , Feminino , Humanos , Masculino , Prevalência , Estudos Retrospectivos , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/terapia , Taxa de Sobrevida
17.
Med Sci Monit ; 24: 6113-6118, 2018 Sep 02.
Artigo em Inglês | MEDLINE | ID: mdl-30173244

RESUMO

BACKGROUND The purpose of this study was to investigate the treatment outcomes and evaluate the prognostic factors of adult sinonasal sarcomas. MATERIAL AND METHODS A retrospective review was performed on consecutive patients with adult sinonasal sarcomas treated in our institution from 2005 to 2016. The Kaplan-Meier method was used to evaluate local recurrence-free survival (LRFS), distant metastases-free survival (DMFS), and overall survival (OS). Univariate and multivariate analyses using Cox proportional hazard models were performed to determine the prognostic factors associated with survival outcomes. RESULTS A total of 49 patients were followed up for 6-122 months, with a median time of 36 months. The 5-year LRFS, DMFS, and OS rates of all patients were 68.3%, 62.8%, and 43.2%, respectively. The results of univariate analysis revealed that patients with an advanced stage of primary tumor and those who received incomplete surgical resection had worse LRFS (p=0.013; p=0.026). Patients with the histological type rhabdomyosarcoma (RMS) and existing regional lymph node metastasis had worse DMFS (p=0.000; p=0.001). The histological type RMS, advanced stage of primary tumor, existing regional lymph node metastasis, and receiving incomplete surgical resection had an unfavorable effect on OS (p=0.001; p=0.002; p=0.008; p=0.011). The results of multivariate analysis showed that histological type and degree of surgical resection were the independent prognostic factors for OS. CONCLUSIONS Our results suggest that the histological type RMS and receiving incomplete surgical resection are independent prognostic factors for worse OS.


Assuntos
Neoplasias do Seio Maxilar/mortalidade , Neoplasias do Seio Maxilar/terapia , Rabdomiossarcoma/mortalidade , Adolescente , Adulto , Idoso , Intervalo Livre de Doença , Feminino , Humanos , Estimativa de Kaplan-Meier , Masculino , Pessoa de Meia-Idade , Análise Multivariada , Recidiva Local de Neoplasia , Estadiamento de Neoplasias , Prognóstico , Modelos de Riscos Proporcionais , Estudos Retrospectivos , Rabdomiossarcoma/patologia , Sarcoma/mortalidade , Sarcoma/patologia , Sarcoma/terapia , Taxa de Sobrevida , Resultado do Tratamento
18.
Thorac Cancer ; 9(9): 1180-1184, 2018 09.
Artigo em Inglês | MEDLINE | ID: mdl-30062849

RESUMO

BACKGROUND: This study was conducted to explore the influence of clinical features of rhabdomyosarcoma (RMS) and a refined therapeutic protocol on the therapeutic efficacy and prognosis in children in the past five years. METHODS: Forty children diagnosed with RMS were retrospectively studied, using a version of the therapeutic protocol refined by Shanghai Children's Medical Center (version 2009.9.1). The patients' demographic characteristics, clinical manifestations, pathological features, therapeutic efficacy, and prognosis were analyzed. RESULTS: Of the 40 children, 17 abandoned treatment. Of the remaining 23 cases, two children were rated as low risk, 12 as medium risk, and nine as high risk, and all received treatment. Patients in the low and medium-risk groups had better prognosis than those in the high-risk group, and treated patients had higher survival rates and longer survival than untreated patients. CONCLUSION: Children with RMS should be treated positively. Combined treatment shows better therapeutic efficacy and prognosis. The refined therapeutic protocol seems more effective than the standard treatment, with a significant impact on long-term RMS prognosis.


Assuntos
Rabdomiossarcoma/diagnóstico , Rabdomiossarcoma/terapia , Protocolos de Quimioterapia Combinada Antineoplásica/efeitos adversos , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Criança , Pré-Escolar , Terapia Combinada , Feminino , Humanos , Lactente , Masculino , Gradação de Tumores , Estadiamento de Neoplasias , Prognóstico , Rabdomiossarcoma/mortalidade , Resultado do Tratamento
19.
J Clin Oncol ; 36(27): 2770-2777, 2018 09 20.
Artigo em Inglês | MEDLINE | ID: mdl-30091945

RESUMO

Purpose Intermediate-risk rhabdomyosarcoma (RMS) includes patients with either nonmetastatic, unresected embryonal RMS (ERMS) with an unfavorable primary site or nonmetastatic alveolar RMS (ARMS). The primary aim of this study was to improve the outcome of patients with intermediate-risk RMS by substituting vincristine and irinotecan (VI) for half of vincristine, dactinomycin, and cyclophosphamide (VAC) courses. All patients received a lower dose of cyclophosphamide and earlier radiation therapy than in previous trials. Patients and Methods Patients were randomly assigned at study entry to either VAC (cumulative cyclophosphamide dose, 16.8 g/m2) or VAC/VI (cumulative cyclophosphamide dose, 8.4 g/m2) for 42 weeks of therapy. Radiation therapy started at week 4, with individualized local control plans permitted for patients younger than 24 months. The primary study end point was event-free survival (EFS). The study design had an 80% power (5% one-sided α-level) to detect an improved long-term EFS from 65% (with VAC) to 76% (with VAC/VI). Results A total of 448 eligible patients were enrolled in the study. At a median follow-up of 4.8 years, the 4-year EFS was 63% with VAC and 59% with VAC/VI ( P = .51), and 4-year overall survival was 73% for VAC and 72% for VAC/VI ( P = .80). Within the ARMS and ERMS subgroups, no difference in outcome by treatment arm was found. Severe hematologic toxicity was less common with VAC/VI therapy. Conclusion The addition of VI to VAC did not improve EFS or OS for patients with intermediate-risk RMS. VAC/VI had less hematologic toxicity and a lower cumulative cyclophosphamide dose, making VAC/VI an alternative standard therapy for intermediate-risk RMS.


Assuntos
Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico , Rabdomiossarcoma/tratamento farmacológico , Neoplasias de Tecidos Moles/tratamento farmacológico , Criança , Pré-Escolar , Ciclofosfamida/administração & dosagem , Ciclofosfamida/efeitos adversos , Dactinomicina/administração & dosagem , Dactinomicina/efeitos adversos , Feminino , Humanos , Lactente , Recém-Nascido , Irinotecano/administração & dosagem , Irinotecano/efeitos adversos , Masculino , Intervalo Livre de Progressão , Rabdomiossarcoma/mortalidade , Rabdomiossarcoma/patologia , Neoplasias de Tecidos Moles/mortalidade , Neoplasias de Tecidos Moles/patologia , Vincristina/administração & dosagem , Vincristina/efeitos adversos
20.
Ann Diagn Pathol ; 36: 50-60, 2018 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-30098515

RESUMO

The present study was aimed at evaluating clinicopathologic and immunohistochemical (IHC) features of 300 rhabdomyosarcomas (RMSs), including differential IHC expression and prognostic value of myogenin and MyoD1 across various subtypes of RMSs. IHC expression of myogenin and MyoD1 was graded on the basis of percentage of tumor cells displaying positive intranuclear immunostaining i.e. grade 1 (1-25%); grade 2 (26-50%); grade 3 (51-76%) and grade 4 (76-100%).Clinical follow-up was available in 238 (79.3%) patients. Various clinicopathologic parameters were correlated with 3-year disease free survival (DFS) and overall survival (OS). There were 140 cases (46.7%) of alveolar RMS (ARMS), 90 of embryonal RMS (ERMS) (30%), 61 (20.3%) of spindle cell/sclerosing RMS and 9 cases (3%) of pleomorphic RMS. Most cases, barring pleomorphic RMSs, occurred in the first two decades (228 cases) (76%), frequently in males, in the head and neck region (126) (42%). By immunohistochemistry, desmin was positive in 292/299 (97.6%) tumors; myogenin in 238/267 (89.1%) and MyoD1 in 192/266 (72.2%) tumors. High myogenin expression (in ≥51% positive tumor cells) was significantly associated with ARMSs (95/121, 78.5%), as compared to other subtypes (48/117, 41%) (p value < 0.001). High MyoD1 expression (≥51% tumor cells) was seen in more cases of pure sclerosing, combined with spindle cell/sclerosing RMSs (10/10, 100%), as compared to the other subtypes (91/141, 67.4%) (p = 0.032). There was no significant difference between high myogenin expression and clinical outcomes. Patients without metastasis and harbouring tumors, measuring ≤5 cm showed a significant increase in OS, with p values = 0.01 and <0.001, respectively. ARMS was the most frequent subtype. There was a significant association between high myogenin expression and ARMSs and high MyoD1 expression and spindle cell/sclerosing RMSs. High myogenin expression did not correlate with clinical outcomes. Patients with smaller sized tumors and without metastasis had significantly better clinical outcomes.


Assuntos
Biomarcadores Tumorais/análise , Músculo Esquelético/metabolismo , Miogenina/metabolismo , Rabdomiossarcoma/patologia , Adulto , Criança , Diagnóstico Diferencial , Intervalo Livre de Doença , Feminino , Humanos , Imuno-Histoquímica/métodos , Masculino , Pessoa de Meia-Idade , Músculo Esquelético/patologia , Rabdomiossarcoma/diagnóstico , Rabdomiossarcoma/mortalidade
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