Objective： Primary uterine leiomyosarcomas （ULMS） are rare, and the optimal treatment is controversial. We aimed to assess the outcome and prognostic factors in a multicenter population of women treated for primary...Objective： Primary uterine leiomyosarcomas （ULMS） are rare, and the optimal treatment is controversial. We aimed to assess the outcome and prognostic factors in a multicenter population of women treated for primary ULMS. Methods： We retrospectively collected data of 110 women treated in 19 institutions of the Rare Cancer Network （RCN）. Inclusion criteria consisted of a pathology report confirming the diagnosis of ULMS, aged 18-80 years, complete International Federation of Gynecology and Obstetrics （FIGO） stage information, complete information on treatment, and a minimum follow-up of 6 months. Local control （LC） and locoregional control （LRC）, overall survival （OS） and disease-free survival （DFS） rates were computed using the Kaplan-Meier method. Univariate analysis was implemented using the log rank test, and multivariate analysis using the Cox model. Results： All patients underwent surgery. Seventy-five patients （68%） received adjuvant radiotherapy （RT）, including brachytherapy in 18 （I 6%）. Seventeen patients （15 %） received adjuvant chemotherapy. Median follow-up was 58 （range, 6-240） months. Five-year OS and DFS rates were 50% and 34%, and LC and LRC rates were 88% and 72%, respectively. On multivariate analysis, independent favorable prognostic factors were younger age, FIGO stage I, small tumor size, previous uterine disease, and no vascular invasion for OS and DFS. FIGO stage was the only favorable factor influencing LRC. Adjuvant local or systemic treatments did not improve the outcomes. Eight patients treated with RT presented a grade 3 acute toxicity, and only one patient with grade 3 late toxicity. Conclusions： In this large population of primary ULMS patients, we found good results in terms of LC and LRC. Nevertheless, OS remains poor, mainly due to the occurrence of distant metastases. An early diagnosis seemed to improve the prognosis of the patients. Adjuvant local or systemic treatments, or more aggressive surgical procedures such as the Wertheim procedure, did not seem to impact the outcome.展开更多
The metastases of uterine leiomyosarcoma to the brain are exceptional and such cases are seldomly reported in the literature. The diagnosis is based on CT and brains MRI findings, in association with a gynecological h...The metastases of uterine leiomyosarcoma to the brain are exceptional and such cases are seldomly reported in the literature. The diagnosis is based on CT and brains MRI findings, in association with a gynecological history of cancer. Their management still stays without guidelines, and the surgical total resection is known to be the only way to influence positively the prognosis and save patient lives. We report a rare case of a 46 years old woman who underwent a hysterectomy and bilateral salpingo-oophorectomy 5 years earlier and presented with the right hemiparesis and whose CT scan and MRI of the brain showed cerebral lesions related to brain location of uterine leiomyosarcoma. The patient underwent surgery for gross total tumor resection, followed by adjuvant radiotherapy, and was doing well after surgery. Three months later she was admitted for recurrences and died after two months of palliative care.展开更多
Uterine leiomyosarcoma is an uncommon malignant neoplasm of smooth muscle origination and is associated with a poor prognosis. We report two cases of uterine leiomyosarcoma that presented with pulmonary metastases.2-d...Uterine leiomyosarcoma is an uncommon malignant neoplasm of smooth muscle origination and is associated with a poor prognosis. We report two cases of uterine leiomyosarcoma that presented with pulmonary metastases.2-deoxy-2-（18F）fluorodeoxyglucose （FDG） positron emission tomography （PET）/computed tomography （CT） was performed to identify the primary carcinoma and found the focus located in the uterus. The follow-up magnetic resonance imaging （MRI） confirmed the diagnosis was uterine leiomyosarcoma.展开更多
Uterine tumors are the most common type of gynecologic neoplasm.Uterine leiomyosarcoma(LMS)is rare,accounting for 2%to 5%of tumors of the uterine body.Uterine LMS develops more often in the muscle tissue layer of the ...Uterine tumors are the most common type of gynecologic neoplasm.Uterine leiomyosarcoma(LMS)is rare,accounting for 2%to 5%of tumors of the uterine body.Uterine LMS develops more often in the muscle tissue layer of the uterine body than in the uterine cervix.The development of gynecologic tumors is often correlated with female hormone secretion;however,the development of uterine LMS is not substantially correlated with hormonal conditions,and the risk factors are not yet known.Radiographic evaluation combined with PET/CT can be useless in the diagnosis and surveillance of uterine LMS.Importantly,a diagnostic biomarker,which distinguishes malignant LMS and benign tumor leiomyoma(LMA)is yet to be established.Accordingly,it is necessary to analyze risk factors associated with uterine LMS in order to establish a method of treatment.LMP2-deficient mice spontaneously develop uterine LMS,with a disease prevalence of~40%by 14 months of age.It is therefore of interest whether human uterine LMS shows a loss of LMP2 expression.We found LMP2 expression is absent in human LMS,but present in human LMA.Therefore,defective LMP2 expression may be one of the risk factors for LMS.LMP2 is potentially a diagnostic biomarker for uterine LMS,and gene therapy with LMP2-encording DNA may be a new therapeutic approach.展开更多
BACKGROUND Leiomyosarcoma is a subtype of soft tissue sarcoma with adverse outcomes.Leiomyosarcoma accounts for nearly 70%of all uterine sarcomas and is responsible for a considerable proportion of deaths because of u...BACKGROUND Leiomyosarcoma is a subtype of soft tissue sarcoma with adverse outcomes.Leiomyosarcoma accounts for nearly 70%of all uterine sarcomas and is responsible for a considerable proportion of deaths because of uterine cancer.Clinical characteristics and relevant diagnosis of pelvic leiomyosarcoma should be further explored.AIM To identify the outcome and relevant perioperative evaluation of patients with pelvic leiomyosarcoma.METHODS The Kaplan-Meier method was used to determine progression-free survival and overall survival rates.Factors predictive of outcomes were identified using univariate and multivariate Cox proportional hazards models.RESULTS Fifty-one patients with pelvic leiomyosarcoma were enrolled and divided into two groups including uterine leiomyosarcoma and non-uterine leiomyosarcoma.Overall,28.6%and 45.5%of uterine leiomyosarcoma and non-uterine leiomyosarcoma patients,respectively,had elevated carbohydrate antigen 125 levels,whereas 45.7%and 68.8%,respectively,underwent ultrasonography.Although 68.8%of uterine leiomyosarcoma patients were initially diagnosed with hysteromyoma,72.7%of non-uterine leiomyosarcoma patients had pelvic and abdominal masses.Moreover,93.3%of the recurrent lesions were detected using ultrasonography.Patients with International Federation of Gynaecology and Obstetrics(FIGO)stages III–IV disease had poorer progression-free survival values than those with FIGO stages I–II(P=0.027)disease.FIGO stage was significantly associated with poor progression-free survival in the univariate(hazard ratio=2.64,P=0.03)and multivariate(hazard ratio=2.49,P=0.048)analyses.CONCLUSION Serum tumour biomarkers cannot be used for pelvic leiomyosarcoma diagnosis.FIGO stage is critical to predict the outcome of uterine leiomyosarcoma.Ultrasonography is more reliable for postoperative follow-up than preoperative diagnosis.展开更多
文摘Objective： Primary uterine leiomyosarcomas （ULMS） are rare, and the optimal treatment is controversial. We aimed to assess the outcome and prognostic factors in a multicenter population of women treated for primary ULMS. Methods： We retrospectively collected data of 110 women treated in 19 institutions of the Rare Cancer Network （RCN）. Inclusion criteria consisted of a pathology report confirming the diagnosis of ULMS, aged 18-80 years, complete International Federation of Gynecology and Obstetrics （FIGO） stage information, complete information on treatment, and a minimum follow-up of 6 months. Local control （LC） and locoregional control （LRC）, overall survival （OS） and disease-free survival （DFS） rates were computed using the Kaplan-Meier method. Univariate analysis was implemented using the log rank test, and multivariate analysis using the Cox model. Results： All patients underwent surgery. Seventy-five patients （68%） received adjuvant radiotherapy （RT）, including brachytherapy in 18 （I 6%）. Seventeen patients （15 %） received adjuvant chemotherapy. Median follow-up was 58 （range, 6-240） months. Five-year OS and DFS rates were 50% and 34%, and LC and LRC rates were 88% and 72%, respectively. On multivariate analysis, independent favorable prognostic factors were younger age, FIGO stage I, small tumor size, previous uterine disease, and no vascular invasion for OS and DFS. FIGO stage was the only favorable factor influencing LRC. Adjuvant local or systemic treatments did not improve the outcomes. Eight patients treated with RT presented a grade 3 acute toxicity, and only one patient with grade 3 late toxicity. Conclusions： In this large population of primary ULMS patients, we found good results in terms of LC and LRC. Nevertheless, OS remains poor, mainly due to the occurrence of distant metastases. An early diagnosis seemed to improve the prognosis of the patients. Adjuvant local or systemic treatments, or more aggressive surgical procedures such as the Wertheim procedure, did not seem to impact the outcome.
文摘The metastases of uterine leiomyosarcoma to the brain are exceptional and such cases are seldomly reported in the literature. The diagnosis is based on CT and brains MRI findings, in association with a gynecological history of cancer. Their management still stays without guidelines, and the surgical total resection is known to be the only way to influence positively the prognosis and save patient lives. We report a rare case of a 46 years old woman who underwent a hysterectomy and bilateral salpingo-oophorectomy 5 years earlier and presented with the right hemiparesis and whose CT scan and MRI of the brain showed cerebral lesions related to brain location of uterine leiomyosarcoma. The patient underwent surgery for gross total tumor resection, followed by adjuvant radiotherapy, and was doing well after surgery. Three months later she was admitted for recurrences and died after two months of palliative care.
文摘Uterine leiomyosarcoma is an uncommon malignant neoplasm of smooth muscle origination and is associated with a poor prognosis. We report two cases of uterine leiomyosarcoma that presented with pulmonary metastases.2-deoxy-2-（18F）fluorodeoxyglucose （FDG） positron emission tomography （PET）/computed tomography （CT） was performed to identify the primary carcinoma and found the focus located in the uterus. The follow-up magnetic resonance imaging （MRI） confirmed the diagnosis was uterine leiomyosarcoma.
基金supported in part by grants from the Ministry of Education,Culture,Science and Technology,and The Foundation of Osaka Cancer Research,The Ichiro Kanehara Foundation of the Promotion of Medical Science and Medical Care,Foundation for Promotion of Cancer Research,Kanzawa Medical Research Foundation,The Shinshu Medical Foundation,and Takeda Science Foundation.
文摘Uterine tumors are the most common type of gynecologic neoplasm.Uterine leiomyosarcoma(LMS)is rare,accounting for 2%to 5%of tumors of the uterine body.Uterine LMS develops more often in the muscle tissue layer of the uterine body than in the uterine cervix.The development of gynecologic tumors is often correlated with female hormone secretion;however,the development of uterine LMS is not substantially correlated with hormonal conditions,and the risk factors are not yet known.Radiographic evaluation combined with PET/CT can be useless in the diagnosis and surveillance of uterine LMS.Importantly,a diagnostic biomarker,which distinguishes malignant LMS and benign tumor leiomyoma(LMA)is yet to be established.Accordingly,it is necessary to analyze risk factors associated with uterine LMS in order to establish a method of treatment.LMP2-deficient mice spontaneously develop uterine LMS,with a disease prevalence of~40%by 14 months of age.It is therefore of interest whether human uterine LMS shows a loss of LMP2 expression.We found LMP2 expression is absent in human LMS,but present in human LMA.Therefore,defective LMP2 expression may be one of the risk factors for LMS.LMP2 is potentially a diagnostic biomarker for uterine LMS,and gene therapy with LMP2-encording DNA may be a new therapeutic approach.
基金Supported by National Natural Science Foundation of China,No81501530Scientific Research Projects of Hubei Health Commission,No. WJ2019M130Scientific Research Fund of Tongji Hospital,No. 2018B02
文摘BACKGROUND Leiomyosarcoma is a subtype of soft tissue sarcoma with adverse outcomes.Leiomyosarcoma accounts for nearly 70%of all uterine sarcomas and is responsible for a considerable proportion of deaths because of uterine cancer.Clinical characteristics and relevant diagnosis of pelvic leiomyosarcoma should be further explored.AIM To identify the outcome and relevant perioperative evaluation of patients with pelvic leiomyosarcoma.METHODS The Kaplan-Meier method was used to determine progression-free survival and overall survival rates.Factors predictive of outcomes were identified using univariate and multivariate Cox proportional hazards models.RESULTS Fifty-one patients with pelvic leiomyosarcoma were enrolled and divided into two groups including uterine leiomyosarcoma and non-uterine leiomyosarcoma.Overall,28.6%and 45.5%of uterine leiomyosarcoma and non-uterine leiomyosarcoma patients,respectively,had elevated carbohydrate antigen 125 levels,whereas 45.7%and 68.8%,respectively,underwent ultrasonography.Although 68.8%of uterine leiomyosarcoma patients were initially diagnosed with hysteromyoma,72.7%of non-uterine leiomyosarcoma patients had pelvic and abdominal masses.Moreover,93.3%of the recurrent lesions were detected using ultrasonography.Patients with International Federation of Gynaecology and Obstetrics(FIGO)stages III–IV disease had poorer progression-free survival values than those with FIGO stages I–II(P=0.027)disease.FIGO stage was significantly associated with poor progression-free survival in the univariate(hazard ratio=2.64,P=0.03)and multivariate(hazard ratio=2.49,P=0.048)analyses.CONCLUSION Serum tumour biomarkers cannot be used for pelvic leiomyosarcoma diagnosis.FIGO stage is critical to predict the outcome of uterine leiomyosarcoma.Ultrasonography is more reliable for postoperative follow-up than preoperative diagnosis.
