scholarly journals Local and Distant Recurrence in Resected Sacral Chordomas: A Systematic Review and Pooled Cohort Analysis

2018 ◽  
Vol 9 (2) ◽  
pp. 191-201 ◽  
Author(s):  
Daniel Kerekes ◽  
C. Rory Goodwin ◽  
A. Karim Ahmed ◽  
Jorrit-Jan Verlaan ◽  
Chetan Bettegowda ◽  
...  

Study Design: Systematic review. Objectives: Sacral chordomas are rare, primary tumors of the spine, best treated with en bloc resection. The purpose of this study was to assess the literature for resected sacral chordoma and to quantify the prevalence of, risk factors for, and treatment outcomes of local and distant recurrence therein. Methods: We searched 5 online databases from January 1980 to May 2016 to find articles that report survival, recurrence outcomes, and/or prognostic factors for the resected sacral chordoma patient population. Characteristics and clinical outcomes of the pooled cohort are reported. Fisher exact tests, unpaired t tests, and one-way analysis of variance were used to investigate patient- and treatment-associated prognostic factors for local and distant recurrence. Survival analyses were performed for time to local recurrence and death. The protocol’s PROSPERO ID is CRD42015024384. Results: Fifty-seven studies, with 1235 unique sacral chordoma patients, were included in this review. Local and distant recurrence occurred in 42.6% and 22.4% of patients with adequate follow-up, respectively. Kaplan-Meier overall median survival for patients with and without recurrence were 98 and 209 months after surgery, respectively. Wide surgical margin was associated with a lower rate of local recurrence; and wide surgical margin, female sex, and patient age ≥65 years were associated with lower rates of distant recurrence. Conclusions: While surgical margin remains the most significant prognostic factor for local and distant recurrence, combined surgical approach may be associated with local recurrence. Male sex and age <65 years may be associated with distant recurrence. Patients with risk factors for recurrence should undergo close monitoring to maximize survival.

Blood ◽  
2018 ◽  
Vol 132 (Supplement 1) ◽  
pp. 3806-3806 ◽  
Author(s):  
Elissa Engel ◽  
Manuela Albisetti ◽  
Leonardo R. Brandao ◽  
Ernest Amankwah ◽  
Anthony Nguyen ◽  
...  

Abstract BACKGROUND: Post-thrombotic syndrome (PTS) is the most common long-term complication in pediatric deep venous thrombosis (DVT), affecting approximately 25% of children with an extremity DVT. PTS leads to a high physical, psychological and financial burden in affected patients. Although several risk factors have been associated with the development of pediatric PTS, few of them have been validated in the pediatric literature. A better understanding of the prognostic factors leading to PTS is a vital step for early identification of those children at greatest risk in order to develop risk-stratified interventions aimed at preventing this complication. AIM: To perform a systematic review and meta-analysis of available published evidence from the pediatric literature on prognostic factors for pediatric PTS. METHODS: A systematic search of MEDLINE, EMBASE, and the Cochrane Library from 1960 to December 2017 was performed. MeSH terms and search strategy employed were as follows: "postthrombotic syndrome" OR "postphlebitic syndrome" AND "all child 0-18 years" AND "young adult 19-24 years". A study was eligible for inclusion if it evaluated the development of PTS in pediatric patients (<21 years of age) with a confirmed extremity DVT and reported on at least one prognostic factor for the development of PTS. Single case reports, narrative reviews, and commentaries were excluded. Studies assessing the efficacy/safety of thrombolysis, and studies including patients >21 years of age with outcomes not reported by age group, were also excluded. Two reviewers independently screened all studies and extracted the data of interest. Data were analyzed using STATA v.15 statistical software. Meta-analyses were conducted for risk factors reported in at least three studies. Summary odds ratios (ORs) and 95% confidence intervals (CI) were calculated from the effect estimates from the individual studies using a random effects model. Statistical heterogeneity was quantified by I2 statistic. RESULTS: A total of 12 studies met the final inclusion criteria (Figure 1), nine cohort studies, two cross-sectional studies, and one case-control study. These studies reported a total of 1,160 patients with venous thromboembolism (VTE), of whom 938 (81%) were assessed for PTS (Table 1). Median age across studies ranged from 0.02 - 15.5 years. VTE was considered provoked in nearly 80% of patients. The most common reported risk factor for VTE was the presence of a central venous catheter (CVC, 54%) followed by congenital heart disease (26%). PTS was diagnosed in 46% (n=434) of patients with an extremity DVT. The median time from DVT diagnosis to PTS diagnosis ranged from 12 to 33 months across studies. Among studies reporting this information, mild PTS was most frequently diagnosed, followed by moderate and severe PTS (35%, 5% and 0.6% of patients respectively). Most common prognostic factors associated with PTS in individual studies included patient characteristics: age and gender; and DVT characteristics: recurrent DVT, symptomatic DVT, DVT degree of occlusion, and time between DVT diagnosis and PTS assessment. Three studies investigated the association of elevated factor VIII and d-dimer levels with PTS. Elevated levels of these biomarkers were found to be associated with development of adverse VTE outcomes in one study but this finding was not confirmed in the other studies. Meta-analysis of reported prognostic factors identified the presence of a CVC and occlusive DVT as significant risk factors for the development of pediatric PTS (OR= 1.8, 95%CI=1.08-2.98, and OR=1.89, 95%CI=1.04-3.46 respectively; Figure 2). CONCLUSION: Among 12 studies evaluating prognostic factors for PTS in children and meeting criteria for this meta-analysis, CVC-related DVT and complete occlusion were associated with pediatric PTS. Overall, high-quality evidence on pediatric PTS is lacking. Collaborative prospective cohort studies and trials that use validated pediatric PTS measures and standardized prognostic factor definitions are needed to better understand the risk factors associated with PTS. Disclosures No relevant conflicts of interest to declare.


2021 ◽  
Author(s):  
Tessel Meike van Rossen ◽  
Rogier E. Ooijevaar ◽  
Christina M.J.E. Vandenbroucke-Grauls ◽  
Olaf M. Dekkers ◽  
Ed J. Kuijper ◽  
...  

Background Clostridioides difficile infection (CDI), its subsequent recurrences (rCDI), and severe CDI (sCDI) provide a significant burden for both patients and the healthcare system. Treatment consists of oral antibiotics. Fidaxomicin, bezlotoxumab and fecal microbiota transplantion (FMT) reduce the number of recurrences compared to vancomycin, but are more costly. Identifying patients diagnosed with initial CDI who are at increased risk of developing sCDI/rCDI could lead to more cost-effective therapeutic choices. Objectives In this systematic review we aimed to identify clinical prognostic factors associated with an increased risk of developing sCDI or rCDI. Methods PubMed, Embase, Emcare, Web of Science and COCHRANE Library databases were searched from database inception through March, 2021. Study selection was performed by two independent reviewers on the basis of predefined selection criteria; conflicts were resolved by consensus. Cohort and case-control studies providing an analysis of clinical or laboratory data to predict sCDI/rCDI in patients ≥18 years diagnosed with CDI, were included. Risk of bias was assessed with the Quality in Prognostic Research (QUIPS) tool and the quality of evidence by the Grading of Recommendations Assessment, Development and Evaluation (GRADE) tool, modified for prognostic studies. Overview tables of prognostic factors were constructed to assess the number of studies and the respective direction of an association (positive, negative, or no association). Results and conclusions 136 studies were included for final analysis. Higher age and the presence of multiple comorbidities were prognostic factors for sCDI. Identified risk factors for rCDI were higher age, healthcare-associated CDI, prior hospitalization, PPIs started during/after CDI diagnosis and previous rCDI. Some variables that were found as risk factors for sCDI/rCDI in previous reviews were not confirmed in the current review, which can be attributed to differences in methodology. Risk stratification for sCDI/rCDI may contribute to a more personalized and optimal treatment for patients with CDI.


2006 ◽  
Vol 24 (18_suppl) ◽  
pp. 9572-9572
Author(s):  
A. Kawai ◽  
R. Nakayama ◽  
A. Matsumine ◽  
S. Matsumoto ◽  
T. Ueda ◽  
...  

9572 Background: Clear cell sarcoma (CCS) of tendons and aponeuroses is a rare malignant tumor that occurs most commonly in the extremities of young adults. Because of the rarity of the disease (1% of all soft tissue sarcomas), most reports about CCS have dealt with a few cases during a long time period. We conducted a multi-institutional study of CCS to clarify the clinical findings and prognostic factors of CCS treated during the era of modern multidisciplinary treatment. Methods: The records of 75 consecutive patients with histologically confirmed CCS treated at the JMOG affiliated institutions between 1980 and 2004 (follow-up; average 44 months) were reviewed. There were 41 men and 34 women with an average age of 36 years (range, 10–71 years). 65 tumors were in the extremities (foot 22, hand 13, thigh 9, others 21) and 10 were in the trunk. The tumor size ranged from 1 to 11 cm (mean, 4 cm). 52 patients presented with localized disease (M0) and 23 with metastatic disease (M1). Results: Surgical excision of the primary tumor was performed in 71 patients (limb-sparing surgery; 56, amputation; 15). Microscopic surgical margin was negative in 60 and positive in 11. 56 patients received chemotherapy (30 with measurable disease) and 17 had radiotherapy. Local recurrence occurred in 16 patients and 48 patients developed metastasis. The 5-year overall survival rate was 47% (M0; 55%, M1; 20%). On univariate analysis, sex (p=0.02), size (p=0.001), depth (p=0.002), TNM stage (p=0.001), IRS group (p=0.001) and surgical margin (p=0.04) were prognostic factors. On multivariate analysis, size (p=0.02) remained to be a significant prognostic factor. Objective response to chemotherapy was observed in 8 (27%) patients. In the group of M0 patients, those who received adjuvant chemotherapy had better prognosis (5-year survival, 65%) than those without chemotherapy (5-year survival, 23%)(p=0.03). Conclusions: The results support the contention that early diagnosis and initial excision with negative surgical margin are essential for favorable outcome of CCS. The role of chemotherapy for CCS should be further investigated. No significant financial relationships to disclose.


2017 ◽  
Vol 27 (7) ◽  
pp. 1446-1454 ◽  
Author(s):  
Ozan Cem Guler ◽  
Sezin Yuce Sari ◽  
Sumerya Duru Birgi ◽  
Melis Gultekin ◽  
Ferah Yildiz ◽  
...  

ObjectiveThe aim of the study was to investigate the prognostic factors for survival and treatment-related toxicities in older (≥65 years) cervical cancer patients treated with definitive chemoradiotherapy. In addition, we sought to compare the outcomes between the older elderly (≥75 years) and their younger old counterparts (age, 65–74 years).Materials and MethodsWe retrospectively reviewed medical records from 269 biopsy-proven nonmetastatic cervical cancer patients treated with external radiotherapy and intracavitary brachytherapy at the departments of radiation oncology in 2 different universities. The prognostic factors for survival, local control, and distant metastasis (DM) were analyzed.ResultsThe median follow-up time was 38.8 months (range, 1.5–175.5 months) for the entire cohort and 70.0 months (range, 6.1–175.7 months) for survivors. The 2- and 5-year overall survival (OS), disease-free survival (DFS), and cause-specific survival rates were 66% and 42%, 63% and 39%, and 72% and 55%, respectively. Patients 75 years or older showed significantly worse OS compared with patients aged 65 to 74 years but showed no significant difference in DFS. The 2- and 5-year local control rates were 86% and 71%, respectively. The incidences of DMs at 2 and 5 years were 22% and 30%, respectively. In multivariate analysis, vaginal infiltration and lymph node metastasis were predictive of OS, DFS, local recurrence, and DM. Concomitant chemotherapy was predictive of OS, DFS, and local recurrence, and larger tumor (>4 cm) was a significant prognostic factor for local recurrence. None of the patients had toxicity that necessitated the discontinuation of radiotherapy. All patients were evaluable for acute toxicity, and no grade higher than 3 adverse events occurred during external beam radiation therapy or brachytherapy.ConclusionsAlthough age limited the delivery of aggressive treatment, concurrent chemoradiotherapy in elderly patients associated with improved outcomes similar as in younger counterparts without increasing serious acute and late toxicities.


Sarcoma ◽  
2019 ◽  
Vol 2019 ◽  
pp. 1-8 ◽  
Author(s):  
Hjalmar Teurneau ◽  
Jacob Engellau ◽  
Iman Ghanei ◽  
Fredrik Vult von Steyern ◽  
Emelie Styring

Background. Myxofibrosarcoma (MFS) is one of the more common types of soft-tissue sarcoma (STS) in patients over 60 years of age. Local recurrence (LR) rates have been reported to be higher compared to other STS types. Patients and Methods. Using a population-based series from the southern Sweden health care region, 56 consecutive patients with MFS and localized disease at diagnosis were analyzed with respect to LR and distant metastases after surgery ± adjuvant treatment. Results. The overall local recurrence (n = 15) and metastasis (n = 13) rates were 27% and 21%, respectively; 6 patients had both. Surgical margin was the only statistically significant prognostic factor for LR. Patients operated with a marginal margin had an HR of 4.5 (CI 1.3–15.1, p=0.02) and those operated with an intralesional margin 9.4 (CI 2.0–43.5, p=0.004) compared to those operated with a wide surgical margin. There was no difference in the LR rate depending on radiotherapy or not, although the latter group had smaller and more superficial tumors. 23 patients received radiotherapy, 9 of whom developed LR, all within the irradiated field. A tumor size >5 cm and intralesional surgical margin were shown to be risk factors for distant metastases. Conclusions. The rate of LR for patients with myxofibrosarcoma was high. The impact of RT on local tumor control was unclear. The surgical margin was important for both local and distant tumor control. Large tumor size was a risk factor for distant metastasis.


2000 ◽  
Vol 18 (10) ◽  
pp. 2087-2094 ◽  
Author(s):  
Jonathan J. Lewis ◽  
Cristina R. Antonescu ◽  
Denis H. Y. Leung ◽  
David Blumberg ◽  
John H. Healey ◽  
...  

PURPOSE: Synovial sarcoma is a high-grade tumor that is associated with poor prognosis. Previous studies analyzing prognostic factors are limited because of inclusion of heterogeneous cohorts of patients with nonextremity and recurrent tumors. The objective of this study was to determine independent prognostic factors of primary synovial sarcoma localized to the extremity. PATIENTS AND METHODS: Between July 1, 1982, and June 30, 1996, 112 patients underwent surgical resection for cure at our institution and then were followed-up prospectively. Clinical and pathologic factors examined for prognostic value included age, sex, tumor site and location, depth, size, microscopic status of surgical margins, invasion of bone or neurovascular structures, and monophasic or biphasic histology. The end points analyzed were the time to first local recurrence that was not preceded by a distant recurrence, time to any distant recurrence, and time to disease-related mortality. These end points were modeled using the method of Kaplan and Meier and analyzed by the log-rank test and Cox regression. RESULTS: The median duration of follow-up among survivors in this cohort of 112 patients was 72 months. The 5-year local-recurrence, distant-recurrence, and mortality rates were 12%, 39%, and 25%, respectively. Tumor size ≥ 5 cm (P = .001; relative risk [RR] = 2.7; 95% confidence interval [CI], 1.5 to 5.2) and the presence of bone or neurovascular invasion (P = .04; RR = 2.3; 95% CI, 1.0 to 5.3) were independent adverse predictors of distant recurrence. Tumor size ≥ 5 cm (P = .003; RR = 2.3; 95% CI, 1.4 to 6.3) and the presence of bone or neurovascular invasion (P = .03; RR = 2.7; 95% CI, 1.0 to 6.5) were also independent adverse predictors of mortality. CONCLUSION: The natural history of primary synovial sarcoma of the extremity is related to tumor size and invasion of bone and neurovascular structures.


1996 ◽  
Vol 14 (5) ◽  
pp. 1679-1689 ◽  
Author(s):  
P W Pisters ◽  
D H Leung ◽  
J Woodruff ◽  
W Shi ◽  
M F Brennan

PURPOSE To identify specific independent adverse clinicopathologic factors for event-free survival in a cohort of consecutively treated patients with extremity soft tissue sarcomas. PATIENTS AND METHODS Prospectively collected data from a population of 1,041 adult patients with localized (American Joint Committee on Cancer [AJCC] stage IA to IIIB) extremity soft tissue sarcomas were analyzed. Patients were treated at a single institution between 1982 and 1994. Patient, tumor, and pathologic factors were analyzed by univariate and multivariate techniques to identify independent prognostic factors for the end points of local recurrence, distant recurrence, disease-specific survival, and post-metastasis survival. RESULTS The 5-year survival rate for this cohort of patients was 76%, with a median follow-up time of 3.95 years. Significant independent adverse prognostic factors for local recurrence were age greater than 50 years, recurrent disease at presentation, microscopically positive surgical margins, and the histologic subtypes fibrosarcoma and malignant peripheral-nerve tumor. For distant recurrence, intermediate tumor size, high histologic grade, deep location, recurrent disease at presentation, leiomyosarcoma, and nonliposarcoma histology were independent adverse prognostic factors. For disease-specific survival, large tumor size, high grade, deep location, recurrent disease at presentation, the histologic subtypes leiomyosarcoma and malignant peripheral-nerve tumor, microscopically positive surgical margins, and lower extremity site were adverse factors. For post-metastasis survival, only large tumor size ( > 10 cm) was an adverse prognostic factor. CONCLUSION The independent adverse prognostic factors for distant recurrence and disease specific survival differ from those identified for subsequent local recurrence. Patients with microscopically positive surgical margins or patients who present with locally recurrent disease are at increased risk for subsequent local recurrence and tumor-related mortality. Specific histopathologic subtypes are associated with increased risks for local failure and tumor-related mortality.


Author(s):  
Arvid von Konow ◽  
Iman Ghanei ◽  
Emelie Styring ◽  
Fredrik Vult von Steyern

Abstract Background Approximately 80% of soft tissue sarcoma (STS) recurrences, local and metastatic disease, are diagnosed within the first 3 years after primary diagnosis and treatment. Recurrences, however, can present after a longer period of remission. Our goal was to identify factors that may predict the risk of late recurrence. Methods We identified 677 patients with STS of the extremities and trunk wall from a population-based sarcoma register. Of these, 377 patients were alive and event-free at 3 years and were included for analysis of possible risk factors for late recurrence. Results Fifty-five of 377 (15%) patients developed late recurrence: 23 local recurrence, 21 metastasis, and 11 both manifestations. With R0 wide surgical margin as reference, R0 marginal (hazard ratio [HR] 2.6; p = 0.02) and R1 (HR 5.0; p = 0.005) margins were risk factors for late local recurrence. Malignancy grade (HR 8.3; p = 0.04) and R0 marginal surgical margin (HR 2.3; p = 0.04) were risk factors for late metastasis. We could not find a statistically significant correlation of late recurrence with many of the generally known risk factors for local recurrence and metastasis in STS. Outcome after treatment of late recurrences was better compared with outcome after treatment of early events. Conclusions Late recurrences, albeit relatively rare, do occur. Outcome after treatment was good compared with outcome after early events. Long surveillance of all patients with high-grade STS, especially if R0 wide surgical margin is not achieved in the primary treatment, appear to be well justified.


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