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European Journal of Cancer Prevention :... May 2024This systematic review aims to synthesize the available literature to determine the association between birthweight and the risk of nonneurological childhood cancers.
OBJECTIVES
This systematic review aims to synthesize the available literature to determine the association between birthweight and the risk of nonneurological childhood cancers.
METHODS
We conducted a systematic search of PubMed, Web of Science, and Scopus databases up to May 2023 to identify observational studies. Heterogeneity between studies was evaluated using the I2 statistics. Publication bias was assessed using Begg and Egger tests. We calculated the odds ratio (OR) or risk ratio (RR) with a 95% confidence interval (CI) using a random-effects model.
RESULTS
Of 11 034 studies retrieved from the search, 56 studies (including 10 568 091 participants) were eligible. The ORs (95% CI) of low (<2500 g) versus normal birthweight (2500-4000 g) and childhood cancers were as follows: leukemia, 0.92 (0.77-1.11); acute lymphoblastic leukemia, 0.82 (0.72-0.94); acute myeloid leukemia, 0.98 (0.77-1.24); lymphoma, 0.99 (0.47-2.10); Hodgkin, 0.79 (0.61-1.03); non-Hodgkin, 0.85 (0.60-1.20); neuroblastoma, 1.34 (1.14-1.58); retinoblastoma, 0.95 (0.68-1.32); rhabdomyosarcoma, 0.86 (0.61-1.20); embryonal, 0.97 (0.66-1.43); alveolar, 1.92 (0.43-8.51); and Wilms tumor, 1.01 (0.83-1.24). The ORs (95% CI) of high (>4000 g) versus normal birthweight and childhood cancers were as follows: leukemia, 1.30 (1.18-1.42); acute lymphoblastic leukemia, 1.27 (1.16-1.39); acute myeloid leukemia, 1.13 (0.98-1.30); lymphoma, 1.69 (0.72-3.94); Hodgkin, 1.22 (1.02-1.46); non-Hodgkin, 1.22 (0.80-1.86); neuroblastoma, 1.20 (1.02-1.41); retinoblastoma, 1.17 (0.93-1.48); rhabdomyosarcoma, 1.07 (0.90-1.27); embryonal, 1.22 (1.00-1.49); alveolar, 1.02 (0.46-2.27); and Wilms tumor, 1.49 (1.34-1.67).
CONCLUSION
This meta-analysis identified high birth weight as a potential risk factor for some childhood cancers, while low birth weight might be protective against a few.
PubMed: 38837193
DOI: 10.1097/CEJ.0000000000000894 -
Pediatric Blood & Cancer Nov 2023Several cancer predisposition syndromes (CPS) are reported to predispose to rhabdomyosarcoma, most frequently in children with embryonal rhabdomyosarcoma. There are... (Review)
Review
Several cancer predisposition syndromes (CPS) are reported to predispose to rhabdomyosarcoma, most frequently in children with embryonal rhabdomyosarcoma. There are lingering questions over the role of CPS in individuals with alveolar rhabdomyosarcoma (ARMS), which are frequently driven by FOXO1 fusion oncoproteins. We conducted a systematic review to identify patients with FOXO1 fusion-positive ARMS (FP-ARMS) who underwent germline DNA sequencing. We estimated the prevalence of pathogenic/likely pathogenic (P/LP) variants in cancer predisposing genes (CPGs) and of CPSs. We included 19 publications reporting on 191 patients with FP-ARMS. P/LP variants in CPGs were identified in 26/191 (13.6%) patients, nine (4.9%) of which were associated with a CPS diagnosis. Evidence for causal associations between CPSs and FP-ARMS could not be assessed with available data from this review. Only one patient was affected with a CPS known to predispose to rhabdomyosarcoma, Li-Fraumeni syndrome. Typical CPS associations with rhabdomyosarcoma are rare, but not nonexistent, in patients with FP-ARMS. FOXO1 fusion status, alone, is insufficient for clinicians to rely on to distinguish between patients with/without CPS.
Topics: Child; Humans; Prevalence; Rhabdomyosarcoma; Rhabdomyosarcoma, Alveolar; Genotype; Germ Cells; Forkhead Box Protein O1
PubMed: 37638828
DOI: 10.1002/pbc.30651 -
Journal of the Korean Association of... Apr 2023This systematic review aimed to analyze the clinicopathological profile and relevant prognostic factors of head and neck rhabdomyosarcoma in pediatric patients. The... (Review)
Review
This systematic review aimed to analyze the clinicopathological profile and relevant prognostic factors of head and neck rhabdomyosarcoma in pediatric patients. The search was carried out in the electronic search portals PubMed, Lilacs, Embase, Scopus, and Web of Science. The search yielded studies that were then analyzed regarding study topic, data extraction, and risk of bias using the STROBE (Strengthening the Reporting of Observational Studies) guidelines. Finally, three studies were included for qualitative analysis. Most of the cases involved embryonic and alveolar rhabdomyosarcoma. Expression of MYOD1 was highly correlated with diagnosis of spindle cell/sclerosing rhabdomyosarcoma, which appears to have a poor prognosis in children. Furthermore, tumor size <5 cm and absence of metastasis accompanied by complete resection and administration of adjuvant therapies such as chemotherapy and radiotherapy favored a better prognosis.
PubMed: 37114443
DOI: 10.5125/jkaoms.2023.49.2.61 -
Oral Surgery, Oral Medicine, Oral... Sep 2022This systematic review aimed to identify the molecular alterations of head and neck rhabdomyosarcomas (HNRMS) and their prognostic values. (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
This systematic review aimed to identify the molecular alterations of head and neck rhabdomyosarcomas (HNRMS) and their prognostic values.
STUDY DESIGN
An electronic search was performed using PubMed, Embase, Scopus, and Web of Science with a designed search strategy. Inclusion criteria comprised cases of primary HNRMS with an established histopathological diagnosis and molecular analysis. Forty-nine studies were included and were appraised for methodological quality using the Joanna Briggs Institute Critical Appraisal tools. Five studies were selected for meta-analysis.
RESULTS
HNRMS predominantly affects pediatric patients (44.4%), and the parameningeal region (57.7%) is the most common location. The alveolar variant (43.2%) predominates over the embryonal and spindle cell/sclerosing types, followed by the epithelioid and pleomorphic variants. PAX-FOXO1 fusion was observed in 103 cases of alveolar RMS (79.8%). MYOD1 mutation was found in 39 cases of sclerosing/spindle cell RMS (53.4%). FUS/EWSR1-TFCP2 gene fusions were identified in 21 cases of RMS with epithelioid and spindle cell morphologies (95.5%). The 5-year overall survival rate of patients was 61.3%, and MYOD1 mutation correlated with significantly higher mortality.
CONCLUSION
The genotypic profile of histologic variants of HNRMS is widely variable, and MYOD1 mutation could be a potential prognostic factor, but more studies are required to establish this.
Topics: Child; DNA-Binding Proteins; Humans; Mutation; Rhabdomyosarcoma; Transcription Factors
PubMed: 35840496
DOI: 10.1016/j.oooo.2021.12.128 -
Cancer Radiotherapie : Journal de La... Dec 2020Alveolar rhabdomyosarcoma (ARMS) represents the most common childhood soft tissue sarcoma, but they are rarely seen among adults. Most of the protocols for adults are...
Alveolar rhabdomyosarcoma (ARMS) represents the most common childhood soft tissue sarcoma, but they are rarely seen among adults. Most of the protocols for adults are adapted from pediatric protocols. Here we report a case of a 53-year-old woman diagnosed with a nasal alveolar rhabdomyosarcoma, stage IV at diagnosis, treated by chemotherapy (a regimen inspired from the pediatric protocole pEpSSG RMS 2005) which led to partial response followed by chemo-radiotherapy. We performed a systematic review of adult head and neck ARMS and found 29 cases. Primary chemotherapy with different protocols (VAC, VAI or VIE) should be done followed by surgery and/or external beam radiotherapy (preferably with IMRT). EBRT seems beneficial to every ARMS with a dose around 50Gy in a conventional fractionation, eventually completed with a boost on residual tumor. The target volume must be defined on pre-chemotherapy imaging. Brachytherapy and proton therapy are under evaluation.
Topics: Antineoplastic Combined Chemotherapy Protocols; Chemoradiotherapy; Combined Modality Therapy; Dactinomycin; Doxorubicin; Female; Humans; Ifosfamide; Middle Aged; Nose Neoplasms; Radiotherapy, Intensity-Modulated; Rhabdomyosarcoma, Alveolar; Vincristine
PubMed: 33172776
DOI: 10.1016/j.canrad.2020.03.015 -
A&A Practice Jan 2020Cytoreductive surgery (CS) and hyperthermic intraperitoneal chemotherapy (HIPEC) present a challenging task for anesthesia providers. Anesthesia management may be...
Anesthetic Management During Pediatric Cytoreductive Surgery and Hyperthermic Intraperitoneal Chemotherapy With Cisplatin in a Small Child: A Case Report and Systematic Literature Review.
Cytoreductive surgery (CS) and hyperthermic intraperitoneal chemotherapy (HIPEC) present a challenging task for anesthesia providers. Anesthesia management may be complicated by hyperthermia, fluid shifts, and distinct inflammatory response. Only a few reports dealing with the anesthesia management of pediatric CS and HIPEC have been published. We report a case of a 2-year-old child with a relapse of an alveolar rhabdomyosarcoma of the uterus and peritoneal carcinomatosis treated with CS and HIPEC. For children, careful temperature measurement, intraoperative prevention of hyperthermia, and sufficient volume management are important, as well as postoperative pediatric intensive care with experience CS and HIPEC patients.
Topics: Anesthetics; Body Temperature; Child, Preschool; Cisplatin; Combined Modality Therapy; Critical Care; Cytoreduction Surgical Procedures; Fatal Outcome; Female; Humans; Hyperthermia, Induced; Neoplasm Recurrence, Local; Peritoneal Neoplasms; Postoperative Care; Rhabdomyosarcoma, Alveolar; Uterine Neoplasms
PubMed: 31651415
DOI: 10.1213/XAA.0000000000001122 -
BioMed Research International 2018Previous literatures have investigated the change of miR-20a expression level in the progression of multiple cancers and its influence on patients' survival outcome, but... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Previous literatures have investigated the change of miR-20a expression level in the progression of multiple cancers and its influence on patients' survival outcome, but results of now-available evidence are inconsistent.
OBJECTIVE
To elucidate the prognostic value of circulating and tissue-based miR-20a for patients with various cancers.
METHODS
A systematic search and review of eligible publications were carried out in three electronic databases including the Cochrane Library, PubMed, and Embase, and the methodological quality of included studies was assessed according to Newcastle-Ottawa Scale (NOS). Hazard ratios (HRs) and corresponding 95% confidence intervals (CIs) for overall survival (OS), recurrence-free survival (RFS), disease-free survival (DFS), and progressive-free survival (PFS) of each study were pooled using a random effect model.
RESULTS
In total, 24 studies involving 4186 samples of multiple cancers published in 20 articles were included in the statistical analysis. As for circulating miR-20a, five kinds of cancers containing gastric cancer, lymphoma, glioblastoma, prostate cancer, and non-small-cell lung cancer reported upregulated level in patients compared with normal healthy control, and overexpressed circulating miR-20a could confer an unfavorable factor for OS (HR = 1.71, 95% CIs: 1.43 -2.04, < 0.01) and DFS (HR = 1.90, 95% CIs: 1.45-2.49, < 0.01). As for tissue-based samples, 6 kinds of malignancies including colorectal cancer, salivary adenoid cystic carcinoma, gallbladder carcinoma, colon cancer, gastrointestinal cancer, and alveolar rhabdomyosarcoma revealed upregulated miR-20a expression level compared with paired nontumorous tissue, of which high expression of miR-20a was significantly associated with poor OS (HR = 2.74, 95% CIs: 1.38-5.42, < 0.01) and DFS (HR = 2.68, 95% CIs: 1.32-5.45, < 0.01); meanwhile, other 5 tumors containing breast cancer, cutaneous squamous cell carcinoma, hepatocellular carcinoma, oral squamous cell carcinoma, and epithelial ovarian cancer demonstrated downregulated miR-20a expression level compared with benign tissue, of which low miR-20a expression was significantly related to shorter OS (HR = 3.48, 95% CIs: 2.00-6.06, < 0.01) and PFS/RFS (HR = 4.05, 95% CIs: 2.89-5.66, < 0.01).
CONCLUSION
Change of circulating and tissue-based miR-20a expression possesses vital prognostic implication for human cancers. Augmented expression of circulating miR-20a predicts poor survival outcome for patients with cancers. Tissue-based miR-20a level may be upregulated or downregulated depending on cancer types; in the former condition, high expression of tissue miR-20a is a risk factor for unfavorable prognosis and in the latter condition low expression of tissue miR-20a is associated with shorter survival.
Topics: Animals; Disease-Free Survival; Down-Regulation; Humans; MicroRNAs; Neoplasms; Prognosis; Up-Regulation
PubMed: 30596096
DOI: 10.1155/2018/6124927 -
Archives of Gynecology and Obstetrics Mar 2018Recently, conservative approaches such as wide local excisions and neoadjuvant chemotherapy are being considered to select young adult females with gynecologic RMS who...
PURPOSE
Recently, conservative approaches such as wide local excisions and neoadjuvant chemotherapy are being considered to select young adult females with gynecologic RMS who have strong desire to preserve fertility. This analysis aims to identify prognosticators affecting survival outcomes and defining potential candidacy for fertility-preservation. Another focus is to explore the role of chemotherapy in reducing the need for aggressive surgery and the role of radiotherapy in decreasing rates of local failure.
METHODS
A comprehensive database search identified 137 females > 16 years old with primary non-metastatic gynecologic RMS, who were included in a multivariate survival analysis.
RESULTS
5-year overall survival rate was 65%. Patients < 50 years old, with cervix uteri primaries, well-defined/polypoid presentations, embryonal histology and superficial tumors were more likely to survive. Deeply invasive disease and alveolar/pleomorphic histology significantly increased risks of treatment failure and tumor recurrence. Chemotherapy use was a significant multivariate predictor of better overall and metastasis-free survival. Radical surgery did not add local control or overall survival benefit for patients with superficial lesions (minimal/no cervical stromal invasion and no myometrial invasion).
CONCLUSIONS
While high-quality clinical trial evidence is missing, existing evidence seems to support holding back on radical surgery for selected candidates with well-defined, polypoid, superficial, embryonal cervical/endometrial RMS lesions that could be completely excised with conservative surgery; further local resections with/without radiotherapy are then warranted based on margin status. Experience on the use of neoadjuvant chemotherapy in the conservative management of uterine RMS in adults is very limited, though this approach is golden-standard in pediatrics. A suggested scheme is introduced for the management of uterine RMS.
Topics: Adolescent; Adult; Child; Disease-Free Survival; Female; Fertility; Fertility Preservation; Gynecologic Surgical Procedures; Humans; Middle Aged; Neoadjuvant Therapy; Neoplasm Recurrence, Local; Rhabdomyosarcoma; Survival Analysis; Survival Rate; Treatment Outcome; Uterine Cervical Neoplasms; Young Adult
PubMed: 29159540
DOI: 10.1007/s00404-017-4591-6 -
Critical Reviews in Oncology/hematology Oct 2015The objective of this systematic review is to provide an unprecedented summary of the prognostic impact of PAX3/7-FOXO1 fusion status in alveolar rhabdomyosarcoma. (Meta-Analysis)
Meta-Analysis Review
PURPOSE
The objective of this systematic review is to provide an unprecedented summary of the prognostic impact of PAX3/7-FOXO1 fusion status in alveolar rhabdomyosarcoma.
METHODS
Studies evaluating PAX3/7-FOXO1 fusion gene or its variants as a prognostic marker were systematically searched and comparative meta-analysis of overall survival was carried out.
RESULTS
A total of 7 studies comprising 993 patients with rhabdomyosarcoma were included. Three eligible studies showed no significant difference of survival between fusion positive and negative alveolar rhabdomyosarcoma. Four eligible studies showed that PAX3-FOXO1 fusion variant may indicate a lower survival probability than PAX7-FOXO1, although the effect did not reach a level of statistical significance (pooled hazard ratios, 1.66; 95% CI, 0.95-2.89; p=0.07).
CONCLUSIONS
There was no significant difference in the overall survival between patients with the positive and negative fusion gene, but there were indications of PAX3-FOXO1 being an unfavorable prognostic factor.
Topics: Forkhead Box Protein O1; Gene Fusion; Humans; PAX3 Transcription Factor; PAX7 Transcription Factor; Prognosis; Proportional Hazards Models; Rhabdomyosarcoma, Alveolar
PubMed: 26008753
DOI: 10.1016/j.critrevonc.2015.04.012