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Cancers Apr 2021The aim of this systematic review is to assess the latest age at diagnosis for detection of familial retinoblastoma in order to evaluate at what age screening of at-risk...
The aim of this systematic review is to assess the latest age at diagnosis for detection of familial retinoblastoma in order to evaluate at what age screening of at-risk children could be discontinued. Extended screening beyond this age would result in unnecessary patient burden and costs. However, discontinuing screening prematurely would have the adverse effect of missing tumors. We performed a literature search (PubMed, Embase, CINAHL and the Cochrane Library) up until February of 2021 and systematically included studies where patients had a family history of retinoblastoma, a known age at diagnosis, and who were ophthalmologically screened for retinoblastoma from birth. A total of 176 familial retinoblastoma patients from 17 studies were included in this review. Based on 48 months of age being the latest age of diagnosis, ophthalmological screening for familial retinoblastoma could safely be discontinued at age four years.
PubMed: 33920538
DOI: 10.3390/cancers13081942 -
Clinical & Translational Oncology :... Oct 2021Intra-arterial chemotherapy is a new retinoblastoma treatment associated with high rates of globe salvage that has been widely adopted for primary treatment of...
BACKGROUND
Intra-arterial chemotherapy is a new retinoblastoma treatment associated with high rates of globe salvage that has been widely adopted for primary treatment of retinoblastoma but is less frequently used as secondary treatment for refractory retinoblastoma. This systematic review aims to summarize the reported outcomes of intra-arterial chemotherapy for refractory retinoblastoma.
METHODS
We conducted a systematic review of studies published on PubMed, Medline, and Embase from 2011 to 2021 reporting globe salvage rates following intra-arterial chemotherapy for secondary treatment of refractory retinoblastoma.
RESULTS
Our search yielded 316 studies, and 24 met inclusion criteria. The 24 included studies were comprised of 1366 patients and 1757 eyes. Among these, 1184 (67%) eyes received secondary indication treatment, and globe salvage was achieved for 776 of these 1184 eyes (64%). Sixteen studies reported cannulation success rates from 71.8 to 100%. Pooled analysis of subjects revealed 21 patients (2.6%) with metastatic disease and 26 deaths (3%) during study follow-up periods (7-74 months). The most common ocular complications were vitreous hemorrhage (13.2%), loss of eyelashes (12.7%), and periocular edema (10.5%). The most common systemic complications were nausea/vomiting (20.5%), neutropenia (14.1%), fever (8.2%), and bronchospasm (6.2%).
CONCLUSIONS
Intra-arterial chemotherapy is associated with high rates of globe salvage and low rates of serious complications in patients with refractory retinoblastoma. Unfortunately, current literature is predominantly comprised of retrospective case studies, and further high-quality evidence is necessary to inform clinical practice.
Topics: Antineoplastic Agents; Bronchial Spasm; Carboplatin; Drug Resistance, Neoplasm; Edema; Eyelashes; Febrile Neutropenia; Humans; Infusions, Intra-Arterial; Melphalan; Methotrexate; Nausea; Retinal Neoplasms; Retinoblastoma; Salvage Therapy; Topotecan; Vitreous Hemorrhage; Vomiting
PubMed: 33826082
DOI: 10.1007/s12094-021-02610-z -
Asia-Pacific Journal of Ophthalmology...The aim of this study was to review the literature on various screening programs, devices, and applications described for the early detection of retinoblastoma.
PURPOSE
The aim of this study was to review the literature on various screening programs, devices, and applications described for the early detection of retinoblastoma.
DESIGN
Systematic review article.
METHODS
A PubMed® search was performed to identify articles published with specific reference to screening of neonates, infants and children for retinoblastoma.
RESULTS
Various devices and mobile phone-based applications based on altered red reflex are finding their way into community screening. Diagnosis of retinoblastoma by newborn eye screening is emphasized in several countries, and red reflex is the most widely employed technique.
CONCLUSIONS
Several screening programs for early detection of retinoblastoma are evolving in the developing countries, but the practices are not uniform. Universal newborn screening should be the norm. Newer tools and software can be utilized to screen infants on a community scale. Focussed research on revolutionizing digital imaging for a versatile screening tool holds promise for early diagnosis of retinoblastoma.
Topics: Child; Diagnostic Imaging; Early Diagnosis; Humans; Infant; Infant, Newborn; Neonatal Screening; Retinal Neoplasms; Retinoblastoma
PubMed: 33793441
DOI: 10.1097/APO.0000000000000378 -
Asia-Pacific Journal of Ophthalmology... Jan 2021To analyze the risk and benefit of high-dose chemotherapy followed by stem cell transplantation (HDCT-SCT) treatment in patients with advanced retinoblastoma.
PURPOSE
To analyze the risk and benefit of high-dose chemotherapy followed by stem cell transplantation (HDCT-SCT) treatment in patients with advanced retinoblastoma.
DESIGN
Systematic review.
METHODS
A comprehensive literature search from 4 online databases, including PubMed, Scopus, EBSCO, and Cochrane was done for original studies evaluating the use of HDCT followed by SCT in the treatment of patients with advanced retinoblastoma. The last search was performed on April 15, 2020.
RESULTS
A total of 35 studies consisting of 160 patients were considered suitable for inclusion. After HDCT-SCT treatment, 108/160 (67.5%) patients were alive with no evidence of disease at the last follow-up. The incidence of secondary malignancy in our data was also relatively low, which was 16/160 (10%) patients. The side effects were mainly hematological and gastrointestinal toxicities. The prognosis for metastatic cases especially the one to the central nervous system (CNS) remains poor, as shown in our data that 22 of 44 (50%) patients died due to the evidence of disease, and 12 of 44 (27%) patients acquired CNS relapse and died.
CONCLUSIONS
HDCT-SCT is a promising treatment option in patients with advanced retinoblastoma. The use of HDCT-SCT in CNS metastases needs to be carefully considered, possibly by adding thiotepa or topotecan to improve tumor control. Further randomized clinical trials are needed to draw firm conclusion regarding its safety and efficacy.
Topics: Antineoplastic Combined Chemotherapy Protocols; Combined Modality Therapy; Humans; Neoplasm Recurrence, Local; Retinal Neoplasms; Retinoblastoma; Stem Cell Transplantation
PubMed: 33481395
DOI: 10.1097/APO.0000000000000372 -
Pediatric Blood & Cancer Dec 2020Childhood cancer outcomes in low- and middle-income countries (LMICs) lag behind those in high-income countries (HICs), in part due to late presentation and diagnosis....
BACKGROUND
Childhood cancer outcomes in low- and middle-income countries (LMICs) lag behind those in high-income countries (HICs), in part due to late presentation and diagnosis. Though several interventions targeting early detection of childhood cancer have been implemented in LMICs, little is known about their efficacy.
METHODS
We conducted a systematic review to identify studies describing such interventions. We searched multiple databases from inception to December 4, 2019. Studies were included if they reported on LMIC interventions focused on: (a) training of health care providers on early recognition of childhood cancer, or (ii) public awareness campaigns. We used preferred reporting items for systematic reviews and meta-analyses (PRISMA) guidelines to conduct our review. The risk of bias in nonrandomized studies of interventions (ROBINS-I) checklist was used to assess quality of studies.
RESULTS
Twelve studies met inclusion criteria (n = 5 full text, n = 7 abstract only). Five studies focused on retinoblastoma only, while the others focused on all types of childhood cancer. The majority studied multiple interventions of which early detection was one component, but reported overall outcomes. All identified studies used pre-post evaluative designs to measure efficacy. Five studies reported statistically significant results postintervention: decrease in extraocular spread of retinoblastoma, decrease in rates of refusal/abandonment of treatment, increase in number of new referrals, increase in knowledge, and an absolute increase in median 5-year survival. Other studies reported improvements without tests of statistical significance. Two studies reported no difference in survival postintervention. The ROBINS-I checklist indicated that all studies were at serious risk of bias.
CONCLUSION
Though current evidence suggests that LMIC interventions targeting early detection of childhood cancer through health professional training and/or public awareness campaigns may be effective, this evidence is limited and of poor quality. Robust trials or quasi-experimental designs with long-term follow up are needed to identify the most effective interventions. Such studies will facilitate and inform the widespread uptake of early detection interventions across LMIC settings.
Topics: Child; Delivery of Health Care; Developing Countries; Early Detection of Cancer; Health Personnel; Humans; Neoplasms; Poverty
PubMed: 33037867
DOI: 10.1002/pbc.28761 -
Acta Diabetologica Feb 2021Diabetes mellitus (DM) is widely recognized as a risk factor for diverse cancers in adults. However, the association between maternal diabetes and risk of childhood... (Meta-Analysis)
Meta-Analysis
AIMS
Diabetes mellitus (DM) is widely recognized as a risk factor for diverse cancers in adults. However, the association between maternal diabetes and risk of childhood cancer in the offspring has so far not been well studied. We thus conducted a meta-analysis to evaluate the role of maternal diabetes on the risk of childhood cancer.
METHODS
We performed a comprehensive literature search to identify eligible studies published up to June 20, 2020, including the PubMed, Web of science and Embase databases. Summary odds ratios (OR) and 95% confidence intervals (CI) were computed using a random-effects model (I ≥ 25%) or a fixed-effect model (I < 25%).
RESULTS
Totally, sixteen case-control and six cohort studies on the risk of childhood cancer associated with maternal diabetes were included. Overall, children of diabetic women had a significantly increased risk in childhood malignancy (OR, 1.30; 95% CI, 1.10-1.53). Notably, a significantly elevated risk of childhood cancer in the offspring was found for women with pre-existing diabetes (OR, 1.41; 95% CI, 1.17-1.70), but not for women with gestational diabetes mellitus (GDM) (OR, 1.10; 95% CI, 0.94-1.28). For site-specific cancers, maternal diabetes was associated with a higher risk of leukemia in offspring (OR, 1.30; 95% CI, 1.15-1.48), especially for acute lymphoblastic leukemia (OR, 1.44; 95% CI, 1.27-1.64). However, no significant associations were observed between maternal diabetes and the risk of lymphomas and retinoblastoma.
CONCLUSIONS
Our meta-analysis indicates that maternal diabetes is associated with an increased risk of childhood cancer in the offspring, particularly for acute lymphoblastic leukemia. Future study should investigate the underlying biological mechanisms behind the association.
Topics: Adult; Case-Control Studies; Child; Child of Impaired Parents; Cohort Studies; Diabetes, Gestational; Female; Humans; Infant, Newborn; Male; Neoplasms; Observational Studies as Topic; Pregnancy; Risk Factors
PubMed: 32915298
DOI: 10.1007/s00592-020-01598-2 -
Neuroradiology Apr 2021Preoperative MRI detection of post-laminar optic nerve invasion (PLONI) offers guidance in assessing the probability of total tumor resection, an estimation of the... (Meta-Analysis)
Meta-Analysis
PURPOSE
Preoperative MRI detection of post-laminar optic nerve invasion (PLONI) offers guidance in assessing the probability of total tumor resection, an estimation of the extent of surgery, and screening of candidates for eye-preserving therapies or neoadjuvant chemotherapies in the patients with retinoblastoma (RB). The purpose of this systematic review and meta-analysis was to evaluate the diagnostic performance of MRI for detecting PLONI in patients with RB and to demonstrate the factors that may influence the diagnostic performance.
METHODS
Ovid-MEDLINE and EMBASE databases were searched up to January 11, 2020, for studies identifying the diagnostic performance of MRI for detecting PLONI in patients with RB. The pooled sensitivity and specificity of all studies were calculated followed by meta-regression analysis.
RESULTS
Twelve (1240 patients, 1255 enucleated globes) studies were included. The pooled sensitivity was 61%, and the pooled specificity was 88%. Higgins I statistic demonstrated moderate heterogeneity in the sensitivity (I = 72.23%) and specificity (I = 78.11%). Spearman correlation coefficient indicated the presence of a threshold effect. In the meta-regression, higher magnetic field strength (3 T than 1.5 T), performing fat suppression, and thinner slice thickness (< 3 mm) were factors causing heterogeneity and enhancing diagnostic power across the included studies.
CONCLUSIONS
MR imaging was demonstrated to have acceptable diagnostic performance in detecting PLONI in patients with RB. The variation in the magnetic field strength and protocols was the main factor behind the heterogeneity across the included studies. Therefore, there is room for developing and optimizing the MR protocols for patients with RB.
Topics: Humans; Magnetic Resonance Imaging; Neoplasm Invasiveness; Optic Nerve; Retinal Neoplasms; Retinoblastoma; Sensitivity and Specificity
PubMed: 32865636
DOI: 10.1007/s00234-020-02538-1 -
Cancer Cell International 2020Retinoblastoma is the most common malignant rare intraocular tumor of childhood. Long noncoding RNAs (lncRNAs) have been reported participating in its progression, but... (Review)
Review
BACKGROUND
Retinoblastoma is the most common malignant rare intraocular tumor of childhood. Long noncoding RNAs (lncRNAs) have been reported participating in its progression, but their significance remains inconclusive. We conducted this systematic review and meta-analysis to explore specific lncRNA biomarker in patients with retinoblastoma.
MATERIALS AND METHODS
Eligible articles were searched from the Pubmed, Web of Science, Embase and the Cochrane library. Hazard ratios (HRs) and odds ratios (ORs) were extracted or calculated to evaluate the relationship between lncRNAs and retinoblastoma. The meta-analysis part was conducted with STATA v.15 software.
RESULTS
A total of 9 articles with 834 retinoblastoma patients are yielded. Heterogeneity among HRs of overall survival (OS) is notably high (I = 91.3%, p < 0.001). Subgroup analysis suggests that elevated expression of lncRNA BDNF-AS and MT1JP are favorable factors in OS (pooled HR = 1.89, 95% CI 1.72-2.07, I = 0%). Six articles included optic nerve invasion as a clinicopathological outcome and showed a notable correlation (pooled HR = 2.38, 95% CI 1.26-3.50, I = 0.0%). We validate our analysis via the public dataset and also sum up the studies of lncRNA BDNF-AS and MT1JP in other cancers.
CONCLUSION
Differential expression of lncRNAs has been reported in retinoblastoma. Some of them showed potential in retinoblastoma prognosis and progression.
PubMed: 32514246
DOI: 10.1186/s12935-020-01281-0 -
Fetal and Pediatric Pathology Dec 2021Recently, epidemiological studies investigating the association of MTHFR 677 C > T, 1298 A > C and MTR 2756 A > G polymorphism with retinoblastoma... (Meta-Analysis)
Meta-Analysis
Association of MTHFR 677C > T, 1298A > C and MTR 2756A > G Polymorphisms with Susceptibility to Childhood Retinoblastoma: A Systematic Review and Met-Analysis.
Recently, epidemiological studies investigating the association of MTHFR 677 C > T, 1298 A > C and MTR 2756 A > G polymorphism with retinoblastoma susceptibility reported controversial results. Data were collected from several electronic databases such as PubMed, EMBASE, and Google Scholar databases, with the last search up to December 05, 2019. A total of eleven case-control studies including four studies with 324 cases and 490 controls on MTHFR 677 C > T, four studies with 324 cases and 490 controls on MTHFR 1298 A > C, and three studies with 283 cases and 485 controls on MTR 2756 A > G were selected. There was a significant association between MTHFR 677 C > T and MTR 2756 A > G polymorphisms and an increased risk of retinoblastoma. However, MTHFR 1298 A > C polymorphism was not significantly associated with risk of retinoblastoma. This meta-analysis demonstrated that MTHFR 677 C > T and MTR 2756 A > G polymorphisms might play important roles in the development of retinoblastoma. No association with MTHFR 1298 A > C polymorphism was observed.
Topics: 5-Methyltetrahydrofolate-Homocysteine S-Methyltransferase; Case-Control Studies; Child; Genetic Predisposition to Disease; Genotype; Humans; Methylenetetrahydrofolate Reductase (NADPH2); Polymorphism, Single Nucleotide; Retinal Neoplasms; Retinoblastoma
PubMed: 32064992
DOI: 10.1080/15513815.2020.1721738 -
Journal of Neurointerventional Surgery Dec 2019Intra-arterial chemotherapy for retinoblastoma has been adopted as a first-line treatment option by numerous tertiary centers. The effect of intra-arterial chemotherapy... (Meta-Analysis)
Meta-Analysis
BACKGROUND AND PURPOSE
Intra-arterial chemotherapy for retinoblastoma has been adopted as a first-line treatment option by numerous tertiary centers. The effect of intra-arterial chemotherapy on future rates of metastatic disease as well as on globe salvage in advanced eyes remains relatively unknown.
METHODS
A search of PubMED, MEDLINE, EMBASE, and Web of Science electronic databases was conducted from inception until January 2019 for studies with a minimum of 10 patients reporting outcomes and complications following intra-arterial chemotherapy for retinoblastoma.
RESULTS
A total of 20 studies met the inclusion criteria for analysis, comprising 873 patients and 1467 eyes. Only one study was comparative; there was substantial heterogeneity in reported outcomes and several overlapping patient cohorts that were published. Across all studies, 174 of 1467 eyes were enucleated (11.8%). Metastatic disease occurred in 8 of 513 patients (1.6%). Globe salvage was achieved in 318 of 906 (35.6%) cases of advanced retinoblastoma. The most common ocular complication was retinal detachment, occurring in 23% of eyes, and the most common systemic complications were transient fever and nausea/vomiting.
CONCLUSIONS
There is a paucity of higher-level evidence with adequate follow-up surrounding the long-term safety of intra-arterial chemotherapy and effect on metastasis in retinoblastoma. Studies to date have been limited by short-term follow-up. Longitudinal prospective studies could provide greater insight into the ability of intra-arterial chemotherapy to reduce the risk of retinoblastoma metastasis.
Topics: Antineoplastic Agents; Child, Preschool; Female; Humans; Infant; Infusions, Intra-Arterial; Male; Melphalan; Prospective Studies; Retinal Neoplasms; Retinoblastoma; Retrospective Studies; Salvage Therapy
PubMed: 31103993
DOI: 10.1136/neurintsurg-2019-014909