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PloS One 2017Our objective was to evaluate quality of conduct and reporting of published systematic reviews and meta-analyses in paediatric surgery. We also aimed to identify... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
Our objective was to evaluate quality of conduct and reporting of published systematic reviews and meta-analyses in paediatric surgery. We also aimed to identify characteristics predictive of review quality.
BACKGROUND
Systematic reviews summarise evidence by combining sources, but are potentially prone to bias. To counter this, the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) was published to aid in reporting. Similarly, the Assessing the Methodological Quality of Systematic Reviews (AMSTAR) measurement tool was designed to appraise methodology. The paediatric surgical literature has seen an increasing number of reviews over the past decade, but quality has not been evaluated.
METHODS
Adhering to PRISMA guidelines, we performed a systematic review with a priori design to identify systematic reviews and meta-analyses of interventions in paediatric surgery. From 01/2010 to 06/2016, we searched: MEDLINE, EMBASE, Cochrane, Centre for Reviews and Dissemination, Web of Science, Google Scholar, reference lists and journals. Two reviewers independently selected studies and extracted data. We assessed conduct and reporting using AMSTAR and PRISMA. Scores were calculated as the sum of reported items. We also extracted author, journal and article characteristics, and used them in exploratory analysis to determine which variables predict quality.
RESULTS
112 articles fulfilled eligibility criteria (53 systematic reviews; 59 meta-analyses). Overall, 68% AMSTAR and 56.8% PRISMA items were reported adequately. Poorest scores were identified with regards a priori design, inclusion of structured summaries, including the grey literature, citing excluded articles and evaluating bias. 13 reviews were pre-registered and 6 in PRISMA-endorsing journals. The following predicted quality in univariate analysis:, word count, Cochrane review, journal h-index, impact factor, journal endorses PRISMA, PRISMA adherence suggested in author guidance, article mentions PRISMA, review includes comparison of interventions and review registration. The latter three variables were significant in multivariate regression.
CONCLUSIONS
There are gaps in the conduct and reporting of systematic reviews in paediatric surgery. More endorsement by journals of the PRISMA guideline may improve review quality, and the dissemination of reliable evidence to paediatric clinicians.
Topics: Child; Humans; Pediatrics; Surgical Procedures, Operative
PubMed: 28384296
DOI: 10.1371/journal.pone.0175213 -
Journal of Medical Internet Research Dec 2021The emergence of genetic and genomic sequencing approaches for pediatric patients has raised questions about the genomic health literacy levels, attitudes toward... (Review)
Review
BACKGROUND
The emergence of genetic and genomic sequencing approaches for pediatric patients has raised questions about the genomic health literacy levels, attitudes toward receiving genomic information, and use of this information to inform treatment decisions by pediatric patients and their parents. However, the methods to educate pediatric patients and their parents about genomic concepts through digital health interventions have not been well-established.
OBJECTIVE
The primary objective of this scoping review is to investigate the current levels of genomic health literacy and the attitudes toward receiving genomic information among pediatric patients and their parents. The secondary aim is to investigate patient education interventions that aim to measure and increase genomic health literacy among pediatric patients and their parents. The findings from this review will be used to inform future digital health interventions for patient education.
METHODS
A scoping review using PRISMA-ScR (Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews) guidelines and protocols was completed using the following databases: MEDLINE, Embase, CINAHL, and Scopus. Our search strategy included genomic information inclusive of all genetic and genomic terms, pediatrics, and patient education. Inclusion criteria included the following: the study included genetic, genomic, or a combination of genetic and genomic information; the study population was pediatric (children and adolescents <18 years) and parents of patients with pediatric illnesses or only parents of patients with pediatric illnesses; the study included an assessment of the knowledge, attitudes, and intervention regarding genomic information; the study was conducted in the last 12 years between 2008 and 2020; and the study was in the English language. Descriptive data regarding study design, methodology, disease population, and key findings were extracted. All the findings were collated, categorized, and reported thematically.
RESULTS
Of the 4618 studies, 14 studies (n=6, 43% qualitative, n=6, 43% mixed methods, and n=2, 14% quantitative) were included. Key findings were based on the following 6 themes: knowledge of genomic concepts, use of the internet and social media for genomic information, use of genomic information for decision-making, hopes and attitudes toward receiving genomic information, experiences with genetic counseling, and interventions to improve genomic knowledge.
CONCLUSIONS
This review identified that older age is related to the capacity of understanding genomic concepts, increased genomic health literacy levels, and the perceived ability to participate in decision-making related to genomic information. In addition, internet-searching plays a major role in obtaining genomic information and filling gaps in communication with health care providers. However, little is known about the capacity of pediatric patients and their parents to understand genomic information and make informed decisions based on the genomic information obtained. More research is required to inform digital health interventions and to leverage the leading best practices to educate these genomic concepts.
Topics: Adolescent; Aged; Child; Communication; Genomics; Health Literacy; Humans; Parents; Pediatrics
PubMed: 34951592
DOI: 10.2196/26684 -
Journal of Pediatric Surgery Apr 2016A small percentage of pediatric solid cancers arise as a result of clearly identified inherited predisposition syndromes and nongenetic lesions. Evidence supports... (Review)
Review
OBJECTIVE
A small percentage of pediatric solid cancers arise as a result of clearly identified inherited predisposition syndromes and nongenetic lesions. Evidence supports preemptive surgery for children with genetic [multiple endocrine neoplasia type 2 (MEN2), familial adenomatous polyposis syndrome (FAP), hereditary nonpolyposis colorectal cancer (HNPCC), and hereditary diffuse gastric cancer (HDGC) and nongenetic [thyroglossal duct cysts (TGDC), congenital pulmonary airway malformations (CPAM), alimentary tract duplication cysts (ATDC), and congenital choledochal cysts (CCC)] developmental anomalies. Our aim was to explore the utility of risk reduction surgery to treat and prevent cancer in children.
METHODS
A systematic review of the available peer-reviewed literature on PubMed was performed using a PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) search strategy, where possible. Search items included "risk reduction surgery", "hereditary cancer predisposition syndrome", "multiple endocrine neoplasia type 2", "familial adenomatous polyposis", "hereditary nonpolyposis colorectal cancer", "hereditary diffuse gastric cancer", "thyroglossal duct cysts", congenital pulmonary airway malformations", "alimentary tract duplication cysts", "malignant transformation", and "guidelines".
RESULTS
We identified 67 articles that met the inclusion criteria describing the indications for prophylactic surgery in surgical oncology. For the genetic predisposition syndromes, 7 studies were related to professional endorsed guidelines, 7 were related to surgery for MEN2, 11 were related to colectomy for FAP, 6 were related to colectomy for HNPCC, and 12 related to gastrectomy for HDGC. Articles for the nongenetic lesions included 5 for techniques related to TGDC resection, 9 for surgery for CPAMs, and 10 for resection of ATDCs. Guidelines and strategies varied significantly especially related to the extent and timing of surgical intervention; the exception was for the timing of thyroidectomy in children with MEN2.
CONCLUSION
Current evidence supporting prophylactic surgery in the management of pediatric cancer predisposition syndromes and nongenetic lesions is best delineated for thyroidectomy to prevent medullary thyroid cancer in children with MEN2 (Strength of Recommendation Grade B/C). Despite the lack of pediatric specific evidence-based recommendations regarding the appropriate extent and timing for risk-reduction surgery for FAP, HNPCC, HDGC and nongenetic anomalies, our review represents an opportunity towards understanding the postgenomic development of these lesions and provides current indications and techniques for preemptive cancer prevention surgery in children.
Topics: Child; Humans; Neoplastic Syndromes, Hereditary; Pediatrics; Prophylactic Surgical Procedures; Surgical Oncology; Treatment Outcome
PubMed: 26898681
DOI: 10.1016/j.jpedsurg.2016.01.004 -
Graefe's Archive For Clinical and... Apr 2022Recently, endothelial keratoplasty (EK) has been increasingly considered the first intervention in pediatrics with isolated corneal endothelial dysfunction. This... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Recently, endothelial keratoplasty (EK) has been increasingly considered the first intervention in pediatrics with isolated corneal endothelial dysfunction. This systematic review aims to investigate the current evidence about the advantages, disadvantages, technical challenges, and clinical outcomes of EK in the pediatric group.
METHOD
All the English literature relevant to pediatric EK was searched in PubMed, Embase, Scopus, and Cochrane databases with appropriate keywords. Relevant data were pooled to conduct an individual participant data meta-analysis.
RESULTS
Of 1646 articles found initially, 35 articles were finally eligible to be included in our study. A total of 154 eyes of 107 patients underwent Descemet's stripping automated endothelial keratoplasty (DSAEK). Congenital hereditary endothelial dystrophy (CHED) was the most reported indication for pediatric DSAEK (108 eyes). Descemet's membrane endothelial keratoplasty (DMEK) was performed in 2 eyes of 2 cases, one with PPCD and another one in a patient with Kearns-Sayre syndrome. Owing to some specific anatomical and physiological pediatric characteristics, some modifications in a standard procedure were suggested. The average follow-up period was 23.80 ± 20.18 months (3 months to 8.5 years). Seventy-six eyes who had a mean best-corrected visual acuity (BCVA) of 1.36 ± 0.70 (0.49 to 3) logMAR preoperatively found mean BCVA of 0.51 ± 0.33 (0.04 to 2) logMAR postoperatively. Graft dislocation was the most reported complication (26 eyes). The rate of other complications was low. Endothelial cell loss was reported from 8.3 to 63.7% after pediatric EK with follow-up duration from 3 months to 8.3 years.
CONCLUSION
EK procedures, despite some technical challenges, are feasible surgical techniques with acceptable visual and anatomical outcomes in the management of pediatrics with corneal endothelial dysfunction and minimal stromal involvement.
Topics: Child; Descemet Membrane; Descemet Stripping Endothelial Keratoplasty; Endothelium, Corneal; Fuchs' Endothelial Dystrophy; Humans; Pediatrics; Retrospective Studies; Visual Acuity
PubMed: 34709453
DOI: 10.1007/s00417-021-05459-8 -
Annals of the Rheumatic Diseases Sep 2015: Autoinflammatory diseases are characterised by fever and systemic inflammation, with potentially serious complications. Owing to the rarity of these diseases,... (Review)
Review
: Autoinflammatory diseases are characterised by fever and systemic inflammation, with potentially serious complications. Owing to the rarity of these diseases, evidence-based guidelines are lacking. In 2012, the European project Single Hub and Access point for paediatric Rheumatology in Europe (SHARE) was launched to optimise and disseminate regimens for the management of children and young adults with rheumatic diseases, facilitating the clinical practice of paediatricians and (paediatric) rheumatologists. One of the aims of SHARE was to provide evidence-based recommendations for the management of the autoinflammatory diseases cryopyrin-associated periodic syndromes (CAPS), tumour necrosis factor (TNF) receptor-associated periodic syndrome (TRAPS) and mevalonate kinase deficiency (MKD). These recommendations were developed using the European League Against Rheumatism standard operating procedure. An expert committee of paediatric and adult rheumatologists was convened. Recommendations derived from the systematic literature review were evaluated by an online survey and subsequently discussed at a consensus meeting using Nominal Group Technique. Recommendations were accepted if more than 80% agreement was reached. In total, four overarching principles, 20 recommendations on therapy and 14 recommendations on monitoring were accepted with ≥80% agreement among the experts. Topics included (but were not limited to) validated disease activity scores, therapy and items to assess in monitoring of a patient. By developing these recommendations, we aim to optimise the management of patients with CAPS, TRAPS and MKD.
Topics: Consensus; Cryopyrin-Associated Periodic Syndromes; Fever; Hereditary Autoinflammatory Diseases; Humans; Mevalonate Kinase Deficiency; Practice Guidelines as Topic
PubMed: 26109736
DOI: 10.1136/annrheumdis-2015-207546 -
BMC Pediatrics Apr 2022Vedolizumab use in pediatrics is still off-label and the data are limited. We conducted a systematic review evaluating the efficacy and safety of vedolizumab in children...
BACKGROUND
Vedolizumab use in pediatrics is still off-label and the data are limited. We conducted a systematic review evaluating the efficacy and safety of vedolizumab in children and adolescents with inflammatory bowel disease (IBD).
METHODS
PubMed, EMBASE and Cochrane databases were systematically searched for studies of vedolizumab in children and adolescents with IBD reporting clinical remission, response, corticosteroid-free (CS-free) remission, mucosal healing, or safety up to December 3 2021.
RESULTS
Ten studies, comprising 455 patients were included. For CD, the pooled clinical remission rates were 25% (19/75) at 6 weeks, 28% (25/85) at 14 weeks, 32% (17/53) at 22 weeks, and 46% (43/92) at 1 year. For UC/IBD-U, the pooled clinical remission rates were 36% (25/70) at 6 weeks, 48% (52/101) at 14 weeks, 53% (24/45) at 22 weeks, and 45% (50/112) at 1 year. Mucosal healing was found in 17%-39% of CD and 15%-34% of UC/IBD-U respectively. Six percent of patients reported serious adverse events.
CONCLUSIONS
According to low-quality evidence based on case series, approximately one-third and one-half of patients for CD and UC/IBD-U respectively achieved remission within 22 weeks, and about half of patients achieved remission at 1 year with reasonable safety profile. Long-term benefit profile data and high quality evidence are still needed.
Topics: Adolescent; Antibodies, Monoclonal, Humanized; Child; Humans; Inflammatory Bowel Diseases; Pediatrics; Remission Induction
PubMed: 35379216
DOI: 10.1186/s12887-022-03229-x -
Resuscitation Mar 2021To evaluate the optimal timing and doses of epinephrine for Infants and children suffering in-hospital or out-of-hospital cardiac arrest. (Meta-Analysis)
Meta-Analysis Review
AIM
To evaluate the optimal timing and doses of epinephrine for Infants and children suffering in-hospital or out-of-hospital cardiac arrest.
METHODS
We searched Medline, EMBASE, and Cochrane Controlled Register of Trials (CENTRAL) for human randomized clinical trials and observational studies including comparative cohorts. Two investigators reviewed relevance of studies, extracted the data, conducted meta-analyses and assessed the risk of bias using the GRADE and CLARITY frameworks. Authors of the eligible studies were contacted to obtain additional data. Critically important outcomes included return of spontaneous circulation, survival to hospital discharge and survival with good neurological outcome.
RESULTS
We identified 7 observational studies suitable for meta-analysis and no randomized clinical trials. The overall certainty of evidence was very low. For the critically important outcomes, the earlier administration of epinephrine was favorable for both in-hospital and out-of-hospital cardiac arrest. Because of a limited number of eligible studies and the presence of severe confounding factors, we could not determine the optimal interval of epinephrine administration.
CONCLUSIONS
Earlier administration of the first epinephrine dose could be more favorable in non-shockable pediatric cardiac arrest. The optimal interval for epinephrine administration remains unclear.
Topics: Child; Epinephrine; Humans; Infant; Out-of-Hospital Cardiac Arrest; Patient Discharge
PubMed: 33529645
DOI: 10.1016/j.resuscitation.2021.01.015 -
Acta Paediatrica (Oslo, Norway : 1992) May 2019To explore, synthesise and discuss currently available digital stethoscopes (DS) and the evidence for their use in paediatric medicine.
AIM
To explore, synthesise and discuss currently available digital stethoscopes (DS) and the evidence for their use in paediatric medicine.
METHODS
Systematic review and narrative synthesis of digital stethoscope use in paediatrics following searches of OVID Medline, Embase, Scopus, PubMed and Google Scholar databases.
RESULTS
Six digital stethoscope makes were identified to have been used in paediatric focused studies so far. A total of 25 studies of DS use in paediatrics were included. We discuss the use of digital stethoscope technology in current paediatric medicine, comment on the technical properties of the available devices, the effectiveness and limitations of this technology, and potential uses in the fields of paediatrics and neonatology, from telemedicine to computer-aided diagnostics.
CONCLUSION
Further validation and testing of available DS devices is required. Comparison studies between different types of DS would be useful in identifying strengths and flaws of each DS as well as identifying clinical situations for which each may be most appropriately suited.
Topics: Humans; Pediatrics; Stethoscopes
PubMed: 30536440
DOI: 10.1111/apa.14686 -
European Journal of Pediatrics May 2018As more new drugs are discovered, traditional designs come at their limits. Ten years after the adoption of the European Paediatric Regulation, we performed a systematic... (Review)
Review
UNLABELLED
As more new drugs are discovered, traditional designs come at their limits. Ten years after the adoption of the European Paediatric Regulation, we performed a systematic review on the US National Library of Medicine and Excerpta Medica database of sequential trials involving newborns. Out of 326 identified scientific reports, 21 trials were included. They enrolled 2832 patients, of whom 2099 were analyzed: the median number of neonates included per trial was 48 (IQR 22-87), median gestational age was 28.7 (IQR 27.9-30.9) weeks. Eighteen trials used sequential techniques to determine sample size, while 3 used continual reassessment methods for dose-finding. In 16 studies reporting sufficient data, the sequential design allowed to non-significantly reduce the number of enrolled neonates by a median of 24 (31%) patients (IQR - 4.75 to 136.5, p = 0.0674) with respect to a traditional trial. When the number of neonates finally included in the analysis was considered, the difference became significant: 35 (57%) patients (IQR 10 to 136.5, p = 0.0033).
CONCLUSION
Sequential trial designs have not been frequently used in Neonatology. They might potentially be able to reduce the number of patients in drug trials, although this is not always the case. What is known: • In evaluating rare diseases in fragile populations, traditional designs come at their limits. About 20% of pediatric trials are discontinued, mainly because of recruitment problems. What is new: • Sequential trials involving newborns were infrequently used and only a few (n = 21) are available for analysis. • The sequential design allowed to non-significantly reduce the number of enrolled neonates by a median of 24 (31%) patients (IQR - 4.75 to 136.5, p = 0.0674).
Topics: Clinical Trials as Topic; Humans; Infant, Newborn; Neonatology; Research Design
PubMed: 29453599
DOI: 10.1007/s00431-018-3110-5 -
European Journal of Clinical... Mar 2017Stress is defined as a state of threatened or perceived as threatened homeostasis. A broad spectrum of extrinsic or intrinsic, real or perceived stressful stimuli,... (Review)
Review
BACKGROUND
Stress is defined as a state of threatened or perceived as threatened homeostasis. A broad spectrum of extrinsic or intrinsic, real or perceived stressful stimuli, called 'stressors', activates a highly conserved system, the 'stress system', which adjusts homeostasis through central and peripheral neuroendocrine responses. Inadequate, excessive or prolonged adaptive responses to stress may underlie the pathogenesis of several disease states prevalent in modern societies. The development and severity of these conditions primarily depend on the genetic vulnerability of the individual, the exposure to adverse environmental factors and the timing of the stressful event(s), given that prenatal life, infancy, childhood and adolescence are critical periods characterized by increased vulnerability to stressors.
MATERIALS AND METHODS
We conducted a systematic review of original articles and reviews published in MEDLINE from 1975 through June 2016. The search terms were 'childhood stress', 'pediatric stress', 'stress and disorders' and 'stress management'.
RESULTS
In this review, we discuss the historical and neuroendocrine aspects of stress, and we present representative examples of paediatric stress system disorders, such as early-life adversity, obesity and bullying. We also discuss the adverse impact of a socio-economic crisis on childhood health. The tremendous progress of epigenetics has enabled us to have a deeper understanding of the molecular mechanisms underlying paediatric stress-related disorders.
CONCLUSIONS
The need for early successful stress management techniques to decrease the incidence of paediatric stress-related diseases, as well as to prevent the development of several pathologic conditions in adolescence and adulthood, is imperative.
Topics: Child; Humans; Neuroendocrinology; Neurosecretory Systems; Obesity; Stress, Physiological; Stress, Psychological
PubMed: 28074555
DOI: 10.1111/eci.12724