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      Cervical aortic arch in the pediatric population: a meta-analysis of individual patient's data

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          Abstract

          Background

          This is the first meta-analysis to analyze all reports of published pediatric cases of cervical aortic arch (CAA) by highlighting the clinical characteristics and treatment outcomes using the reported individual data of the patients. The aim of the study is to investigate the clinical features and surgical outcomes of such a rare disease in the pediatric population.

          Methods

          A comprehensive search was conducted in various academic databases, including PubMed, ScienceDirect, SciELO, DOAJ, and Cochrane Library, until June 2022 for case reports describing the presence of cervical aortic arch in the pediatric age. Case reports and series were included if the following criteria were met: (1) description of the cervical aortic arch; (2) patient of pediatric age; and (3) published in the English language. All other types of publications that lacked patient-specific information were excluded from the analysis. This systematic review was conducted in accordance with the PRISMA guidelines. The primary outcome measure of the analysis was early and late mortality.

          Results

          The literature search identified 2,272 potentially eligible articles, 72 of which met our inclusion criteria with 96 patients including the author's institutional case. At a median of 365 (90–730) days, the overall cohort registered a 7.3% (7/96) mortality rate. In the subset of patients who underwent surgery, the mortality rate was also 7.3% (4/55), and the mortality rate following surgery to treat only CAA was 2.4% (1/42). Dyspnea was identified as an independent determinant of mortality by employing the univariable Firth bias-reduced logistic regression method.

          Conclusion

          Cervical aortic arch is a rare congenital heart disease that poses treatment challenges due to the high anatomical variability, diverse clinical presentations, and the presence of other concomitant diseases. The surgical treatment appears to be a safe and effective approach for resolving the symptoms, although it needs to be tailored individually for each patient.

          Systematic Review Registration

          https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=346826, Identifier: CRD42022346826.

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          Most cited references80

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          PRISMA 2020 explanation and elaboration: updated guidance and exemplars for reporting systematic reviews

          The methods and results of systematic reviews should be reported in sufficient detail to allow users to assess the trustworthiness and applicability of the review findings. The Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) statement was developed to facilitate transparent and complete reporting of systematic reviews and has been updated (to PRISMA 2020) to reflect recent advances in systematic review methodology and terminology. Here, we present the explanation and elaboration paper for PRISMA 2020, where we explain why reporting of each item is recommended, present bullet points that detail the reporting recommendations, and present examples from published reviews. We hope that changes to the content and structure of PRISMA 2020 will facilitate uptake of the guideline and lead to more transparent, complete, and accurate reporting of systematic reviews.
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            The CARE guidelines: consensus-based clinical case reporting guideline development.

            A case report is a narrative that describes, for medical, scientific, or educational purposes, a medical problem experienced by one or more patients. Case reports written without guidance from reporting standards are insufficiently rigorous to guide clinical practice or to inform clinical study design. Develop, disseminate, and implement systematic reporting guidelines for case reports. We used a three-phase consensus process consisting of (1) pre-meeting literature review and interviews to generate items for the reporting guidelines, (2) a face-to-face consensus meeting to draft the reporting guidelines, and (3) post-meeting feedback, review, and pilot testing, followed by finalization of the case report guidelines. This consensus process involved 27 participants and resulted in a 13-item checklist-a reporting guideline for case reports. The primary items of the checklist are title, key words, abstract, introduction, patient information, clinical findings, timeline, diagnostic assessment, therapeutic interventions, follow-up and outcomes, discussion, patient perspective, and informed consent. We believe the implementation of the CARE (CAse REport) guidelines by medical journals will improve the completeness and transparency of published case reports and that the systematic aggregation of information from case reports will inform clinical study design, provide early signals of effectiveness and harms, and improve healthcare delivery. © 2013 Gagnier et al.; licensee Wiley Periodicals, Inc.
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              Chapter 7: Systematic Reviews of Etiology and Risk

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                Author and article information

                Contributors
                URI : https://loop.frontiersin.org/people/1399335/overviewRole: Role: Role: Role: Role: Role: Role: Role: Role: Role: Role:
                Role: Role: Role: Role:
                Role: Role: Role:
                Role: Role: Role:
                URI : https://loop.frontiersin.org/people/69391/overviewRole: Role: Role:
                URI : https://loop.frontiersin.org/people/1090463/overviewRole: Role: Role:
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                URI : https://loop.frontiersin.org/people/1059273/overviewRole: Role: Role: Role: Role: Role: Role: Role:
                Journal
                Front Cardiovasc Med
                Front Cardiovasc Med
                Front. Cardiovasc. Med.
                Frontiers in Cardiovascular Medicine
                Frontiers Media S.A.
                2297-055X
                28 September 2023
                2023
                : 10
                : 1266956
                Affiliations
                [ 1 ]Department of Congenital Cardiac Surgery, IRCCS Policlinico San Donato , San Donato Milanese, Italy
                [ 2 ]Department of Cardiac Surgery, ASST Spedali Civili di Brescia, University of Brescia , Brescia, Italy
                [ 3 ]Department of Pediatric and Adult Congenital Cardiology, IRCCS Policlinico San Donato , San Donato Milanese, Italy
                Author notes

                Edited by: Arpit Kumar Agarwal, Baylor College of Medicine, United States

                Reviewed by: Cosimo Marco Campanale, Bambino Gesù Children's Hospital (IRCCS), Italy Maruti Haranal, U N Mehta Institute of Cardiology and Research, India

                [* ] Correspondence: Mauro Lo Rito mauro.lorito@ 123456gmail.com
                Article
                10.3389/fcvm.2023.1266956
                10580808
                37855019
                cade70da-9ea6-4e25-902d-73dfef54dafb
                © 2023 Baudo, Varrica, Reali, Saracino, Carminati, Frigiola, Giamberti and Lo Rito.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                : 25 July 2023
                : 11 September 2023
                Page count
                Figures: 3, Tables: 3, Equations: 0, References: 80, Pages: 0, Words: 0
                Funding
                Funded by: The authors declare that no financial support was received for the research, authorship, and/or publication of this article.
                This work was supported by IRCCS Policlinico San Donato, a Clinical Research Hospital partially funded by the Italian Ministry of Health.
                Categories
                Cardiovascular Medicine
                Systematic Review
                Custom metadata
                Pediatric Cardiology

                cervical aortic arch,congenital heart disease,cardiac surgery,pediatrics,meta-analysis

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