儿科人群中的颈主动脉弓：个体患者数据的荟萃分析
Cervical aortic arch in the pediatric population: a meta-analysis of individual patient's data.
作者信息
Baudo Massimo, Varrica Alessandro, Reali Matteo, Saracino Antonio, Carminati Mario, Frigiola Alessandro, Giamberti Alessandro, Lo Rito Mauro
机构信息
Department of Congenital Cardiac Surgery, IRCCS Policlinico San Donato, San Donato Milanese, Italy.
Department of Cardiac Surgery, ASST Spedali Civili di Brescia, University of Brescia, Brescia, Italy.
出版信息
Front Cardiovasc Med. 2023 Sep 28;10:1266956. doi: 10.3389/fcvm.2023.1266956. eCollection 2023.
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.
背景
这是第一项荟萃分析，通过突出使用所报告患者个体数据的临床特征和治疗结果，来分析已发表的小儿颈主动脉弓（CAA）病例的所有报告。本研究的目的是调查这种罕见疾病在儿科人群中的临床特征和手术结果。
方法
在包括PubMed、ScienceDirect、SciELO、DOAJ和Cochrane图书馆在内的各种学术数据库中进行了全面检索，直至2022年6月，以查找描述小儿年龄患者存在颈主动脉弓的病例报告。如果满足以下标准，则纳入病例报告和系列研究：（1）颈主动脉弓的描述；（2）小儿年龄患者；（3）以英文发表。所有缺乏患者特定信息的其他类型出版物均被排除在分析之外。本系统评价按照PRISMA指南进行。分析的主要结局指标是早期和晚期死亡率。
结果
文献检索确定了2272篇潜在符合条件的文章，其中72篇符合我们的纳入标准，包括作者机构的病例在内共96例患者。在中位时间365（90 - 730）天，整个队列的死亡率为7.3%（7/96）。在接受手术的患者亚组中，死亡率也为7.3%（4/55），仅治疗CAA的手术后死亡率为2.4%（1/42）。通过使用单变量Firth偏倚减少逻辑回归方法，呼吸困难被确定为死亡率的独立决定因素。
结论
颈主动脉弓是一种罕见的先天性心脏病，由于其高度的解剖变异性、多样的临床表现以及其他伴随疾病的存在，给治疗带来了挑战。手术治疗似乎是缓解症状的一种安全有效的方法，尽管需要为每个患者进行个体化定制。
系统评价注册
https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=346826，标识符：CRD42022346826。
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