Plonkowski Alexander T, Naidu Priyanka, Celie Karel-Bart, Munabi Naikhoba C O, Nagengast Eric, Tapia Maria Fernanda, Ntirenganya Faustin, Yao Caroline A, Magee William P
From the Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles, Los Angeles, CA.
Operation Smile Incorporated, Virginia Beach, VA.
Plast Reconstr Surg Glob Open. 2025 May 1;13(5):e6721. doi: 10.1097/GOX.0000000000006721. eCollection 2025 May.
Cleft lip and/or palate (CLP) represents one of the most common congenital anomalies among live births globally. Morbidity associated with CLP is well documented in the literature and stratified through measures such as disability-adjusted life years. However, a paucity of literature details mortality rates among different subgroups of patients with CLP.
A meta-analysis was performed using preferred reporting items for systematic reviews and meta-analysis guidelines. Included studies reported on mortality rates of patients with CLP in a liveborn cohort. Studies that reported only intraoperative mortality, included fetal mortality, or included non-CLP conditions in mortality rates were excluded. Meta-analysis was performed through subgroup analysis using random-effects models.
Twenty-one studies were included. Mortality rates ranged from 1.22% to 19.8% between studies. Patients with isolated cleft lip had lower odds of mortality compared with CLP (odds ratio [OR] = 0.28 [0.14, 0.56], = 0.005) or isolated palate (OR = 0.34 [0.24, 0.48], = 0.0005). Increased odds of mortality were found in patients with comorbidities (OR = 19.79 [11.37, 34.43], < 0.0001) compared with otherwise healthy CLP patients. Across age groups, neonates (0-28 d) had the highest mortality rate (0.7%-19.8%) followed by infants (0.2%-6.6%), both with values less than 0.05.
Mortality rates in patients with CLP are higher for those with an additional comorbidity or younger age (<1 y). Further studies stratifying data by cleft phenotype and age are required to better understand factors that contribute to CLP mortality.
唇腭裂(CLP)是全球活产儿中最常见的先天性畸形之一。与CLP相关的发病率在文献中有充分记载,并通过伤残调整生命年等指标进行分层。然而,关于CLP不同亚组患者死亡率的文献却很少。
按照系统评价和Meta分析的首选报告项目指南进行Meta分析。纳入的研究报告了活产队列中CLP患者的死亡率。仅报告术中死亡率、包括胎儿死亡率或死亡率中包含非CLP疾病的研究被排除。通过随机效应模型进行亚组分析来进行Meta分析。
纳入21项研究。各研究之间的死亡率在1.22%至19.8%之间。与唇腭裂(CLP)患者相比,单纯唇裂患者的死亡几率较低(比值比[OR]=0.28[0.14,0.56],P=0.005),与单纯腭裂患者相比也是如此(OR=0.34[0.24,0.48],P=0.0005)。与健康的CLP患者相比,合并症患者的死亡几率增加(OR=19.79[11.37,34.43],P<0.0001)。在各个年龄组中,新生儿(0 - 28天)的死亡率最高(0.7% - 19.8%),其次是婴儿(0.2% - 6.6%),两者P值均小于0.05。
合并其他疾病或年龄较小(<1岁)的CLP患者死亡率较高。需要进一步按腭裂表型和年龄对数据进行分层研究,以更好地了解导致CLP患者死亡的因素。