Mortality in Cleft Lip and Palate Patients: A Systematic Review and Meta-analysis

Abstract

Background: 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.

Methods: 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.

Results: 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], P = 0.005) or isolated palate (OR = 0.34 [0.24, 0.48], P = 0.0005). Increased odds of mortality were found in patients with comorbidities (OR = 19.79 [11.37, 34.43], P < 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 P values less than 0.05.

Conclusions: 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.

Fig. 1.

PRISMA flow diagram of the search strategy. PRIMA, preferred reporting items for systematic reviews and meta-analysis.

Fig. 2.

Forest plot of overall mortality among included articles. Confidence intervals are at the 95% confidence level. The solid red line represents the median mortality proportion, and the dashed lines are the 95% confidence interval of the median value.