This review aimed to evaluate the association between craniofacial features in children and adolescents with pediatric obstructive sleep apnea (OSA).Seven databases were searched to fulfill our research objectives. Clinical studies that included participants younger than 18 years with fully diagnosed OSA or without OSA and that evaluated skeletal, soft craniofacial features, or dental arch morphology were considered for this review. The risk of bias and certainty of evidence were assessed. A meta-analysis was performed when low methodological and clinical heterogeneity were detected. This review followed the protocols recommended by the Preferred Reporting Items for a Systematic Review and Meta-analysis (PRISMA-2020) guidelines.Nine studies were identified at the end of the selection process, from which 5 did not report differences. Four studies reported differences between craniofacial features when OSA was compared to an asymptomatic control group. Mandibular retrognathia, reduced anteroposterior linear dimensions of the bony nasopharynx (decreased pharyngeal diameters at the levels of the adenoids), longer facial profile, and a narrower intercanine width were described among children with OSA. A meta-analysis was performed considering the studies with a similar methodological approach, and no differences were observed in all the considered cephalometric angles (SNA, SNB, ANB, NSBa, U1-L1, U1-SN). All the included studies were considered at low risk of bias even though some limitations were noted.Due to the very low to moderate level of certainty, neither an association nor a lack thereof between craniofacial morphology and pediatric OSA can be supported by these data.Fagundes NCF, Gianoni-Capenakas S, Heo G, Flores-Mir C. Craniofacial features in children with obstructive sleep apnea: a systematic review and meta-analysis. J Clin Sleep Med. 2022;18(7):1865-1875.