Sagittal Synostosis and Its Association With Cognitive, Behavioral, and Psychological Functioning

Autor: Diana Dorstyn, David J. David, Rachel M. Roberts, Ben G Grave, A. J. Osborn
Jazyk: angličtina
Rok vydání: 2021
Předmět:
Zdroj: JAMA Network Open
ISSN: 2574-3805
Popis: Key Points Question Do individuals with sagittal synostosis experience greater cognitive, behavioral, and/or psychological difficulties, compared with their healthy peers? Findings In this meta-analysis, data from 32 independent studies involving a pooled sample of 1422 children and adults and examining 16 domains were analyzed. Overall, results were highly variable, with individual study results ranging from moderately positive findings for global development, where the children with sagittal synostosis were functioning at better levels than their peers, to large negative differences between groups for general cognition. Meaning These findings suggest that some children with sagittal synostosis experience negative outcomes; thus, ongoing monitoring and referral to support services as required are critical.
This meta-analysis reviews research on individuals with sagittal synostosis to determine whether, and to what extent, they experience cognitive, behavioral, and psychological difficulties compared with their healthy peers or normative data for each measure.
Importance Findings on the cognitive, behavioral, and psychological functioning of individuals with sagittal synostosis (SS) are highly disparate, limiting their clinical utility. Objective To identify and review research on individuals with SS and to determine whether, and to what extent, they experience cognitive, behavioral, and psychological difficulties compared with their healthy peers or normative data for each measure. Data Sources PubMed, Scopus, Embase, and PsycINFO were searched through January 2021 with no date restrictions. Scopus citation searches and manual checks of the reference lists of included studies were conducted. Study Selection Studies included participants of any age who had received a diagnosis of single-suture (isolated or nonsyndromic) SS or scaphocephaly and who had been assessed on cognitive, behavioral, and psychological outcomes. Data Extraction and Synthesis Data were independently extracted by 2 reviewers. Case-control outcomes (individuals with SS vs healthy peers or normative data) were compared using random-effects models with 3 effect sizes calculated: weighted Hedges g (gw), odds ratios (ORs), and mean prevalence rates. This study follows the Meta-analysis of Observational Studies in Epidemiology (MOOSE) reporting guidelines. Main Outcomes and Measures Findings were categorized by surgical status (conservatively managed, presurgery, postsurgery, or combined); domain (eg, general cognition); type of cognitive, behavioral, or psychological measure (objective or subjective); and source of comparison data (peers or normative data). Results Data from 32 studies, involving a pooled sample of 1422 children and adults with SS (mean [SD] age at assessment, 5.7 [6.6] years; median [interquartile range] age, 3.3 [0.5-10.3] years), were analyzed. Data on sex were available for 824 participants, and 642 (78%) were male. Individual study results varied substantially. Objective tests identified significant moderate group differences on 3 of 16 examined domains: presurgical motor functioning (3 studies; gw = −0.42; 95% CI, −0.67 to −0.18; P
Databáze: OpenAIRE