Assessing Psychological Adjustment to Congenital Craniofacial Anomalies: An Illustration of Methodological Challenges

Abstract

Background:

Recognition of the challenges inherent in psychology research related to cleft lip and/or palate (CL/P) and other craniofacial anomalies (CFAs) is far from novel; yet these challenges continue to limit progress within the field. The aim of the present article was to illustrate these challenges by utilizing data extracted from 2 recent literature reviews pertaining to psychological adjustment within CL/P and CFA research.

Design:

Data relating to 148 CL/P and 41 CFA studies were extracted, summarized, and compared, using percentages, figures, and χ² calculations.

Results:

Comparable patterns were observed in both populations in relation to small sample sizes, wide age ranges, a reliance on data collection from single sites, and limited global coverage. Similarly, only one-third of all studies had used a comparison group, 42% of studies did not include the patient perspective, and fewer than 10% of studies had collected data longitudinally. Qualitative research was lacking across both populations, but particularly in relation to CFA. A higher proportion of CFA studies utilized validated measures and were less likely to exclude patients with additional anomalies. CFA studies most frequently focused on behavior and overall quality of life, while CL/P studies tended to investigate emotional well-being and social experiences.

Conclusions:

Findings illustrate the variability in research approaches, sampling, measurement, and analysis across both populations. There is a pressing need to address key methodological issues within craniofacial research and to examine the possible similarities and condition-specific differences between CL/P and other congenital craniofacial anomalies.