Zolkwer, Morgan Baynes; (2025) Psychological impact and adjustment to rare birthmarks. Doctoral thesis (Ph.D), UCL (University College London).

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Abstract

Research into the psychological impact of rare birthmarks on children and their parents is in its infancy. Despite this, in many parts of the world assumptions of adverse psychological impact are frequently used to justify surgical interventions in children, to attempt to “improve” aesthetic appearance. Such assumptions usually involve more specific components, including that girls will suffer more adverse impact than boys, and that visible lesions on the face will confer more adverse impact than those elsewhere on the body. The lack of evidence also impacts resource allocation and the clinical management recommendations for supporting the psychological adjustment of this population. The aim of this research was therefore to investigate the psychological impact of rare birthmarks from multiple angles, to increase understanding of the risk and protective characteristics, and to start to formulate evidence-based clinical recommendations to support the adjustment of affected individuals and their families. Four complimentary studies were designed and conducted. The first two utilised a sequential exploratory mixed methods design to investigate parental adjustment to their child’s rare disease. The third used a battery of validated and purpose made questionnaires to measure the psychological impact of rare birthmarks on adolescents, and identify factors associated with adjustment. The final study used matched data from adolescents and their parents to investigate the concordance between adolescent self-reports and parent reports of adolescent adjustment, to explore the validity of parental reports which have previously been used as surrogates. These results were then compared to Dermatologists’ perspectives of the psychological impact of the same birthmarks, to investigate the validity of physicians’ preconceptions. Intrinsic to the research design was the inclusion of two clinically well-delineated but different conditions, congenital melanocytic naevi (CMN) and arteriovenous malformations (AVM), and a heterogenous group of other rare birthmarks to assess the generalisability of findings between different birthmark groups which are treated at the same specialist service. This work provides novel documentation of the experience of parenting a child with rare birthmarks, and the pivotal role of specialist care in parental adjustment. Findings indicate significant risk-factors for parental adjustment included uncertainty, concern about transitional stages, and parental stressor difficulty; significant protective-factors included parent age, parenting self-efficacy, and the parent-HCP relationship. These factors are measurable in a clinical setting, and the PhD proposes that these should be used to help identify those parents at greatest need of psychological support. The findings related to adolescent adjustment provide novel insights into the wide range of impacts that rare birthmarks have on patients and challenge the frequent assumptions that female sex and birthmark visibility have a negative impact. Significant risk-factors for adolescent adjustment to rare birthmarks instead included appearance orientation, self-reported physical discomfort, psychological impact of condition, and number of siblings. Strikingly, visibility of birthmarks was identified as a significant protective-factor, irrespective of age or sex. These data could have a major impact internationally on the early life decision-making of doctors and parents around the need for aesthetic surgery for birthmarks. They could also allow better identification of those at need of psychological support and form the basis of future therapeutic intervention to optimise adjustment. Major differences between the psychological impact of different rare birthmark diagnoses were not observed. Instead, the functional impact of rare birthmark conditions was found to be a better predictor of differences between adjustment. Analysis of concordance rates between parents, adolescents, and physicians documented pervasive misalignment between self-reports and parent-reports of psychological impact of birthmarks, calling into question the validity of previous studies which focus on parental reports. Furthermore, the impact of misalignment between self- and parent-reports in either direction is likely to be important for adjustment and will be pertinent to the design of future interventions. Data from physicians clearly confirmed the reliance on increased birthmark visibility as an indicator for psychology referral, which did not always align with the experiences of patients themselves. The identification of risk- and protective-factors associated with adjustment will be taken forward into clinical assessment to improve early identification of patients and parents at-risk of poor adjustment, to justify improved targeted care provision and to inform future research. Taken together, the findings will have a major impact on the understanding, assessment and management of children and adolescents with rare birthmarks, and their parents.

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