Abstract

OBJECTIVE: Parents of patients with inherited metabolic diseases (IMDs) report impaired well-being and a heavy burden in managing their children’s disease. Until now, studies focused more on general quality of life, while research on more specific psychological aspects is needed to provide more tailored care. Nevertheless, systematic studies on this topic are not present in the literature. The object of this systematic review is to investigate the instruments used to evaluate stress and coping in parents of patients with different IMDs.

MATERIALS AND METHODS: The literature search was performed on PubMed and Scopus. Inclusion criteria: the presence of instruments exploring stress and/or coping in parents of patients with IMDs, diagnosis of IMD, publication in peer-reviewed journals and English language. Exclusion criteria: sample mostly composed of caregivers different from parents, dissertations, reviews/meta-analysis, full-text not available. Data extracted from the studies: authors, year, country, disease, number and age of patients and parents, design and aim of the study, name and characteristics of instruments for parental stress and/or coping.

RESULTS: Among the included studies (n=39) reported, the use of quantitative and qualitative instruments were different for stress and coping evaluation: almost all studies on parental stress used questionnaires to collect quantitative data, such as standardized questionnaires, instead a larger proportion of studies on coping employed qualitative data (i.e., ad hoc questionnaires and interviews).

CONCLUSIONS: Qualitative and quantitative instruments can be used to investigate stress and coping in parents of patients with IMDs. Investigating these specific psychological aspects in parents is pivotal in research and clinical practice to provide more tailored family-centered care of IMDS, characterized by the wide variability of symptoms and parental burden.