Vascular Malformations and Health-Related Quality of Life: A Systematic Review and Meta-analysis

Importance Patients with vascular malformations (VAMs) and vascular overgrowth syndromes have lower health-related quality of life (HRQoL) attributable to social stigmatization, poor mental health, severity, and pain. However, the factors that contribute to this decreased HRQoL are not clear. Objective To perform a systematic review and meta-analysis of studies that used validated HRQoL instruments to compare the HRQoL of persons with VAMs with the US general population. Data Sources A comprehensive search was performed in MEDLINE, Embase, PsycINFO, CINAHL, and Scopus from 1946 to March 31, 2017, with the consultation of an experienced librarian. Study Selection All VAM studies with validated HRQoL instruments published in the English language were included. Case reports, review articles, non–English-language publications, and studies about the development of new HRQoL instruments were not included. Data Extraction and Synthesis Two reviewers assessed studies’ eligibility and the risk of bias and performed data extraction. The meta-analysis was performed using the random-effects model. Comparisons of means were performed using the unpaired, 2-sample t test. Main Outcomes and Measures The outcome was HRQoL. Results Eleven studies met the inclusion criteria for a total of 692 patients with VAMs. Six studies (320 patients) were included in the meta-analysis, whereas 5 studies were included in the qualitative analysis (372 patients). Those with VAMs had lower 36-Item Short-Form Health Survey scores in bodily pain (mean difference, −11.87; 95% CI, −21.45 to −2.29; I2 = 92%; P = .02) and mental health (mean difference, −6.04; 95% CI, −11.55 to −0.52; I2 = 83%; P = .03) compared with the US general population. Conclusions and Relevance Patients with VAMs had increased pain and psychosocial distress compared with the US general population. Pain and psychological morbidity are associated with poorer HRQoL and may serve as indicators for quality of life.