Health-related quality of life and associated risk factors in patients with Multiple Osteochondromas: a cross-sectional study

Qual Life Res. 2024 May;33(5):1323-1334. doi: 10.1007/s11136-024-03604-4. Epub 2024 Mar 8.

Abstract

Purpose: To evaluate the health-related quality of life and associated risk factors for Multiple Osteochondromas patients.

Methods: A cross-sectional, observational study was conducted from May to December 2022 during the routine visit to the referral center for rare skeletal disorders. All patients with Multiple Osteochondromas aged ≥ 3 years were included. EuroQol 5-dimension questionnaires, and demographic, clinical, and surgical history data were collected. Descriptive statistics, Fisher's exact test, One-sample t-test, Spearman's correlation, and multiple linear and logistic regression were performed to analyze the data. Results are reported following STROBE guidelines.

Results: A total of 128 patients were included in the study, with a mean age of 14 [SD, 10] years. The mean EQ-5D Index Value was 0.863 [SD, 0.200] and the EQ-VAS was 84 [SD, 19] with a positive correlation between two scores [r = 0.541, p < 0.001]. Patients frequently referred problems in pain/discomfort [78.8%], anxiety/depression [50%], and usual activities [38.8%] dimensions. Increasing age was the common risk factor for health-related quality of life [p < 0.000], as well as Index Value and VAS scores were significantly lower in surgical patients [p = 0.001 and p < 0.001, respectively].

Conclusion: Increasing age and surgical procedures were found highly associated with reduced health-related quality of life in Multiple Osteochondromas patients. Our findings provide relevant information to support the establishment of patient-centered healthcare pathways and pave the way for further research into medical and non-medical therapeutic strategies for these patients.

Keywords: HRQoL; Healthcare pathway; Multiple Osteochondromas; PROs; Rare disease; Skeletal dysplasia.

Publication types

  • Observational Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Child, Preschool
  • Cross-Sectional Studies
  • Exostoses, Multiple Hereditary / psychology
  • Female
  • Humans
  • Male
  • Middle Aged
  • Quality of Life*
  • Risk Factors
  • Surveys and Questionnaires
  • Young Adult