Factors associated with health-related quality of life (HRQOL) in adults with short stature skeletal dysplasias

Authors

Nitasha Dhiman, Virginia Tech Carilion School of Medicine, Roanoke
Alia Albaghdadi, Johns Hopkins University School of Medicine, Baltimore
Cheryl K. Zogg, Yale School of Medicine, New Haven
Meesha Sharma, Harvard T.H. Chan School of Public Health, Boston
Julie E. Hoover-Fong, The Johns Hopkins Children's Center, Baltimore
Michael C. Ain, The Johns Hopkins School of Medicine, Baltimore
Adil H. Haider, Harvard T.H. Chan School of Public Health, BostonFollow

Document Type

Article

Department

General Surgery

Abstract

Introduction: Numerous factors associate with health disparities. The extent to which such factors influence health-related quality of life (HRQOL) among adults with short stature skeletal dysplasias (SD) is unknown. In an effort to update and clarify knowledge about the HRQOL of adults with SD, this study aimed to quantify HRQOL scores relative to the American average and assess whether specific indicators are associated with lower scores.
Methods: Members (>18 years) of Little People of America were invited to complete an online survey assessing HRQOL using the SF-12 supplemented with indicator-specific questions. SF-12 components (Physical Component Summary, PCS; Mental Component Summary, MCS) were compared to the standardized national American mean. Scores were divided at the median to identify factors associated with lower scores using multivariable logistic regression, adjusting for age, gender, race, education, and employment.
Results: A total of 189 surveys were completed. Mean and median PCS and MCS were below the national mean of 50 (p < 0.001). Advancing decade of age corresponded to a significant decline in PCS (p < 0.001) but not MCS (p = 0.366). Pain prevalence was high (79.4%); however, only 5.9% visited a pain specialist. Significant factors for lower PCS included age >40 years (p = 0.020), having spondyloepiphyseal dysplasia congenita (SED) or diastrophic dysplasia relative to achondroplasia (p = 0.023), pain (p < 0.001), and "partial" versus "full" health insurance coverage (p = 0.034). For MCS, significant factors included a lack of social support (p = 0.002) and being treated differently/feeling stigmatized by health care providers (p = 0.022).
Conclusions: Individuals with SD face documented disparities and report lower HRQOL. Further research and interventions are needed to modify nuanced factors influencing these results and address the high prevalence of pain.

Comments

This work was published before the author joined Aga Khan University

Publication (Name of Journal)

Quality of life research

Recommended Citation

Dhiman, N., Albaghdadi, A., Zogg, C. K., Sharma, M., Hoover-Fong, J. E., Ain, M. C., Haider, A. H. (2017). Factors associated with health-related quality of life (HRQOL) in adults with short stature skeletal dysplasias. Quality of life research, 26(5), 1337-1348.
Available at: https://ecommons.aku.edu/pakistan_fhs_mc_surg_gen/150