title: Factors associated with health-related quality of life (HRQOL) in adults with short stature skeletal dysplasias.
abstract: 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.
  
  
  
  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.
  
  
  
  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).
  
  
  
  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.
authors.affiliation: Virginia Tech Carilion School of Medicine, Roanoke, VA, USA.
authors.forename: Nitasha
authors.initials: N
authors.lastname: Dhiman
authors.affiliation: Department of Surgery, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
authors.forename: Alia
authors.initials: A
authors.lastname: Albaghdadi
authors.affiliation: Yale School of Medicine, New Haven, CT, USA.|Department of Surgery, Center for Surgery and Public Health, Brigham & Women's Hospital, Harvard Medical School, Harvard T.H. Chan School of Public Health, Boston, MA, USA.
authors.forename: Cheryl K
authors.initials: CK
authors.lastname: Zogg
authors.affiliation: Department of Surgery, Center for Surgery and Public Health, Brigham & Women's Hospital, Harvard Medical School, Harvard T.H. Chan School of Public Health, Boston, MA, USA.
authors.forename: Meesha
authors.initials: M
authors.lastname: Sharma
authors.affiliation: Greenberg Center for Skeletal Dysplasias, McKusick-Nathans Institute for Genetic Medicine, The Johns Hopkins Children's Center, Baltimore, MD, USA.
authors.forename: Julie E
authors.initials: JE
authors.lastname: Hoover-Fong
authors.affiliation: Department of Orthopaedic Surgery, The Johns Hopkins School of Medicine, Baltimore, MD, USA.
authors.forename: Michael C
authors.initials: MC
authors.lastname: Ain
authors.affiliation: Department of Surgery, Center for Surgery and Public Health, Brigham & Women's Hospital, Harvard Medical School, Harvard T.H. Chan School of Public Health, Boston, MA, USA. ahhaider@partners.org.|Department of Surgery, Brigham & Women's Hospital, Kessler Director Center for Surgery and Public Health, 1620 Tremont Street, One Brigham Circle, 4th Floor, Suite 4-020, Boston, MA, 02120, USA. ahhaider@partners.org.
authors.forename: Adil H
authors.initials: AH
authors.lastname: Haider
mesh_terms: D000328:Adult; D000368:Aged; D000369:Aged, 80 and over; D005260:Female; D006801:Humans; D008297:Male; D008875:Middle Aged; D010009:Osteochondrodysplasias / Q000151:congenital* / Q000523:psychology; D018434:Sickness Impact Profile; D011795:Surveys and Questionnaires; D055815:Young Adult
keywords: Disparities; Dwarfism; Health-related quality of life; Skeletal dysplasia