Problem joints are associated with worse health-related quality of life (HRQoL) and work-productivity loss in adults with hemophilia, according to a study published in Haemophilia.
“Recurrent hemarthrosis causes irreversible joint damage in people with hemophilia,” the researchers wrote in their report. “In order to address both patient-centric and research-oriented needs for more precision, the ‘problem joint’ concept was developed and defined as chronic joint pain and/or limited range of movement due to compromised joint integrity.”
In the study, the investigators aimed to quantify the humanistic burden of problem joints in people with hemophilia to validate of the problem joint outcome measure.
They used multivariable regression models to evaluate the relationship between problem joints and HRQoL and work-productivity loss using data from 3 population studies, including the Cost of HaEmophilia: a Socioeconomic Survey (CHESS) II and CHESS US+ in adults and CHESS-Paeds in children and adolescents.
The CHESS II sample included 292 people with hemophilia with HRQoL data (mean age, 38.6 years). Of those, 39% reported ≥1 problem joints and 61% reported no problem joints. Work-productivity loss data was available for 134 of these individuals.
The CHESS US+ population included 345 people with hemophilia with HRQoL data (mean age, 35 years). Of those, 43% reported ≥1 problem joints, while 57% reported no problem joints. Work-productivity loss data was available for 239 of these individuals.
The CHESS-Paeds population included 198 people with hemophilia aged 4-17 years (mean age, 11.5 years). Of those, 19% reported ≥1 problem joints, while 81% reported no problem joints. Only HRQoL data was available for these individuals.
The researchers found the presence of problem joints was associated with worse HRQoL in both the CHESS II and CHESS US+ populations (P <.001 for both). They observed no significant association between problem joints and HRQoL in the CHESS-Paeds population.
The team found upper body problem joints were significantly correlated with work-productivity loss in the CHESS II population (P <.05) and ≥1 problem joints or upper and lower body problem joints were significantly correlated with work-productivity loss in the CHESS US+ population (P <.05 for both).
“This study has shown that an increase in the number of [problem joints] was associated with an increasing humanistic burden, that is, worse HRQoL and larger work productivity loss, in [people with hemophilia] of all ages, types and severities across Europe and United States,” concluded the researchers.
They added, “The patient-centric [problem joint] concept of hemophilia-related chronic damage could be important in both clinical and health policy assessments of joint health and treatment effectiveness in order to account for the [people-with-hemophilia] perspective in this lifelong debilitating condition.”
Disclosure: This research was supported by BioMarin and uniQure. Please see the original reference for a full list of disclosures.
Burke T, Rodriguez-Santana I, Chowdary P, et al. Humanistic burden of problem joints for children and adults with haemophilia. Published online December 27, 2022. Haemophilia. doi:10.1111/hae.14731