Timmer M1, Veenhof C2, de Kleijn P1, de Bie R3, Schutgens R1, Pisters MF4
1UMC Utrecht, van Creveldkliniek, Utrecht, Netherlands, 2UMC Utrecht, Rehabilitation, Physical Therapy Science and Sport, Utrecht, Netherlands, 3Maastricht University, Department of Epidemiology, Maastricht, Netherlands, 4UMC Utrecht, Center for Physical Therapy Research and Innovation in Primary Care, Utrecht, Netherlands
Background: Haemophilia is an inherited genetic disorder that impairs the body´s ability to make blood clots. Intra-articular bleeding is the hallmark of haemophilia. Joint bleeds cause haemophilic arthropathy (HA), which is characterized by joint pain, limitations in range of motion and muscle atrophy. Consequently, adults with severe haemophilia walk and run less and sit more than healthy adults. Daily movement behaviour is defined as a combination of day time sedentary behaviour, physical activity and sleep. Previous studies investigate movement behaviour in persons with haemophilia using a single parameter for movement behaviour or have analyzed several parameters separately, therewith ignoring that daily movement behaviour cannot be measured adequately in this way. Moreover, sedentary behaviour and physical activity are independent predictors of health outcome. Identifying subgroups of persons with haemophilia with similar movement behaviour gives a more complete insight in movement behaviour in relation to clinical characteristics.
Purpose: The aim of this study was to identify phenotypes of movement behaviour within persons with haemophilia and compare clinical characteristics between phenotypes of movement behaviour.
Methods: Movement behaviour was measured using the Activ8 activity monitor. The Activ8 has been validated and can distinguish between: lying down, sitting, standing, walking, running and cycling. Clinical characteristics included age, severity of haemophilia, joint health (Hemophilia Joint Health Score), self-perceived limitations in activities (Haemophilia Activity List) walking speed (40 meters self-paces walk test) and pain (Numerical pain Rating Score). Hierarchical cluster analysis was used to identify phenotypes. Clustering variables included 7 parameters of movement behaviour; sitting, standing, walking, biking, running, frequency of active bouts and length of active bouts. Clinical characteristics were compared between the identified clusters with the Kruskall-Wallis test.
Results: A total of 105 persons with haemophilia (70% severe haemophilia; median age 43 years [30.0-54.0]) were included in the study. Cluster analysis identified 3 clusters; “sedentary” (57%), “bikers and runners” (22%) and “walkers” (20%). Persons with haemophilia in the “sedentary” cluster had the lowest HAL score, the “walkers” cluster scored lower than the “bikers & runners” cluster (median [IQR] 74.5 [61.0-92.5], 85.6 [76.7-93.9] and 90.3 [69.1-100] respectively, p=0.04). A trend was found towards lower HJHS score in persons with haemophilia in the “bikers and runners” cluster compared to the “walkers” and the “sedentary” (median [IQR] 4.0 [1.0-29.0], 15.5 [5.8-25.8] and 16.0 [7.5-34.0] respectively, p=0.07). No differences were found between the clusters based on age, NRS and self-paced walking speed (p>0.05).
Conclusion(s): Three phenotypes of movement behaviour were identified in persons with haemophilia. The majority of persons with haemophilia was sedentary. The “bikers and runners” had a better joint health and experienced less limitations in activities than the “walkers” and “sedentary” persons. The “walkers” perceived fewer limitations in activities than the “sedentary” but had comparable joint health.
Implications: The results of this study subscribe the importance of assessment of movement behaviour as a total, as opposed to using a single parameter, in clinical practice and in scientific research. Furthermore, the results indicate that the regular walking and less sitting is feasible and beneficial for persons with haemophilic arthropathy.
Keywords: haemophilia, movement behaviour, phenotypes
Funding acknowledgements: This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Purpose: The aim of this study was to identify phenotypes of movement behaviour within persons with haemophilia and compare clinical characteristics between phenotypes of movement behaviour.
Methods: Movement behaviour was measured using the Activ8 activity monitor. The Activ8 has been validated and can distinguish between: lying down, sitting, standing, walking, running and cycling. Clinical characteristics included age, severity of haemophilia, joint health (Hemophilia Joint Health Score), self-perceived limitations in activities (Haemophilia Activity List) walking speed (40 meters self-paces walk test) and pain (Numerical pain Rating Score). Hierarchical cluster analysis was used to identify phenotypes. Clustering variables included 7 parameters of movement behaviour; sitting, standing, walking, biking, running, frequency of active bouts and length of active bouts. Clinical characteristics were compared between the identified clusters with the Kruskall-Wallis test.
Results: A total of 105 persons with haemophilia (70% severe haemophilia; median age 43 years [30.0-54.0]) were included in the study. Cluster analysis identified 3 clusters; “sedentary” (57%), “bikers and runners” (22%) and “walkers” (20%). Persons with haemophilia in the “sedentary” cluster had the lowest HAL score, the “walkers” cluster scored lower than the “bikers & runners” cluster (median [IQR] 74.5 [61.0-92.5], 85.6 [76.7-93.9] and 90.3 [69.1-100] respectively, p=0.04). A trend was found towards lower HJHS score in persons with haemophilia in the “bikers and runners” cluster compared to the “walkers” and the “sedentary” (median [IQR] 4.0 [1.0-29.0], 15.5 [5.8-25.8] and 16.0 [7.5-34.0] respectively, p=0.07). No differences were found between the clusters based on age, NRS and self-paced walking speed (p>0.05).
Conclusion(s): Three phenotypes of movement behaviour were identified in persons with haemophilia. The majority of persons with haemophilia was sedentary. The “bikers and runners” had a better joint health and experienced less limitations in activities than the “walkers” and “sedentary” persons. The “walkers” perceived fewer limitations in activities than the “sedentary” but had comparable joint health.
Implications: The results of this study subscribe the importance of assessment of movement behaviour as a total, as opposed to using a single parameter, in clinical practice and in scientific research. Furthermore, the results indicate that the regular walking and less sitting is feasible and beneficial for persons with haemophilic arthropathy.
Keywords: haemophilia, movement behaviour, phenotypes
Funding acknowledgements: This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Topic: Musculoskeletal; Rheumatology; Human movement analysis
Ethics approval required: No
Institution: UMC Utrecht
Ethics committee: UMC Utrecht
Reason not required: Data was used from medical health record
All authors, affiliations and abstracts have been published as submitted.
