Cost-utility of a six-month programmed sports therapy (PST) in patients with haemophilia.

INTRODUCTION: Recurrent musculoskeletal haemorrhages in people with haemophilia (PwH) lead to restrictions in the locomotor system and, as a result, in physical performance, too. Due to its physical and psychological benefits, sport is increasingly re-commended for haemophilic patients. Evidence on the cost-effectiveness of sports therapy is still lacking. AIM: The aim of this study was to determine the cost-effectiveness of a 6-month programmed sports therapy (PST). METHODS: The cost-effectiveness of the 6-month PST was assessed from a societal perspective alongside a RCT using cost-utility analysis. The analysis included 50 PwH with moderate-to-severe haemophilia A and B and a training period over 6 months. The health-related quality of life was measured with the EuroQoL-domain questionnaire. Resource utilization was assessed by questionnaire before and after the intervention. A cost-effectiveness acceptability curve was constructed, and sensitivity analyses were performed. RESULTS: During the 6-month study period, mean adjusted total healthcare costs were lower (mean difference: -22 805 EUR; 95%-CI: -73 944-48 463; P = .59) and the number of QALYs was higher in the intervention group (mean difference: 0.3733; 95%-CI: 0.0014-0.0573; P = .04). The probability of an incremental cost-effectiveness ratio <50 000 EUR per QALY was 71%. The performed sensitivity analysis confirmed these results. CONCLUSION: Results showed that the PST is effective in terms of a significant gain of QALYs. Furthermore, results weakly indicate the potential of the PST to reduce healthcare costs. Future studies should expand the observation period to have a closer look at the influence of PST on lifetime costs.

RCT - subjective physical performance and quality of life after a 6-month programmed sports therapy (PST) in patients with haemophilia.

INTRODUCTION: Musculoskeletal bleedings lead to limitations in the locomotor system and consequently, in health-related quality of life (HRQoL) in patients with haemophilia (PwH). Sports therapy is increasingly recommended to improve their physical performance. Until today, randomised controlled studies investigating changes in physical performance in PwH are rare. AIM: This study investigates the impact of programmed sports therapy on the subjective physical performance and the HRQoL in PwH. METHODS: A randomised controlled trial (RCT) was conducted with a training intervention for over 6 months. For this purpose, 64 PwH with moderate (n = 5) or severe (n = 59) haemophilia A (n = 57) or B (n = 7) were randomised into two groups - intervention (IG) or control group (CG). The HRQoL was assessed with the SF-36 questionnaire and the disease-specific Haem-A-QoL before and after the intervention. The subjective physical performance was tested by the HEP-Test-Q. RESULTS: After the 6-month training intervention, PwH in the IG subjectively reported significant better 'endurance' (P = 0.000) in the HEP-Test-Q compared to the CG. In the SF-36, a significant difference in the domains 'general health perceptions' (P = 0.005) and 'mental health' (P = 0.001) was detected. The haemophilia-specific HRQoL questionnaire showed a significant improvement in the dimensions 'feeling' (P = 0.049), 'work' (P = 0.046) and 'family' (P = 0.040). CONCLUSION: In the first RCT evaluating the impact of a 6-month training intervention on the subjective perception of PwH, an increase in subjective physical performance and some domains of HRQoL was demonstrated in the IG. Specific sports therapy should be included into the comprehensive treatment under supervision and monitoring by experienced staff.

RCT of a 6-month programmed sports therapy (PST) in patients with haemophilia - Improvement of physical fitness.

UNLABELLED: Physical fitness is of major importance for patients with haemophilia (PwH) but is highly influenced by bleeding episodes. Although some cohort studies describe an improvement of physical fitness after training intervention, randomized controlled studies (RCT) in PwH are still rare. AIM: The aim of this study was to prove the enhancement of physical work capacity in PwH by programmed sports therapy (PST). METHODS: This RCT includes a training period over six months. Sixty-four PwH with moderate (n = 5) to severe (n = 59) haemophilia A (n = 57) and B (n = 7) were randomized into two groups - training intervention group (IG) and control group (CG) by block randomization. Complex strength measurements, joint score, coordination check and a 12-min walking test were carried out before and after training intervention. RESULTS: Significant differences were tested in M. triceps brachii (Δ+0.62 N kg(-1) ), biceps brachii (Δ+1.25 N kg(-1) ), latissimus dorsi (Δ+0.59 N kg(-1) ), rectus abdominis (Δ+0.51 N kg(-1) ), biceps femoris (right: Δ+0.68 N kg(-1) ; left: Δ+0.59 N kg(-1) ) and the quadriceps femoris (right: Δ+0.71 N kg(-1) ; left: Δ+0.55 N kg(-1) ) after intervention between the two groups (all p ≤ 0.003). Furthermore, an increase in distance (Δ+171.5 m) covered in the 12-min walking test (P = 0.011) was observed. Regarding one-leg stand, a significant improvement (P = 0.037) in the IG (Δ+2.2 s right leg) after intervention could be determined. CONCLUSION: For the first time, a study with a corresponding number of adult PwH in a RCT-design showed that programmed sport therapy with specific instructions over 6 months has a positive effect on physical performance of PwH, independent of constitution and disease process.

Pain sensitivity in patients with haemophilia following moderate aerobic exercise intervention.

BACKGROUND: Physical activity is influenced by pain and vice versa. Although studies recommend exercise therapy for patients with haemophilia (PwH), the influence of physical activity on the pain condition in PwH has not been investigated so far. AIM: Aim of this study was to examine the effect of a treadmill intervention with self-chosen velocity on the acute pain sensitivity in PwH. PATIENTS AND METHODS: Twenty PwH [aged 24-58 years, moderate (n = 3) to severe (n = 17) haemophilia A (n = 17) or B (n = 3)] and 20 control subjects (aged 26-61 years) were included in this study. Eighteen PwH and all controls completed a treadmill intervention for 30 min. Pressure pain thresholds (PPT) in Newton (N) were measured at both the knees, ankles and elbows, sternum and forehead before (pre) and immediately after walking (post). RESULTS: PwH and controls walked with comparable speed (mean speed in km h-1 ; PwH: 3.5, controls: 3.8), resulting in significantly different values of performance-related parameters such as heart rate (mean heart rate per minute; PwH: 102, controls: 86; P ≤ 0.01). Compared to baseline values, PPT remained unaltered at all landmarks in both groups after walking (e.g. pre/post in Newton; knee right: PwH: 63.1/63.0, controls: 93.8/93.8; left knee: PwH: 62.1/62.7, controls: 90.0/93.4), indicating a non-increasing pain condition. CONCLUSION: Findings of unaltered PPT following moderate aerobic exercise showed initial evidence that PwH are able to perform an endurance exercise with self-chosen velocity for 30 min as recommended, without increasing the acute pain condition. By doing so, PwH can benefit from the positive effects of endurance exercise.

Complex strength performance in patients with haemophilia A. Method development and testing.

UNLABELLED: The aim of this study was to develop a complex strength measurement method and to apply this new method for the first time in patients with haemophilia (PwH). 20 PwH with severe haemophilia A and 20 controls were included into the study. All subjects completed ten measurements of maximum isometric strength. Furthermore, the 20 control subjects completed re-test-measurements to evaluate the method. As a result, the method showed a high reliability (ICC 0.764 to 0.934). Between the two groups significant reductions in PwH between -(19-35%) were detected, regarding the relative force of the M. triceps brachii (-19%; p = 0.008), M. biceps brachii (-19%; p = 0.031), M. latissimus dorsi (-17%; p = 0.019), M. biceps femoris right (-20%; p = 0.036) and M. quadriceps femoris (right: -29%; p = 0.004; left: -35%; p = 0.002). No differences were found for M. rectus abdominis and in the hand strength. Thus, there is no general deficit in the muscle strength in PwH. The most obvious deficits exist in the upper and lower extremities and in the back muscles. CONCLUSION: PwH should carry out complex muscle strength training and integrate it early into a comprehensive treatment concept.

Changes in phosphocreatine concentration of skeletal muscle during high-intensity intermittent exercise in children and adults.

PURPOSE: The aim of the present study was to test the hypotheses that a greater oxidative capacity in children results in a lower phosphocreatine (PCr) depletion, a faster PCr resynthesis and a lower muscle acidification during high-intensity intermittent exercise compared to adults. METHODS: Sixteen children (9.4 ± 0.5 years) and 16 adults (26.1 ± 0.3 years) completed a protocol consisting of a dynamic plantar flexion (10 bouts of 30-s exercise at 25 % of one repetition maximum separated by 20-s recovery), followed by 10 min of passive recovery. Changes of PCr, ATP, inorganic phosphate, and phosphomonoesters were measured by means of (31)Phosphorous-magnetic resonance spectroscopy during and post-exercise. RESULTS: Average PCr (percentage of [PCr] at initial rest (%[PCr]i)) at the end of the exercise (adults 17 ± 12 %[PCr]i, children 38 ± 17 %[PCr]i, P < 0.01) and recovery periods (adults 37 ± 14 %[PCr]i, children 57 ± 17 %[PCr]i, P < 0.01) was significantly lower in adults compared to children, induced by a stronger PCr decrease during the first exercise interval (adults -73 ± 10 %[PCr]i, children -55 ± 15 %[PCr]i, P < 0.01). End-exercise pH was significantly higher in children compared to adults (children 6.90 + 0.20, -0.14; adults 6.67 + 0.23, -0.15, P < 0.05). CONCLUSIONS: From our results we suggest relatively higher rates of oxidative ATP formation in children's muscle for covering the ATP demand of high-intensity intermittent exercise compared to adults, enabling children to begin each exercise interval with significantly higher PCr concentrations and leading to an overall lower muscle acidification.