Aquatic Physiotherapy Group Aquatic Physiotherapy Level 3 Course Literature Review The Effectiveness of Aquatic Physiotherapy in Patients with Haemophilia: A Review of the Literature Nicola Blum Senior Physiotherapist Victorian Paediatric Rehabilitation Service Monash Children’s Hospital Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ The Effectiveness of Aquatic Physiotherapy in Patients with Haemophilia: A review of the literature. Nicola Blum Senior Physiotherapist Victorian Paediatric Rehabilitation Service Monash Children’s Hospital _____________________________________________________________________________________ 1 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ Abstract Haemophilia and its secondary complications can have a detrimental impact on the quality of life of its sufferers. Aquatic based interventions are often recommended for these patients because of the therapeutic benefits water based activities offer. Evidence suggests aquatic physiotherapy could be beneficial and effective for haemophilia patients especially following bleeds. This literature review investigates the evidence of aquatic exercise therapy in a haemophilia population to 1) determine the role of this intervention and to 2) determine its effective dosage. A literature review was undertaken using specific search terms and after an inclusion and exclusion process, four articles fit the criteria. Within these studies, participants’ range of motion (ROM), strength, pain and aerobic capacity was measured. It was noted in these studies, that ROM, strength, pain and aerobic capacity all improved within an aquatic intervention. However these results must be taken with caution, as the studies were of poor quality with poor methodological rigour and potential bias. This review highlights there is a lack of supporting evidence for aquatic physiotherapy in patients with haemophilia and further research into this area is indicated especially to determine the best practice guidelines and an effective dosage of such an intervention. Keywords: haemophilia, aquatic physiotherapy, aquatic therapy, hydrotherapy, water therapy, water exercise. Introduction Haemophilia, a congenital genetic disorder (Kargarfard, et al., 2013) is a result of an inherited recessive trait in the X chromosome (Souza, et al., 2011). It occurs in sufferers as a result of a deficiency in coagulation factor VIII (Haemophilia A), and IX (Haemophilia B) (CuestaBarriuso, et al., 2013; Souza, et al., 2011). Haemophilia A is the most common form of Haemophilia with an estimated 90% percent of all cases (Souza, et al., 2011). In Australia, Haemophilia is estimated to affect approximately 3000 people who are mostly males (Haemophilia Foundation Australia, 2014). However, whilst very rare, females can also be affected (Haemophilia Foundation Australia, 2014). A study by Kargarfard, et al (2013) conducted in Iran reported as high as one in every 10 000 newborn has Haemophilia A. _____________________________________________________________________________________ 2 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ There are three types of Haemophilia depending on the percentage of the blood clotting factor (Cuesta-Barriuso, et al., 2013). The smaller the percentage of blood clotting factor, the more severe the form of haemophilia. The three types are seen below: • Mild Haemophilia (>5-40% FVIII/FIX) with bleeding during surgical procedures or from severe injuries, • Moderate Haemophilia (1-5% FVIII/FIX) with bleeding from slight injuries, • Severe Haemophilia (<1% FVIII/FIX) characterised by spontaneous bleeding. Joint bleeds, also known as a haemoarthrosis, is the most prevalent and degenerative complication in patients with haemophilia. It is estimated that 80% of bleeds occur as a haemoarthrosis (Rodrigues-Mechan, 1996). The symptoms of a haemoarthrosis include severe pain, swelling and inflammation, increased skin temperature, reduced range of motion, muscle atrophy as well as synovial involvement (Cuesta-Barriuso, et al., 2013; Kargarfard, et al., 2013). Without the proper treatment, a haemoarthrosis can lead to capsular and tendon contractures of the affected joints attributing to detrimental consequences of the musculoskeletal system and the function of haemophilia patients (Cuesta-Barriuso, et al., 2013). Furthermore, in haemophilia sufferers, chronic arthropathy is also a major cause of morbidity (Cuesta-Barriuso, et al., 2013). The elbow, knee and ankle are the most common joints involved in a haemoarthrosis however the hip and shoulder can also be affected, though infrequently (Rodrigues-Mechan, 1996). Furthermore, a joint that is affected by several bleeds within 3-6 months is susceptible to further articular degeneration (Jansen, et al., 2008) and may lead down a negative path to haemophilic arthritis, with a similar presentation to that of rheumatoid arthritis (Mazloum, et al., 2014). Presently, haemophilia sufferers are often treated with replacement therapy. However prior to the availability of factor VIII and IX, treatment included bed rest, ice, splinting and/or braces and patients who did not have replacement therapy may have had up to five damaged joints by the age of 20 years (Heijnen, 2008). However with the current improvements in medical advances, such as replacement therapy, bleeding is reduced and its secondary complications such as haemoarthropathies can be prevented (Heijnen, 2008). _____________________________________________________________________________________ 3 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ Alongside replacement therapy, regular physical activity is important in patients with haemophilia in preventing further damage and injury to their joints especially in patients who have haemoarthroses (Heijnen, 2008). Souza, et al., (2011) state the importance of physical activity for patients with haemophilia for the benefits of improving strength and skeletal muscle cross- sectional area, flexibility, and decreased body fat. However, in patients where exercising and walking on land may exacerbate pain, further limit range of motion (ROM) and increase swelling or inflammation, exercise undertaken in water provides an effective medium to exercising on land, thus providing more benefits over a land based program. Mazloum, et al., (2014) report that the effect of buoyancy and unloading the lower extremities, encourages patients to exercise more effectively, leading to improved outcomes. Heijnen, (2008) supports the use of aquatic physiotherapy for haemophilia patients who have painful or stiff joints especially in the early stages of rehabilitation following a haemoarthrosis, muscle bleed or chronic arthropathy. Furthermore, Heijnen, (2008) reports aquatic physiotherapy can help patients who have chronic synovitis, bridging the gap of land based mobilisation and following long periods of bed rest or weaning from a splint. This review investigates the current evidence of water based therapy programs in patients with haemophilia and to determine what improvements can be achieved with this intervention. Methods To obtain the articles, five databases were systemically searched (PubMed, Medline, CINAHL, PEDro and Cochrane). The keywords as can be seen in Table 1 were haemophilia and aquatic physiotherapy or aquatic therapy or hydrotherapy or water therapy or water exercise. Articles were included if in the abstract they were pertaining to aquatic physiotherapy or word derivative equivalent alone (aquatic physiotherapy, aquatic therapy, hydrotherapy, water therapy, water exercise) and with land based physiotherapy in a haemophilia population. They were excluded if they were not in English. The process can be seen in figure 1. There were no date limits placed on the databases. Each article was critically appraised using the PEDro scale as can be seen in table 2. _____________________________________________________________________________________ 4 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ Table 1. Keywords used in search strategy Population Haemophilia (AND) Intervention aquatic physiotherapy (OR) aquatic therapy (OR) hydrotherapy (OR) water therapy (OR) water exercise Articles retrieved n= 68 PubMed- 54 Pedro 1 Cochrane 5 Medline- 4 CINAHL- 4 Review of title and abstract • Removal of duplicates and systematic reviews Included if: • Published in English • Study in a Haemophilia population • Involved in aquatic physiotherapy or equivalent intervention Eligible n= 4 Figure 1. The process of searching for articles. _____________________________________________________________________________________ 5 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ Table 2. Break down of the four studies into their methodological parts. Study design Garcia, et al., (2009) Pre-test posttest Mazloum, et al., (2014) Kargarfard, et al., (2013) Vallejo, et al., (2010) Semi experimental pretest post-test Pre-test post-test 4 4 Level of evidence 4 Supervised quasiexperimental and perspective with non- randomised pre-test- post-test control group 4 Eligibility criteria were specified Subjects were randomly allocated to groups (in a crossover study, subjects were randomly allocated an order in which treatments were received) Allocation was concealed No Yes No Yes No No Yes No No No No No Groups were similar at baseline regarding the most important prognostic indicators There was blinding of all subjects There was blinding of all therapists who administered the therapy There was blinding of all assessors who measured at least one key outcome Measures of at least one key outcome were obtained from more than 85% of the subjects initially allocated to groups All subjects for whom outcome measures were available received the treatment or control condition as allocated or, where this was not the case, data for at least one key No Yes No No No No No No No No No No No No No No Yes Yes Yes Yes Yes Yes Yes Yes _____________________________________________________________________________________ 6 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ outcome was analysed by “intention to treat” The results of betweenYes group statistical comparisons are reported for at least one key outcome The study provides both No point measures and measures of variability for at least one key outcome Total of Yes 3 Yes Yes Yes Yes Yes Yes 6 5 5 Results As can be seen in table 3 and table 4 below, the search yielded four studies for review. Of these, three studies investigated the effect on warm water exercise on joint ROM (Table 3). These studies were by Garcia, et al., (2009), Kargarfard, et al., (2013), and Mazloum, et al., (2014). Furthermore Mazloum, et al., (2014) investigated pain and Kargarfard, et al., (2013) looked at knee flexion and extension strength in their studies. In addition a study by Vallejo, et al., (2010) investigated the effects of training in motor performance in patients with haemophilia (Table 4). Table 3. Review of articles investigating focusing on ROM. ROM Article Participants Program intervention Garcia, et al., (2009) 9 children with haemophilia and arthropathies between 5 and 13 years of age. 9 children with no pathology. 30 minute program consisting of: 1. Vertical cycling in suspension at 2.10m depth with a floatation device under the Mazloum, et al., (2014) 40 patients Therapeutic exercise13 Hydrotherapy- 14 Control- 13 Not specified. Kargarfard, et al., (2013) 20 males with moderate haemophilia- 10 in experimental group and 10 in control group 40-60 minutes of water exercise therapy consisting of: • Warm upProgressive aerobic _____________________________________________________________________________________ 7 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ armpits for 10 minutes. 2. Controlled breathing for 3 minutes. 3. Transverse, sagittal and longitudinal rotation control through games for 10 minutes. 4. Activities for buoyancy, balance and immobility control for 7 minutes. Program frequency Program duration Program provided by Is there a control group? Comparison to land intervention Could program be replicated? Outcome measures 2 times per week Not specified. Not specified Not specified 4 weeks Not specified activity 5074% of maximal heart rate. Activity not specified. • Main plan- 10 simple movements in the water – 5 for the upper limb and 5 for the lower limb. For a duration of 1 minute each of maximal power or for patients who were experiencing pain for as far as tolerable. • Cool down- 5 minutes. Flexibility movement however not specified. Exercises were progressed by increasing time and quality of the exercise. 3 times per week 8 weeks Not specified No. Yes Yes No. Yes No No. No. No. Elbow, knee and ankle ROM- measuring the arch of active articular Pain- VAS. Knee Flexion and extension ROM. Elbow flexion and extension ROM. Ankle dorsiflexion _____________________________________________________________________________________ 8 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ movement at each joint. Adherence Long term follow up Not specified. No. Not specified. No. and plantarflexion ROM. Knee extension and flexion ROM strength. Not specified. No. Table 4. Research into the effect of aquatic training on motor performance by Vallejo, et al., (2010). Motor performance Article Vallejo, et al., (2010) Participants 13 patients. Program 1 hour Aquatic trainingintervention • Warm up- 50m crawl or back stroke, running on the spot in the shallow end of the pool and circles forward and backward of the hip and shoulder at shoulder depth. • Main section- 2 blocks: o 1. muscular endurance and muscle strengthening Muscular endurance- 2 sets of 20 repetitions per exercise with active 30sec pauses (slow running on spot)• Flexion- extension of shoulder • Abduction- adduction of shoulder • 45° Abduction- adduction of shoulder in frontal plane • Flexion- extension of elbow. Muscle strengthening- 2 30 second sessions per exercise with a relaxation time 2 minutes where balance exercises were performed. • Horizontal and vertical traction movements at the participants’ maximal performance speed. o 2. Aerobic capacity which was extensive and continuous and was made up of 3 phases with 2 levels within each phase which was progressed by an increase in each participant’s maximal heart rate. Exercises were performed for a length of 50m and repeated 4 times for a duration of 20 minutes. • Backstroke of legs and arms. • Front crawl with arm movements. • Full arm movements in either backstroke or front crawl as selected by the participants. • Swimming backwards • Relaxation- Stretching exercises of the upper and lower limbs- 2 sets of 8 _____________________________________________________________________________________ 9 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ Program frequency Program duration Program provided by Program design Is there a control group? Comparison to land intervention Could program be replicated? Outcome measures Adherence Long term follow up exercises lasting 30 seconds each. 3 times per week. 9 weeks Not specified Pre-test post-test No. No. Yes. Oxygen uptake, relative oxygen uptake, Carbon dioxide, respiratory quotient, heart rate and the distance ran in the Cooper test. 4 abandoned study. No. These studies above do support an aquatic therapy intervention for improvements in ROM and strength of affected joints, improving motor performance and reducing pain. The evidence reveals that ROM does improve with aquatic therapy intervention. Garcia, et al., (2009) noted that in the group of patients with haemophilia, there was an improvement in the ankle and knee ROM, however no change in the elbow ROM (N.B. statistical significance was not reported). They suggest that these improvements may be due to the properties of the water including buoyancy, hydrostatic pressure as well as the thermodynamics of the heated water (Garcia, et al., 2009). In contrast, Kargarfard, et al., (2013) reported on a significant improvement in the ROM of all joints investigated on: left and right knees, ankles and elbow joints. Interestingly, their control group revealed that with a lack of intervention, the affected joints of patients with haemophilia will worsen highlighting the vital need for intervention in this population. Mazloum, et al., (2014) noted a significant improvement in knee flexion and extension ROM in both the land and aquatic exercise groups following intervention but no difference between them. _____________________________________________________________________________________ 10 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ Kargarfard, et al., (2013) as previously outlined above also investigated knee flexion and extension strength prior to and following a water based program. They reported an improvement in the power in knee extension and knee flexion of both knee joints following the intervention. In this literature review, this was the only study to investigate strength of any joint. To date no other studies have investigated strength with an aquatic intervention in haemophilia patients. Mazloum, et al., (2014) as outlined previously also looked at pain intensity when comparing a 4 week land based exercise intervention group with aquatic exercise intervention group compared to a control group. They reported there was an improvement in both the land and aquatic exercise groups when compared to the control, with a bigger improvement in the aquatic exercise therapy compared to the land exercise group (N.B. statistical significance was not reported). Vallejo, et al., (2010) reported that there is an improvement after an aquatic training intervention on patients who had haemophilic arthropathies. Vallejo, et al., (2010) measured the distance covered in the Cooper test, the relative oxygen uptake, carbon dioxide, respiratory quotient and heart rate. The Cooper test, a 12 minute test where the participants runs as far as able, is highly correlated with the VO2Max (Quinn, 2014). Compared to the results before the intervention, there was a significant difference in VO2 and relative VO2, CO2, respiratory quotient and the distance covered in the Cooper test. However there was no difference noted in the heart rate before and after the training. Discussion Of the four articles yielded, improvements post aquatic based intervention were seen in ROM in the elbow, ankle and knee, in knee extension and flexion strength, in motor performance and with a reduction in pain. This is of important clinical significance as patients undertaking aquatic physiotherapy programs may have improvements in their ROM and strength of their affected joints, improvements in their aerobic capacity as well as a reduction in their pain levels and larger improvements in pain over patients undergoing land based interventions. These improvements could ultimately improve their physical activity and participation leading to improvements in the quality of life, however further research is required to support this. As all of the studies were of poor (level 4) quality of evidence on the PEDro scale with small group sizes, _____________________________________________________________________________________ 11 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ had poor methodological rigour as well as potential bias, caution needs to be taken when examining the results. There were several pitfalls in the methodology of these studies. Among these were a lack of randomisation in patient groups, lack of comparison to a control or land based programs, poor comparisons to normal populations and a lack of long term follow up. The studies by Garcia, et al., (2009) and Vallejo, et al., (2010) had no control groups nor was there a land based group comparison for these studies and the study by Kargarfard, et al., (2013). In contrast, the study by Mazloum, et al., (2014) did have a control group and did compare to a land based intervention. Furthermore the study by Garcia, et al., (2009) unjustly compared haemophilia patients to a group of patients with no pathology or arthropathy. Of the four studies reviewed, only one study which was undertaken by Kargarfard, et al., (2013), randomly allocated the subjects. This lack of randomisation in the studies could have potentially led to bias in the results. In all of the four studies reviewed, the provider of the program was not identified. This may have led to further biased results as the tester may have been the program provider and may not have been blinded for the study. It could be suggested that the lack of any specialist training for performing these treatments by the program provider, which may be required for this population, could ultimately have a negative effect on the intervention’s outcomes and ultimately on future patient’s outcomes. In addition, these studies did not report on having a blind assessor. Of the four studies reviewed, none looked at a long term follow up of their intervention on this patient group. Long term follow up studies are required to support the effectiveness of these programs and determine the optimal length of treatment for maintaining improvements. The study by Garcia, et al., (2009), had a control group but the group’s participants did not have haemophilia nor did they have any articular anomalies. A comparison of an intervention group of haemophilia patients to a group of non-haemophilia participants in an aquatic environment does not truly reflect a typical comparison between a control and experimental group in a population undergoing treatments. _____________________________________________________________________________________ 12 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ To date, Mazloum, et al., (2014) is the only article that has measured pain in patients with haemophilia. Research confirms that pain has a detrimental impact on patients with haemophilia and would be an important outcome of an intervention. Mazloum, et al., (2014) found that aquatic based programs lead to bigger improvements in pain reduction. This finding is of important clinical significance, especially for patients with haemophilia who are experiencing high pain levels, where the pool may be a more suitable environment over land. As a result, both clinical and future research should include pain in its outcomes to determine the improvements of aquatic based programs on pain in patients with haemophilia. The American College Sports Medicine guidelines for improvements in strength in the normal population state that training should include 1-3 sets of 8-12 repetitions at a minimum of 2 nonconsecutive days per week (American College of Sports Medicine, 2013). The study by Kargarfard, et al., (2013) which investigated knee flexion and extension strength did not specify its length of program, frequency nor details of program intervention. Despite their result with an increase in knee flexion and extension strength, this program is not reproducible due to a lack of a specified dosage. Lastly, the second aim of this literature review was to determine the dosage of an aquatic physiotherapy intervention for patients with haemophilia. However, due to the lack of specificity and poor quality of evidence this was not achieved. Within the four studies, the program varied between 30 and 60 minutes of intervention, between 2 and 3 times per week and lasting over a range of 4-9 weeks. As can be seen, due to the lack of specificity of the programs’ parameters, a dosage for an aquatic physiotherapy intervention was not determined. These studies did not reveal an optimal duration of a treatment session and overall program length, nor did it state the frequency of treatment and length of session with sessions varying between 30 and 60 minutes of therapy. For example, the study by Garcia did not state how long their intervention was for. Whereas there was conflicting durations for Mazloum, et al., (2014) and Kargarfard, et al., (2013) with 4 weeks and 8 weeks respectively for improvements in ROM. Future research needs to be undertaken to determine if there is an effectiveness of aquatic physiotherapy treatment of 4 weeks compared 8 weeks of intervention. Furthermore, to improve motor performance, 9 weeks has been supported by Vallejo, et al., (2010). However, could these improvements be seen in _____________________________________________________________________________________ 13 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ shorter durations? As previously stated, further research is required to determine the optimal dosage of intervention for patients with haemophilia. Conclusion A review of the literature of aquatic intervention in the haemophilia population reveals that improvements in ROM and strength of affected joints can occur, with improvements in pain as well as improvements with aerobic capacity. However caution should be taken when interpreting these results as the research was conducted in poorly designed studies with small sample sizes with potential bias. Despite these encouraging results, an effective dosage of an aquatic physiotherapy intervention, was not achieved due to the conflicting and minimal evidence supported in the literature. Higher quality studies with larger sample sizes and better methodological rigour whilst limiting bias, need to be undertaken to provide the evidence in determining best practice of aquatic physiotherapy in patients with haemophilia as well as determining the optimal dosage when providing this intervention. _____________________________________________________________________________________ 14 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ References American College of Sports Medicine, 2013. Resistance Training for Health and Fitness. [Online] Available at: https://www.acsm.org/docs/brochures/resistance-training.pdf [Accessed 26th October 2014]. Cuesta-Barriuso, R., Gomez-Conesa, A. & Lopez-Pina, J., 2013. Physiotherapy Treatment in Patients with Haemophilia and Chronic Ankle Arthropathy: A Systematic Review. Rehabilitation Research and Practice, Volume 2013, pp. 1-10. Garcia, M. et al., 2009. Variations of the Articular Mobility of Elbows, Knees and Ankles in Patients with Severe Haemophilia Submitted to Free Active Movimentation in a Pool with Warm Water. Haemophilia, Volume 15, pp. 386- 389. Haemophilia Foundation Australia, 2014. Haemophilia. [Online] Available at: https://www.haemophilia.org.au/bleedingdisorders/haemophilia [Accessed 26th October 2014]. Heijnen, L., 2008. The Role of Rehabilitation and Sports in Haemophilia Patients with Inhibitors. Haemophilia, Volume 14, pp. 45-51. Jansen, N., Roosendaal, G. & Lafeber, F., 2008. Understanding haemophilic arthropathy: an exploration of current open issues. British Journal of Haemotology, Volume 143, pp. 632-640. Kargarfard, M., Dehghadani, M. & Ghias, R., 2013. The Effect of Aquatic Exercise Therapy on Muscle Strength and Joint's Range of Motion in Hemophilic Patients. International Journal of Preventative Medicine, Volume 4, pp. 50-56. Mazloum, V., Rahmana, N. & Khayambashi, M., 2014. Effects of Therapeutic Exercise and Hydrotherapy on Pain Severity and Knee Range of Motion in Patients with Hemophilia: A Randomized Controlled Trial. International Journal of Preventative Medicine, Volume 5, pp. 83-88. Quinn, E., 2014. Fitness Test- 12 Minute Run Fitness Test for Endurance. [Online] Available at: http://sportsmedicine.about.com/od/fitnessevalandassessment/a/12MinRun.htm [Accessed 29th October 2014]. Rodrigues-Mechan, E., 1996. Effects of Haemophilia on Articulations of Children and Adults. Clinical Orthopaedics and Related Research, Volume 328, pp. 7-13. Souza, J. et al., 2011. Haemophilia and Exercise. International Journal of Sports Medicine, Volume 33, pp. 83-88. _____________________________________________________________________________________ 15 Aquatic Physiotherapy and Haemophilia ______________________________________________________________________________ Vallejo, L. et al., 2010. Influence of Aquatic Training on the Motor Performance of Patients with Haemophilic Arthropathy. Haemophilia, Volume 16, pp. 155-161. _____________________________________________________________________________________ 16