Abstract
Documented evidence of the usage of complementary and alternative medicine among patients with rare disease such as hemophilia is limited. Therefore, we explored the types of complementary and alternative medicines used, the associated sociodemographic and clinical factors, and the cost and reasons for usage among severe hemophilia A patients in India. Our study demonstrates an increased usage of complementary and alternative medicine (42.3%) among hemophiliacs. Significant factors associated with usage of these therapies were economic status, comorbidity status, and education of head of households. Among users of complementary and alternative medicine, cost of these therapies was found to be higher (17.22%) compared with hemostatic drugs (5.63%) from the average cost of treatment (Rs 29 029). The findings of this study warrant multicenter research to explore different dimensions of complementary and alternative medicine and also incorporate it in comprehensive hemophilia care programs to address issues of treatment gap and quality of life of hemophiliacs.
Introduction
Complementary and alternative medicine is practiced across many continents such as Asia, Africa, and Latin America and is part of classical documented heritage and ancient civilization. It has become an important aspect in treatment protocols for patients suffering from chronic diseases and is often being used along with or independently from conventional treatment. The medical pluralism of Ayurveda, Yoga, Siddha, Unani, and Homeopathy (AYUSH) has been supported by the Government of India in its policy statements over the years. Except for the Bhore Committee (1943), various other committees such as the Chopra Committee (1948), C. G. Pant Committee (1950), Dave Committee (1954), Mudaliar Committee (1961), Udupa Committee (1968), and the National Health Policy (1983) have supported the idea of integrating modern medicine into the Indian system of medicine with requisite changes in medical curricula and establishment of state-of-the-art facilities for promoting research and services in the field. 1 During the seventh, eighth, and ninth plan periods, 3% of the national health budget was allocated to AYUSH. 1 Accordingly, a policy and institutional framework is provided for the Indian system of medicine in terms of statutory bodies, regulatory bodies, apex research bodies, apex educational institutes, and pharmaceutical committees in Indian contexts vis-à-vis the allopathic system of medicine. For example, since 1969 the Indian Council of Medical Research was divested of its central role in directing clinical research programs on the Indian system of medicine alone, an autonomous body—the Central Council for Research in Indian Medicine and Homeopathy—has been entrusted with the responsibility of conducting research in AYUSH. In 1978, this composite council was dissolved and 4 independent councils were formed in each specialty: the Central Council for Research in Ayurveda and Siddha, the Central Council for Research in Unani Medicine, the Central Council for Research in Yoga and Naturopathy, and the Central Council for Research in Homeopathy. 2 In 1995, the Department of Indian System of Medicine and Homeopathy was established under the Ministry of Health and Family Welfare (renamed as AYUSH in 2003). The National Medicinal Plants Board was established under the department to coordinate activities related to cultivation, marketing, export, and policy making for the development of the medicinal plants sector. 2 These concerted efforts of the Government of India had finally culminated in the form of the National Policy for Indian System of Medicine and Homeopathy in 2002. It aims to promote good health, expand outreach, ensure an affordable Indian system of medicine and homeopathy services, and integrate it within the health care delivery system via providing fiscal incentives and budgetary allocations for expanding centrally sponsored schemes, establishment of pharmacopoeia laboratories, encouraging research, and building centers of excellence and model institutions for its growth and development. 3 Today, AYUSH has a huge network and trained manpower, including 787 000 registered practitioners; 3277 hospitals with a bed strength of 62 649; 24 289 dispensaries; 501 recognized graduate and postgraduate colleges; and 8644 drug manufacturing units under the AYUSH system. 4 Eight national institutes exist at the national level for teaching, research, and clinical activities. 2 To make AYUSH medicine affordable, reimbursement provisions for cost on AYUSH are incorporated under social insurance such as the Employment State Insurance Scheme and the Central Government Health Scheme. In 2005, under the National Rural Health Mission, policy measures were implemented to mainstream AYUSH in the public health system. Various projects in the field have been explored to study in depth the multifaceted aspects of complementary and alternative medicine. For example, recently the Council of Scientific and Industrial Research, of the Government of India led the ayurgenomic project (integration of ayurveda with genomics). That research has established links between ayurveda and modern science, which hold great potential for predictive and personalized medicine at affordable costs. 5
Empirical research studies performed on large-scale surveys by the National Council of Economic and Applied Research (1990); the Indian Council of Medical Research (2004 and 2005); and National Sample Survey Organization, National Health and Family Survey-II (1998-1999) are replete with examples of the usage of complementary and alternative medicine, the costs incurred, and the reasons associated exclusively in the Indian context, 6–9 demonstrating a significant percentage of utilization of complementary and alternative medicine in the Indian population. The specific pathologies related to the usage of complementary and alternative medicine in rheumatoid arthritis and diabetics mellitus are also available in the Indian context. 10–12 These studies testify to a widespread acceptance of complementary and alternative medicine in the Indian community and are tantamount to studies among chronic illness in international contexts. 13–15 Research demonstrating the potential effectiveness of any particular form of these systems of medicine usage such as homeopathy and other forms of alternative therapy usage such as acupuncture and hypnosis in the hemophilic population has been reported in previous studies. 16–20 For example, Kundu and colleagues, 16 in a blind, placebo-controlled, crossover trail of 28 consecutive hemophilic patients with severe (24) or moderately severe (4) hemophilia administered standard management with placebo homeopathy for 1 year and active homeopathic treatment in the subsequent year with the same conventional management. The study was able to demonstrate that the use of homeopathy medicine results in reduced frequency of bleeding, extent of bleeding, blood products consumed, and improved well-being in 24 hemophilic subjects. 16 Two seminal studies based on hemophilia pain management with focus on use of pharmacological and alternative treatment modalities were carried out in the United States. 21 , 22 A national pain study in 2009 replicated the pilot study of 2005, and both studies were prospective and descriptive in research design. Among several objectives, one of the objectives was to describe use of alternative treatment modalities to control acute and persistent pain among hemophilic patients. The pilot study, which analyzed 114 hemophiliacs within 2 hemophilia treatment centers in Michigan, documented the use of nontraditional therapies such as acupuncture, acupressure, herbal remedies, faith, and prayer less than 35% of the time. Use of nonpharmacological methods for pain management that fits into the lifestyle of persons living with hemophilia were more visible and pronounced as documented in a national pain study that identified 700 persons living with hemophilia across 12 regions of the United States. The study has identified the use of homeopathy (5%), naturopathy (2%), faith (22%), and prayer (29%) by hemophilic patients both for acute and persistent pain. Other modalities trialed were massage therapy (84%), heat application (33%), physical therapy (30%), transcutaneous electrical nerve stimulation (10%), chiropractic care (10%), herbal therapies (9%), biofeedback (5%), and acupuncture (4%). Similarly, alcohol (15%) and illicit drug use (8%) have been reported for the management of both acute and persistent pain. These studies underscore the role of hemophilia treatment centers in recognizing the efficacy of these modalities and accordingly develop multimodal guidelines incorporating both pharmacological and nonpharmacological treatment plans that have significant impact on the persons living with hemophilia and experiencing pain.
Hemophilia represents a rare inherited form of bleeding disorder having the second highest global burden of disease in India (total cases = 11 580) with an incidence of 1 in 10 000 in male births. 23 The hemophilia gene is passed on at birth from an affected father to daughter, who then becomes a carrier of the hemophilia gene, and from mother to son, who will have hemophilia. Even though it is genetic in nature, almost 30% of cases are fresh mutations. Hemophilia is characterized by deficiency in clotting factor, either factor VIII (in hemophilia A) or factor IX (in hemophilia B), resulting in the inability of the blood to clot normally. The severity of disorder is dependent on the measured activity of clotting factor—mild (5% to 30% of normal), moderate (1% to 5%), or severe (<1%). 24 Clinical manifestations include repeated bleeding tendencies into deep tissues, especially joints and muscles, eventually leading to chronic hemophilic arthropathy, morbidity, and contributing to progressive functional disability. Other internal bleeding such as intracranial hemorrhage and gastrointestinal hemorrhage are generally fatal. Expensive antihemophilic factor replacement is an important component in the treatment of hemophilia, which is infused at the time of bleeding episode to control it and prevent damage. Due to its low prevalence, limited health budget, and pressing public health priorities, expensive hemophilia treatment does not garner any support from public health functionaries. 25 , 26 The exorbitant cost of antihemophilic factor necessitates patients to resort to suboptimal treatment. Comprehensive hemophilia care is an exception rather than a rule in most parts of the country as treatment facilities are available only at tertiary centers in the city. The fallout of a lack of standardized treatment facilities and delays in treatment manifests in deteriorated health status in terms of high levels of comorbidities, excruciating pain, widespread disability, and diminished quality of life. In this milieu, patients are opting for complementary and alternative medicine or alternative therapies or traditional healers vis-à-vis allopathic treatment. Documentation of complementary and alternative medicine usage and its expenditure is imperative among these rare groups of patients in light of national policy of the Indian System of Medicine & Homeopathy 2002. Complementary and alternative medicine in the present research includes use of ayurvedic and homeopathy as these specialty physicians have been employed by some of the Hemophilia Chapters in the country and many hemophiliacs are known to resort to these kinds of therapies. 16 , 26 However, attempts have also been made to elicit information regarding any other practices, especially traditional healers, that patients have access to. The “traditional healers” are not part of complementary and alternative medicine but include people who practice quackery under the guise of metaphysical and supernatural things. The objectives of the present research were to explore the types of complementary and alternative medicine used associated with sociodemographic and clinical factors, its expenditure, and reasons for usage among hemophiliacs.
Materials and Methods
This is a cross-sectional, single-center, descriptive, and exploratory survey. As hemophilia A represents 85% of the cases compared with hemophilia B, 24 and on an average the severity of hemophilia shows significant relationship across clinical, social, and psychological functioning compared with mild and moderate hemophilia, 27 severe hemophilic A patients were included in the study. Initially, the 2010 registry of the Hemophilia Society was accessed, which shows in their database 1600 persons with hemophilia with various degrees of severity, including patients with mild, moderate, and severe hemophilia A, hemophilia B, Von Willbrand factor disorder, and other inherited bleeding disorders such as factor XII or factor XIII deficiency. From this database, 283 severe hemophilia A patients were found to be residing in Mumbai or its suburbs who were telephonically contacted. Those who reported at least 1 catastrophic bleeding episode during a 1-year retrospective period from January 2010 to December 2010 and subsequent health care resource use were included in the study. Thus, 160 severe hemophilia A patients (56.53%) could be identified and were interviewed by home visits and recruited for this study. Almost 67 (23.67%) patients’ address and contacts were found to be incorrect, 12 (4.24%) patients were dead, 29 (10.24%) patients did not report any catastrophic bleeding episode in the retrospective period, and 15 (5.30%) patients refused to participate in the study. After informed consent of adult hemophilic patient and/or parents of hemophilic children, data were collected on hemophiliacs’ sociodemographic, clinical, and health seeking behavior and type of health care providers accessed for that period using a semistructured interview schedule. Detailed questions were asked about expenses incurred as direct costs, which includes medicine, antihemophilic factor, user fee, consumables, complementary and alternative medicine, travel, food and accommodation during hospitalization, as well as indirect costs, including wages lost by caregivers and hemophilic patients themselves during illness episodes. Due attention was paid to estimating accurate expenditure for treatment while cross-checking with medical records, bills, and records of purchase of antihemophilic factor from the Hemophilia Society. Open-ended questions on why traditional healers were accessed and the reasons for accessing respective types of health care providers and complementary and alternative medicine were asked. The first author interviewed and collected the data, which last for 6 months from January 2011 to May 2011. Data were analyzed with Statistical Packages for Social Sciences, version 20. Data are reported as means and P values <.05 were considered statistically significant.
Results
Sociodemographic and Economic Status Parameters (Table 1)
Of 160 hemophilic patients, 43.1% (n = 69) were users and 56.9% (n = 91) were nonusers of complementary and alternative medicine. The mean age of hemophilic patients among nonusers was 19.74 years, and among users, the mean age was 22.81years. Age, religion, marital status, and education of hemophilic patient were not significantly associated between users and nonusers of complementary and alternative medicine. Hemophilic patients who were university educated used these systems of medicine more (65.9%). However, utilization of complementary and alternative medicine was found to be higher among heads of households who had school level of education than those who had university level of education, and this was statistically significant (P = .009). Furthermore, occupations of head of households were not significantly associated between users and nonusers of complementary and alternative medicine. In comparison with nonusers of complementary and alternative medicine, utilization was found to be higher among higher and middle monthly per capita expenditure group, 39.1% and 37.7%, respectively, than the lower monthly per capita expenditure (23.2%) group, and this was statistically significant (P = .019).
Sociodemographic Parameters of Users and Nonusers of Complementary and Alternative Medicine.
Abbreviations: PWH, person with hemophilia; MPCE, monthly per capita expenditure; ns, not significant; s, significant.
*P < .05. **P < .01.
Characteristics of Complementary and Alternative Medicine Users
Among users of complementary and alternative medicine, 43.5% (n = 30) availed homeopathy, 56.5% (n = 39) availed ayurvedic treatments, and 5.8% used traditional healers vis-à-vis complementary and alternative medicine. Furthermore, 30.4% (n = 21) used complementary and alternative medicine for less than a year, whereas 69.6% (n = 48) reported usage for more than a year. Among users of complementary and alternative medicine, more than 75% reported that the main reason for its usage was to control bleeding episodes, to reduce pain, and to reduce the number of bleeding incidents.
Clinical Parameters (Table 2)
Among users of complementary and alternative medicine, a lower percentage of hemophilic patients (36.2%) reported having a congenital history of hemophilia, compared with nonusers (41.8%), and the difference between the 2 groups was not statistically significant (P = .47). Disability status (52.2%), joint bleeding status (78.3%), and number of bleeding episodes (both less than 5 and more than 5) are reported to be higher among users of complementary and alternative medicine (78.3% and 17.4%, respectively) than nonusers. However, the difference between them was not statistically significant. Lower functional health score derived using the Hemophilia Utilization Group Study was reported to be higher among users (58%) compared with nonusers of complementary and alternative medicine, and the difference was not statistically significant. Comorbidity status was reported to be higher among users (30.4%) than nonusers (16.5%), and the difference between the 2 groups was statistically significant (P = .029).
Clinical Parameters and Usage of Complementary and Alternative Medicine.
Abbreviation: FHS, Functional Health Score.
a S using χ2 test of significance at <.05 level.
Cost for Complementary and Alternative Medicine
From the average cost of treatment of Rs 29 029 among users of complementary and alternative medicine, the mean cost for these treatment modalities was reported to be Rs 4999 (17.22%); (Table 3). Higher mean cost was reported for the ayurvedic system of medicine compared with the homeopathy system of medicine. For treatment obtained from traditional healers, the mean cost was reported to be Rs 603. Furthermore, the mean total direct expense of treatment was higher for complementary and alternative medicine (17.22%) compared with hemostatic drugs (5.63%). Overall, the total direct cost for treatment of hemophilic persons was higher for users compared with nonusers, and the difference between them was statistically significant (P = .025). Additionally, among components of total direct cost for treatment, both direct medical cost and direct nonmedical cost were higher for users of complementary and alternative medicine compared with nonusers, and the difference between the 2 groups was statistically significant only for direct medical cost (P = .024).
Cost of Treatment Among Users and Nonusers of Complementary and Alternative Medicine.
Discussion
Hemophilia treatment protocols are dictated by scarcity and exorbitant cost of antihemophilic factor due to which treatment of hemophiliacs is suboptimal in Indian contexts. 25 , 26 Many patients are opting for either rudimentary forms of treatment, that is, infusion of whole blood or plasma or cryoprecipitate, which is considered highly precarious as there is no effective system of viral inactivation, 28 , 29 or relying on episodic or on-demand treatment albeit with too little and too late infusion of antihemophilic factor. Thus, unmet need of episodic treatment manifests in increased pain, hospitalization, crippling disability, comorbidity, and affects on overall quality of life. The treatment gap is addressed by patients resorting to various forms of complementary and alternative medicine complementary with conventional treatment, and the present study provides evidence for this. Higher usage of complementary and alternative medicine among hemophilic patients (42.3%) reiterates findings from other studies where usages of complementary and alternative medicine is significant in patients suffering from chronic diseases such as diabetics, HIV, rheumatoid arthritis, cancer, and so on. 10 - 15 Treatment with complementary and alternative medicine is individual oriented and hence very specific; therefore, higher usage of these systems of medicine denotes higher perceived benefits from these treatment modalities. Study has reported that 5.8% (n = 4) of users have visited traditional healers vis-à-vis usage of complementary and alternative medicine systems. Accessing traditional healers is associated with their intrinsic faith in these healers along with ignorance, stigma, and superstitions related to the diseases. Due to ignorance about the nature of the disorder, hemophilia is perceived as “wrath of the goddess or witch’s evil eye,” and therefore hemophilic patients and their caregivers approach traditional healers immediately after first signs of bleeding episodes. Furthermore, higher usages of these systems of medicine among higher and middle monthly per capita expenditure groups could be associated with health literacy as access to medical information is always reflected in higher/middle economic class and ability to pay for complementary and alternative medicine among these economic strata of hemophilic patients. Access to information with resultant increase in health literacy leads to adoption of other preventive and curative aspects of health care. Those with congenital history of hemophilia have found lower number of users (36.2%) compared with those without congenital history (41.8%), and the reason for this could be that congenital history has exposed the family to this illness through generations, which culminated in experience of absence of any discernible changes after opting for complementary and alternative medicine. Higher level of prevalence of clinical parameters such as disability and comorbidity status among users could be the reason to use complementary and alternative medicine as a comorbid condition makes a patient experiment with different treatment modalities to either control it or to have a better quality of life. Higher functional health status signifies lower usage of complementary and alternative medicine (42%), and lower functional health status indicates higher usage of complementary and alternative medicine (58%), which indicates increased burden of diseases of hemophilic patients, and to cope with it, more usage of these systems of medicine must have been necessitated. Similar findings have been noted in a study of patient motivations for using complementary and alternative medicine. 30 From average expenditure of treatment among users, highest expenditure for complementary and alternative medicine (17.22%) compared to hemostatic drugs (5.63%) marks the advent of increased professionalism and commercialization of these systems of medicine. Complementary and alternative medicine systems in India are seen to have a potential market along with their established industrial and technological base. In light of the increasing cost of complementary and alternative medicine, the recommendations of the High Level Expert Group on Universal Health Coverage by 2020 of the 12th Planning Commission of India are highly applicable. Besides recommending increased budgetary allocation for AYUSH (provision of 10% of total budget of health sector to AYUSH), it has emphasized incorporating drugs of complementary and alternative medicine in the national list of essential medicine, which will ensure AYUSH drugs are available at primary health centers, thereby enhancing the contribution of AYUSH doctors who are presently handicapped by the lack of AYUSH drug supplies. 4 , 31 This is a ray of hope for not only reducing cost of complementary and alternative medicine but also mitigating overall treatment cost for persons living with hemophilia.
Conclusion
Conventional treatment for hemophiliacs, that is, infusion of antihemophilic factor, is a standard treatment as recommended by the World Health Organization (WHO) and the World Federation of Hemophilia. However, in the backdrop of infectious and noncommunicable diseases, a rare disease such as hemophilia gets poor visibility in the Indian health system and provision of treatment products for hemophiliacs are considered as cost-intensive. Until the Government of India takes the onus of providing free treatment products to hemophiliacs in public health facilities, usage of complementary and alternative medicine will remain a reality for thousands of hemophiliacs. In many parts of the country AYUSH are the only accessible treatment modalities that are within the physical and financial reach of the people, thereby providing leverage to universal access to health care. The present study highlights 2 forms of complementary and alternative medicine usage by hemophilic patients influenced by multiple factors including comorbid conditions, expensive antihemophilic factor, and nonavailability of standard treatment for hemophilia in the Indian context, as India is reported to be have the lowest consumption per capita of clotting factor VIII, that is, 0.03 compared to its global burden worldwide. 23 , 32 The findings of this study warrant more collaborative and multicenter research to explore diverse issues of complementary and alternative medicine in the context of hemophilia such as its efficacy, veracity, its broader social implications, and disclosure of these systems of medicine to treating physician and hemophilia treatment centers in order to augment AYUSH facilities for hemophiliacs and to incorporate it in comprehensive hemophilia care programs to address issues of treatment gap and quality of life of hemophilic patients.
Footnotes
Acknowledgments
The authors thank the patients, respondents, and their families for their cooperation during the study period. The authors also express gratitude to the Hemophilia Society, Mumbai Chapter, for its full cooperation during the course of this study. This study is a part of PhD work done under the guidance of Dr Kanchan Mukherjee and performed in Mumbai and at Tata Institute of Social Sciences.
Author Contributions
Mrs Uma Jadhav conceptualized the study, collected and analyzed the data, and wrote the first draft of article. Dr Kanchan Mukherjee contributed to research design, analysis, and review of the final draft of article. Prof Harshad Thakur contributed to research design and analysis of the data.
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
Ethical Approval
The study was approved by the Committee for Academic Research Ethics of King Edward Memorial Hospital and Seth G. S. Medical College, Mumbai, India (Project No. EC/76/2010; CARE/OUT/661/10, dated October 26, 2010).