Public Perceptions and Attitudes of Bangladeshi Population towards Thalassemia Prevention: A Nationwide Study

Nur-EAlam Mawlana Bhashani Science and Technology University Md Shariful Islam (  sharifbge@uky.edu ) University of Kentucky https://orcid.org/0000-0002-7631-882X Umme Suriea MBSTU: Mawlana Bhashani Science and Technology University Ramisa Binti Mohiuddin MBSTU: Mawlana Bhashani Science and Technology University Md. Muzahidul Islam MBSTU: Mawlana Bhashani Science and Technology University Sumaiya Akter Mawlana Bhashani Science and Technology University Salma Aktar MBSTU: Mawlana Bhashani Science and Technology University Nahid Mahamud Mawlana Bhashani Science and Technology University Omaima Nasif King Saud University Sulaiman Ali Alharbi King Saud University Gaber El-Saber Batiha Damanhour University Md. Nazmul Islam Bappy Sylhet Agricultural University Dipankar Sardar Khulna University Mst. Mahmuda Khatun MBSTU: Mawlana Bhashani Science and Technology University Kamal Chowdhury Cla in University A. K. M. Mohiuddin MBSTU: Mawlana Bhashani Science and Technology University


Introduction
Thalassemia is the most commonly inherited single-gene disorder globally that results from absence or decrease of globin chain production (1). Depending on the nature of the mutation, there are two types of thalassemia: alpha-(α-) thalassemia and beta-(β-) thalassemia (2,3). High prevalence and the lack of cure make thalassemia as a global health concern (4). Globally an estimated 100 million people are carriers of beta-thalassemia, approximately 100,000 children are born and 50,000-100,000 children with thalassemia major die each year in low-and middle-income countries (5,6).
Thalassemia is highly prevalent in Southeast Asia, the Indian subcontinent, Mediterranean, middle Asia, Central Asia and West Africa (7). As a consequence of migration towards non-endemic regions, thalassemia is spreading in Europe and North America (8).
South Asia (India, Pakistan, Bangladesh and Sri Lanka) is a region with a high prevalence of hemoglobinopathy, representing 23% (approximately 1.56 billion) of the world's population (9). Bangladesh is situated in the South Asian region, with a population of over 160 million people. About 10-19 million people of this country (6-12% of the population) carry a thalassemia gene (10). According to World Health Organization (WHO) estimates, approximately 3% of the population (3.6 million) carries β-thalassemia and 4% (4.8 million) carries hemoglobin E (HbE) in Bangladesh (5,11,12). It is assumed that over 7000 children are born with thalassemia each year in Bangladesh (13). Furthermore, beta thalassemia or HbE has been found in 28% of assessed rural women in a recent study (14).
Allogeneic hematopoietic stem cell transplantation (alloHSCT) is currently the only curative therapy for thalassemia despite having limited access in the absence of suitable donors. However, most developing countries lack the necessary medical resources and skills to perform alloHSCT (15). Regular blood transfusions and iron chain treatment with desferrioxamine is the standard management of thalassemia that begin in patients early in life and continue throughout their childhood, adolescence, and adult years (16). In addition, managing thalassemia patients and living with this condition nancially and emotionally for a long time constitutes a heavy burden for patients and their families (17). Therefore, prevention is the best way to reduce the prevalence of this disorder. Different strategies were applied in different countries to reduce thalassemia. Increased awareness of general population, pre-marital screening and genetic counselling (PSGC) and prenatal diagnosis (PND), has led to almost total elimination of thalassemia in Cyprus and, to a considerable extent, in Greece, Italy and Sardinia (1,18).
Although Bangladesh is in the world thalassemia belt, there is a lack of information about the epidemiology, clinical course, mortality, complications and treatment outcome of thalassemia. The general peoples of Bangladesh possess poor knowledge of the disease. This lack of awareness is in uenced by region and population, including gender, marital status, education, employment, and socio-economic status (3). Furthermore, there is no national health insurance system or organized national program in Bangladesh to raise awareness, conduct career screenings or manage thalassemia patients (10).Therefore, this study aimed to assess public knowledge, perceptions and attitudes toward thalassemia and thalassemia screening practice.

Participants and study area
A cross-sectional study was conducted from January 2020 to October 2020 among randomly selected 1,623 peoples (16 ->50 years old) in eight divisional regions (Dhaka, Chittagong, Barisal, Khulna, Rajshahi, Rangpur, Mymensingh and Sylhet) of Bangladesh. We assumed a 50% prevalence of good knowledge and attitudes, with an error margin of 3% at a 95% level of con dence, the minimum number of respondents required to ful l the objectives of this study was 1067 (19). Participants were selected from different places, such as public institutions, houses and local markets in order to capture them from various backgrounds in the community. Only the people who consented were included in this study.
Furthermore, only those who were < 16 years of age and failed to respond to the questions were excluded.
Questionnaire Content Data was collected via a self-administrative close-ended questionnaire which was developed by this research team based on an extensive review of the literature (4,10,20,21). The study questionnaire was rst developed in English and translated into Bengali after which translation accuracy was veri ed by an independent bilingual translator.
The questionnaire was comprised of 27 questions, and divided into four sections which included: i) demographics (9 items), ii) knowledge towards this disease (12 items), iii) attitude (3 items), and iv) practice of thalassemia screening (3 items). The responses to knowledge questions (from Q11 to Q20) were categorized into three groups: (i) correct, which included the right answers, (ii) incorrect which included the wrong answers and (iii) do not know responses. A total knowledge score was calculated by summing the responses for participants who reported having heard of thalassemia.
The total score ranged between 0 and 10. According to our criteria, participants' knowledge was considered adequate when the score was equal or more than 6.

Data Analysis
Categorical variables were described using frequencies and percentages, and continuous variables were summarized using means and standard deviations. One-way ANOVA was performed to assess the association between the demographic variables (gender, literacy, marital status, living place, employment status and socio-economic status) and knowledge of thalassemia among those who heard of the disease. Post-Hoc tests were conducted using Tukey's HSD.
Pearson's chi-square (χ2) test was also used for testing the association between categorical variables. A p-value less than 0.05 was considered to be signi cant. Data were analyzed using IBM SPSS version 20 software.

Ethical Clearance
This study was conducted in accordance with the Declaration of Helsinki. The study was approved by the Dept. of from each participant before the start of the study. Those who were not willing to participate, were not given the questionnaires. Con dentiality of the respondents was maintained.

Results
Participant's characteristics A total of 1623 respondents participated in the study, out of which 856 (52.7%) were male and remaining 767 (47.3%) were females. Participants' ages ranged from 16 ->50 years with a mean of 34.082 (± 12.44) years. Approximately half of the participants (47.9%) were unmarried. Sixty three percent of the participants were from Semi Town/ rural community (village) while rest participating in study were from urban areas. Of the total respondents, 43.2% were students and majority of them (82.9%) belonged to middle class families (Table 1). Approximately 16% of the participants reported a family history of genetic diseases (Fig. 1).
Of the total respondents, only 44.7% (726/1623) had heard of thalassemia (Table 1). These 726 participants were included in our study. The urban residing participants (60.9%) who had heard of thalassemia were nearly twice as high as the participants (35.3%) who lived in semi-urban or rural settings. Textbooks (47.4%) was cited as the most frequently mentioned source of information about thalassemia followed by family/friends (23.1%) and internet/ social media (20.8%), respectively (Supplementary le 1). Of the participants who have heard of thalassemia, 0.8% (n = 06) had thalassemia major and 6.9% (n = 50) had family members or relatives with thalassemia major (Fig. 1).

Attitudes And Practice Towards Thalassemia
In response to the questionnaire, majority of the respondents who have heard of thalassemia showed positive attitudes towards thalassemia. About 68.2% respondents would prefer premarital screening of themselves or their family members to prevent thalassemia. The majority of the participants (85.5%) had given positive response about donating blood to thalassemia patients. Likewise, 96.3% were agreed to spread the information about thalassemia in their community. The participants from Dhaka region showed more positive attitude than other regions of Bangladesh.
Signi cant association was observed between agreement in opinion that prefer premarital screening and spread awareness about thalassemia with region (p < 0.001) ( Table 4).
Respondents were asked whether they had undergone thalassemia screening process, only 7.7% reported that they had been screened for thalassemia. Among these, the majority of the participants (6.6%) were unmarried. No signi cant association (p < 0.05) was observed between the socio-demographic variables and the practice status (Table 5).

Discussion
This study was carried out to determine the public knowledge, attitude and screening practice of thalassemia in Bangladesh. Very few studies have investigated knowledge about thalassemia in different parts of Bangladesh, and in our knowledge, this is the rst study conducted among the general peoples across the country. Our study helped to address the knowledge gaps related to thalassemia.
Our study results demonstrate that only 44.7% of the respondents had heard of thalassemia. As Bangladesh lies in the thalassemia belt, the level of awareness is unexpectedly lower than the countries including Malaysia (76%), Greece (93%), Bahrain (65%) and Italy (85%) (17,20,22,23). The most worrisome nding in this study is that of the participants who declared to know about thalassemia, only 37.7% have adequate knowledge and 32.1% believed that thalassemia is a transfusion transmitted disease. This result re ects a general lack of knowledge among the participants. More importantly, these participants may convey incorrect information to others who do not know about the disease.
The participants' knowledge of thalassemia as an inherited disorder was relatively better in this study. In contrast, a study conducted in Pakistan documented that only 40% were aware of the nature of disease (24). However, an alarming nding of our study was that half of the respondents were unaware about the role of consanguineous marriages in the incidence of thalassemia which was incongruous with a previous study in Pakistan (21). About 70.4% had the correct knowledge that both parents have to be carriers of beta thalassemia to have an affected child. This percentage is more than the study ndings conducted in Bangladesh and Pakistan (10,25). Half of the respondents had misconceptions that if one parent is a carrier, a child is born with thalassemia disease. These knowledge de cits may lead to stigmatization and have profound emotional effects on thalassemia carriers. The ndings from the present study showed, respondents' marital, literacy and employment status had a signi cant relationship with the level of knowledge on thalassemia. However, the study among participants in Bangladesh reported that thalassemia knowledge was found to be signi cantly related to having higher education levels and being a student. These results were consistent with the study from Kolkata and Bahrain (2,22).
Textbooks were selected as the most common sources of information for those who had heard about thalassemia.
Similar nding was reported in a previous study in Bangladesh (10). From this study health professionals (family doctors, obstetricians, and genetic counsellors) contributed very little to spread awareness about thalassemia. About 23% of respondents reported family and friends as a source of information which was similar to the result of the study performed in Italy (23). Some studies have already revealed that physicians can play a greater role in informing the public about thalassemia. In Sardinia, 70% of the target population was informed via physicians (26).
Despite the lack of knowledge, the participants who have heard of thalassemia showed positive attitudes towards the disease. Premarital screening and genetic counseling facilities will contribute to reduce the number of babies born with thalassemia (27). However, about 68% of respondents in this study agreed to do premarital screening with a blood test before marriage which is less than Oman, where 92% participants responded that they will do the test in future (28). A very positive nding of this study was that 85.5% were willing to donate blood to transfusion-dependent thalassemia patients. This study ndings raise the hopes that future awareness programs could easily increase the number of blood donors and family members with thalassemia will be able to nd blood donors. Merely 7.7% of the study population got themselves screened for thalassemia. This nding was similar with the Indian study where 2% of the participants performed premarital testing (24). Ignorance, fear of being stigmatized for positive results, and endangering future prospects of getting married are considered barriers to not perform any screening test. In Bangladesh (and the South Asian region overall), marriage is synonymous with nancial, emotional and physical security for many women (29).
Thalassemia is becoming a rising concern for public health in Bangladesh. Based on our study, it could be recommended that public education about thalassemia should be emphasized for successful thalassemia prevention. It has already been proven in several countries worldwide that implementation of mandatory national premarital screening programs could drastically reduce the incidence of infants born with thalassemia major (17). In 2018, a writ petition was led in the Bangladesh High Court to make pre-marital screening mandatory. Furthermore, the Minister for Health and Family Welfare also announced that "Bangladesh would be thalassemia free by 2028" (10). However, the Ministry of Health should provide adequate health workers and train them to give appropriate advice in an effort to bring about behavioral change among the public to discourage consanguineous marriages. Furthermore, like the polio campaign, Thalassemia prevention programs should be planned and interventions should be made all over Bangladesh to get rid of thalassemia.

Conclusion
This study has identi ed major areas which need to be highlighted and emphasized in rural communities and public education for thalassemia screening and awareness campaigns in Bangladesh. The Ministry of Health, Bangladesh has announced an intention to start a national screening programme for thalassemia. Our study has speci cally pointed out knowledge de cits regarding the genetics and pattern of inheritance of thalassemia. Insights of the report depict that more concise and specially designed programs for disseminating awareness regarding thalassemia should spread across the country.

Limitations
We recognized a few limitations in our study. Firstly, all data of this research were collected via face-to-face interviews; therefore, reporting bias due to socially desirable attitudes and behaviors might exist. Secondly, we faced some di culties to translate the questionnaire from English to Bangla as some English words do not translate exactly into Bangla. Furthermore, a cross-sectional survey of this nature may capture only a snapshot of information about the respondents but cannot be generalized to other populations; the ndings may change over time.

Declarations
Ethics approval and consent to participate

Consent for publication
Not applicable Availability of data sets The datasets used and/or analysed during the current study are available from the corresponding author on reasonable request.

Supplementary Files
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