“Honestly, this problem has affected me a lot”: A qualitative exploration of the socioeconomic impacts of chronic respiratory disease among people and their communities in Sudan and Tanzania

7 Background : Over 500 million people live with chronic respiratory diseases globally and 8 approximately 4 million of these, mostly from the low- and middle-income countries including sub- 9 Saharan Africa, die prematurely every year. Despite high CRD morbidity and mortality, little is known 10 about the socioeconomic impact of CRDs in sub-Saharan Africa. We aimed to gain an in-depth 11 understanding of the socioeconomic impact of CRDs among people with CRD to inform management 12 of CRDs in Sudan and Tanzania. 13 Method : We conducted in-depth interviews with people with known or suspected CRD and focus 14 group discussions with members of the community in Gezira state, Sudan and Dodoma region, 15 Tanzania, to share their understanding and experience with CRD. The data was analysed using 16 thematic framework analysis. 17 Results : People with CRD in both contexts reported a significantly diminished capacity to do hard 18 physical work, resulting in both direct and indirect economic impacts for them and their families. 19 Direct costs were incurred while seeking healthcare, including expenditures on transportation to the 20 health facility and procurement of diagnostic tests and treatments, whilst loss of working hours and 21 jobs resulted in substantial indirect costs. Enacted and internalised stigma leading to withdrawal and 22 social exclusion was described by participants and resulted in part from association of chronic cough 23 with tuberculosis and HIV/AIDS. In Sudan, asthma was described as having a negative impact on 24 marital prospects for young women and non-disclosure related to stigma was a particular issue for 25 young people. Impaired community participation and restrictions on social activity led to 26 psychological stress for both people with CRD and their families. Conclusion : Chronic respiratory diseases have substantial social and economic impacts among 28 people with CRD and their families in Sudan and Tanzania. Stigma is particularly strong and appears


Introduction 34
Over five hundred million people globally live with chronic respiratory diseases (CRDs) such as 35 asthma, chronic obstructive pulmonary disease (COPD) and bronchiectasis(1). Approximately four 36 million of these die prematurely annually, most of whom are from low-and middle-income countries 37 (LMICs)(2), where the estimated socioeconomic burden is also high . In sub-Saharan Africa (SSA), the 38 increasing burden is driven by indoor air pollution due to domestic use of biomass fuel, which is used 39 for cooking by approximately 90% of rural households(3); tobacco smoke; and post tuberculosis lung 40 disease (4). Despite increasing recognition, CRDs are rarely prioritised by communities, health 41 systems, or governments in SSA and robust data with which to inform CRD interventions are 42 lacking(5). Reliable data on the prevalence of CRDs in Sub Saharan Africa (SSA) are scarce. A 43 systematic review(6) estimated the prevalence of COPD in Sub Saharan Africa to be 13.4%, which is 44 similar to Western European and North American settings(7). Asthma incidence has been estimated 45 to be approximately 10% in Khartoum,Sudan,(8) and recent data has shown a COPD prevalence of 46 16.5% in urban Sudanese adults who underwent spirometry(9). Similarly, a study in Tanzania showed 47 and well-functioning community-based referral and follow-up system for people with chronic cough 98 suspected to have TB even when their TB investigations are negative. The community referral 99 system in Tanzania maintains contact with community members through designated community 100 health workers who identify people with chronic cough who were found to be negative for TB but 101 continue to seek care in the health facilities for on-going respiratory symptoms. 102

Participant selection and recruitment 103
For IDIs, purposive sampling was used to select participants with confirmed or suspected CRD(19). In 104 Sudan, patients were purposively selected from the asthma standard case management programme 105 at EPILAB sites to include men and women with varied asthma severity and from catchment 106 communities closer and farther away from the health facility. Diagnosis of asthma was made by 107 clinicians using diagnostic algorithms and peak flow meter measurements at the EPILAB sites. In 108 Tanzania, the research team selected patients in collaboration with health workers in the catchment 109 area health facility with the aim of ensuring balanced representation by gender, socioeconomic 110 status and severity of symptoms. In Tanzania, clients with presumed CRD who had been investigated 111 for TB, were found to be negative but remained ill and continued to visit the health facility for 112 treatment without a definitive CRD diagnosis, were purposively selected from catchment areas of 113 study health facilities. 114 For FGDs, community representatives were selected to reflect the range of literacy levels and 115 socioeconomic backgrounds within the community in both countries. In Sudan, an additional group 116 of adolescent girls was constituted to create a socially safe and trusting environment for open 117 discussion, based on the research team observations that young women and adolescent girls were 118 particularly likely to experience negative social impact of asthma in the community. 119

Data collection 120
We used common data collection methods across the two countries. Topic guides were used to 121 guide semi-structured interviews and explored patients' and community understanding of CRD, 122 impact of CRD on productivity, work life, finances, coping mechanisms, experiences with CRD and 123 priorities for care. Additionally, in Sudan, patients' perceptions and experiences with the asthma 124 management services were also explored. 125 Training of research assistants already experienced in qualitative research was conducted in each 126 study site and covered overall aims of the study, data quality, ethical issues in health facility 127 research, informed consent, privacy and confidentiality. Training included hands-on practical sessions on qualitative interviewing, adapting topic guides and a data collection pilot in the 129 community. The pilot data was analysed and used to inform data collection in the main study. 130 Interviews were audio recorded and conducted in the local language (Swahili in Tanzania and Arabic 131 in Sudan) in a quiet, private and secure room within the health facility premises. FGDs were 132 conducted in mutually agreed venues in the communities. A note taker took notes during the 133 interviews and a debrief session was observed after interviews to reconcile any inconsistencies. All 134 interviews were transcribed by the field team, quality checked and translated into English by 135 professional translators prior to data analysis. 136

Data analysis 137
We used the Framework Method(20) for the management and analysis of data as it facilitates 138 transparent analysis and allows teams of researchers to collaborate on analysis(21). To improve 139 trustworthiness, members of the research teams independently read and re-read manuscripts, 140 inductively identifying emerging codes and developed an analytical coding framework for coding the 141 rest of the data after reviewing a few transcripts together and resolving discrepancies. This process 142 was iteratively repeated till no new codes emerged. Separate analytical frameworks were developed 143 for patient interviews and focus group discussions. Codes were organised into categories and 144 themes for analysis by charting into a spreadsheet matrix. Separate coding frameworks were 145 developed for each country. Similar themes that emerged from the interviews were compared and 146 discussed to identify final common themes. Socioeconomic impacts were defined as those effects of 147 CRS that negatively influenced social life and means of livelihood of the patients with CRS and their 148 families in the community, including the psychological consequences of these impacts(22) . 149 150 151

Demographics of study participants 153
We conducted 23 in-depth interviews, 7 in Tanzania (4 males, 3 females) and 5 in Sudan (2 males, 3 154 females). The composition of the FGDs is shown in table 1 below. There were 128 FGD participants, 155 50 in Sudan and 78 in Tanzania, with each FGD group consisting of 7-12 participants. Youth 156 participants were defined as those aged less than 18 years. The FGD location and age, gender, and 157 number of FGD participants is shown in table 1. 158 In general, participants described how CRDs impacted their activities of daily living, relationships and 162 interactions within the community, and the implications of this on their overall wellbeing. The 163 results are presented in the following themes: limitations on livelihoods; economic impacts of 164 healthcare seeking costs and CRD mortality on patients and their families; social exclusion, 165 discrimination and stigma; and impacts on social wellbeing, mental health, and sexuality. 166 167 People with CRD in both countries reported a significantly diminished capacity to do hard physical 168 work. The impact on livelihoods was the same among males and females as both relied largely on 169 physical work. This resulted in both direct and indirect economic impact for people with CRD and 170 their families due to lost working hours and inability to continue with physical work. For most people 171 this was due to being unable to be as 'productive' in farming or informal work, but for some this 172 involved reduced working hours in formal employment. Many people with CRD had to stop work to 173 attend clinics, be hospitalised, recuperate after treatment and/or attend follow-up appointments. 174

Limitations on livelihoods
While some people with CRD could change their work to involve less vigorous and less physically 175 demanding tasks, others stopped working entirely. Subsistence farming was common in our study sites with any surplus produce being sold for cash'. 181 Therefore, reduced ability or inability to work impacts on both the family's food supply and their 182 cash income. Loss of income also meant inability to pay school fees for dependent children and 183 inability to take care of dependents, including aged or infirm older relatives. A female cleaner 184 working in a local primary school reported that she stopped working when she developed CRD 185 because of the associated fatigue and breathlessness: 186 let alone the doctor's fee and medications and tests" (IDI-Male, Patient, Sudan). In both countries, 203 transportation costs were especially high when the health facility was located far away from the 204 patient's community and this challenge was so significant that some community members suggested 205 that they would be willing to pay specialist consultation fees if the specialist could come to a health 206 facility nearer to them, obviating the need for transportation. In some cases, people with CRD were 207 reportedly accompanied on their multiple health facility visits and admissions by bread-winning 208 family members who also incur transportation costs and loss of working hours during these periods. 209 "The family will also be affected; they (relatives) will accompany the patient when s/he falls ill. All 210 livelihood activities will be put on hold whether these activities are in the private or public work…The 211 whole family will all be at hospital. Financially and physically, they will be exhausted." (FGD-Female-212 Sudan) 213 Participants described multiple visits to seek care in different health facilities. This journey would 214 usually start with the local pharmacy and may end up in high level health facilities often located far 215 away from the person with CRD. This involved not only transportation costs but also costs of 216 medications and tests which are often beyond the ability of the person with CRD to afford . A 217 participant mentioned that treatment could cost an entire month's salary: The death of a relative with CRD was also reported to impact the economy of the extended family in 222 Tanzania. A participant reported that, in the event of death of a patient, relatives would be expected 223 to take over their responsibilities such as care of the family, parenting, payment of school fees, 224 provision of basic needs and management of assets, all of which exert major economic pressures on 225 the patient's relatives. 226 "I had a relative who had such a problem, we came to realize when it was too late, and she had a 227 family. When she felt sick, she used to go to the hospital to perform some tests and get medication. 228 In the end, they realised that her lungs were badly damaged. Unfortunately, she died, and we, as Similarly, in Sudan, asthma was believed to be infectious and people with CRD may be labelled as 269 'TB' because of chronic cough: "For them, asthma is something infectious that needs to be treated or a disease that might be 271 possible to cure. For instance, they tend to say that this person (is) living with tuberculosis or 272 something else." (IDI-Female-Sudan). 'Them' here refers to members of the community. 273 Some people with CRD who internalised their stigma mentioned that they did not disclose their 274 illness to family members and the community. In Sudan, some people with CRD reported that they 275 pretended to be well when in the company of other people, hiding their illness from family and 276 friends. They explained that this was to avoid attracting attention to themselves which may further 277 reinforce stigma, increasing their sense of themselves as a burden to their neighbours or family. A 278 young male with asthma highlighted how important it was to him for the family to be protected 279 from feeling his 'pain': 280

"Yes it does...it hurts me a lot, but I don't want my family to feel my pain." (IDI-Male, Sudan) 281
Of particular importance to female community participants in Sudan was the potential for loss of 282 marriage opportunities due to being discriminated against by potential suitors when a young woman 283 developed CRD or tuberculosis. This is because both CRDs (such as asthma) and tuberculosis are 284 stigmatising illnesses in the community . Marriage is a very important part of the identity of a 285 woman in Sudanese society and a symbol of her status both of her family and her own position 286 within it. A female participant who spoke of the impact of CRD on young unmarried women, 287 described the difficulties a young woman or girl with CRD would have in attracting a suitor: 288 "It will lead to single marital status. It will lower the market value." (FDG, Female adult, Sudan). 289 The impact of CRD-related stigma on marriage was reported to not only be limited to a prospective 290 wife but also to her family. This was reported as having the potential to jeopardise future marital 291 prospects for other women in the family: 292

"It is a social problem, solely a social problem. [Even] if the girl is so beautiful like the moon, they will 293
tell you not to marry from her family as they have so and so." (FGD, Female, Sudan) . 294 Similarly, a male participant emphasised that prospective husbands may be discouraged from 295 marrying young women with asthma for fear that asthma may be passed on to the children: 296 "In another area, people start to avoid and say that person is contagious, this is a social part. They 297 say X shouldn't marry Y because she has asthma and may affect children after that, pulling them to 298 those diseases" (FGD, Male, Sudan). 299 301 Participants reported that inability to play the usual expected roles in the society had impacts on 302 their sense of belonging and contributions to the society. This represents an important intersection 303 between the economic, social and psychological impacts of CRD on the patient and their families. In 304 both contexts, the social impacts of CRD such as impaired community participation and restrictions 305 on social activity, led to psychological stress to both people with CRD and their families. People with 306 CRD used terminologies such as 'moody', 'angry', and 'crying' to describe the impact of CRD on their 307 psyche. A patient in Sudan described himself as always in a bad mood and angry: 308 The sense of uncertainty around the illness was also reported to be shared by the entire family: 315 "if there is someone sick in the house, the whole of the family will be in stressful condition 316 particularly if the patient could not breathe … The whole family will be anxious; not only the patient 317 but all the family members will be in stressful condition." (FGD, Female, Sudan) 318

Impact on psychosocial wellbeing
The impact of CRD symptoms on sexual activity was also reported to be a contributor towards 319 negative impacts on wellbeing. Several male community members in Sudan emphasised that CRD 320 symptoms could impact the desire and capacity for sexual activity, and this was gendered, as it was 321 an important indicator of wellbeing and pride among men in the community. The strong association of chronic cough with TB in these settings is not surprising. Chronic cough was 342 widely presumed to be TB in a similar study among Ugandan communities(23). This highlights the 343 huge importance of TB as an ongoing public health challenge in the sub-Saharan African context. 344 Whilst Tanzania is one of the 30 high TB burden countries, Sudan has a moderate but also significant 345 burden of TB (WHO Global TB report 2020). The common knowledge of TB in the communities may 346 be explained by the historicity of endemic TB disease and by the substantial investments into TB 347 control compared to CRD and other diseases in these contexts. A downside of this association, as 348 seen in this study, is the stigmatization and ostracism of people with CRD in the community. This 349 may lead to failure to attend health facilities for fear of being confirmed as having TB disease, which 350 further delays diagnosis and can lead to increased morbidity and mortality (13,24). Misinformation 351 about chronic cough is another driver of stigma seen in this study. A study investigating knowledge 352 and perceptions of asthma among secondary school students in Tanzania revealed that most 353 information about asthma had been passed on to students by their parents and non-asthmatic 354 students presumed that asthma was infectious and therefore avoided interactions with their 355 asthmatic colleagues for fear of contracting the disease (10). Systematic community sensitization 356 activities with trained and well-informed health personnel could help improve community 357 perception about chronic cough and contribute to tackling stigma in CRDs. Clear messages about the 358 causes of asthma and the availability and effects of treatment on controlling symptoms would help 359 to destigmatise asthma and other CRDs. The effect to seeing people being managed well with 360 affordable medicines and transform their lives can be more powerful than being told about 361 treatment effectiveness. Words also matter and consistent use of terms like 'people who have 362 asthma' rather than asthma 'victims' or CRD 'sufferers' can start to shift mindsets. There are many 363 transferable lessons to be learned from community de-stigmatisation efforts with HIV in the antiretroviral therapy era(25) and, more recently, efforts being applied to COVID-19 related 365 stigma(26). 366 The economic impact of CRD in these communities highlights the vulnerability of people with CRD in 367 mostly rural settings, who are self-employed or working in informal sectors of the economy such as 368 subsistence farming, and without recourse to any social security. Chronic diseases, including CRDs, 369 are known to incur significant economic costs for both patients and the health system(27, 28). In 370 Malawi, the mean care seeking cost for chronic cough per patient was found to be 2.3 times the 371 average cost per capita on health for the country and consisted mainly of transport and drug 372 costs(29). Similarly, TB-related catastrophic costs have been well recognised and documented. A 373 systematic review of financial impact of TB in LMICs revealed that on average the total cost was 374 equivalent to 58% of reported annual individual and 39% of reported annual household income(30). 375 To tackle catastrophic costs in TB context, poverty reduction strategies are increasingly being woven 376 into TB control programmes including social protection initiatives such as cash transfers, food 377 baskets and social insurance (31). Health systems could draw from these experiences and 378 interventions in TB to develop an integrated approach to addressing the economic impacts of CRDs 379 in the sub-Saharan African setting. 380 Transportation costs were strongly highlighted by our study participants as a major source of 381 economic loss, impacting people with CRD and relatives who accompany them on hospital 382 appointments. Our informants themselves made the that the transportation costs were often the 383 equivalent of their monthly salary. Transportation has been identified as an important social 384 determinant of health (32) and a well-documented barrier to engaging in the care of chronic 385 diseases especially among poor, vulnerable populations (33). Studies done in high income settings 386 have shown that interventions such as provision of bus passes, taxi vouchers and reimbursements 387 from insurance covers could improve healthcare utilization (34) but these interventions may not be 388 feasible in our study setting . because of lack of transport infrastructure. Policy makers in our study 389 context could consider decentralisation of services and investment in specialist mobile clinics to rural 390 areas and geographically distant communities as measures to reduce the impact of transportation 391 costs and improve health outcomes of people with CRD. In our study setting, the extended family The finding in this study that young Sudanese women with chronic cough and their families were 412 discriminated against by potential suitors further highlights the gendered nature of stigma 413 associated with chronic cough. The perception that asthma, a major cause of chronic cough well 414 known in this society, was primarily hereditary, has also been described in a previous study of 415 asthma in urban Sudan (41), where 67% of 490 asthmatic patients believed that asthma could be 416 transmitted within the family. The study also highlighted denial of asthma diagnosis and non-use of 417 inhalers by young female asthmatic patients as a coping mechanism to avoid stigma. This highlights 418 the disproportionate social burden of CRD born by young women. A related finding in our study is 419 the choice made by people with CRD to hide their symptoms from family and community members 420 in a bid to keep the emotional burden of illness away from them. Young people with chronic illness 421 have been shown to be particularly circumspect about disclosure of their illness, frequently choosing 422 non-disclosure because of perceived fear of rejection, pity, and perceptions of being seen as 423 vulnerable or different(42). In the context of stigma, non-disclosure could also be a coping 424 mechanism to avoid stigmatization while shielding the family from its effects, as observed in our 425 study. 426

Limitations of the study 427
As this was a qualitative study, quantitative costs were not collected, and quality of life not 428 measured. Quantification of economic impact through a cost of illness study would lay the 429 foundation for a future evaluation of the cost-effectiveness of interventions to address CRD within 430 the health systems in these settings. However, the direct testimony of the people with CRD and members of their communities about their lived experience, provides important information about 432 the range of social, economic and psychological impacts that should be measured in our study 433 setting and insight into potential interventions to address them . Whilst the findings are not 434 statistically generalisable to populations in Sudan, Tanzania or sub-Saharan Africa more widely, the 435 common issues arising within these two different contexts are likely to be generalisable to similar 436 settings, whilst the context-specific issues point to the importance of locally developed 437 interventions. 438

Conclusion 439
Chronic respiratory diseases are associated with significant social and economic impacts among 440 people with CRD and their families in Sudan and Tanzania. While the impacts on livelihood and 441 economy resulted mainly from impairment of physical ability to carry out income-generating 442 activities and from expensive healthcare costs, the long-standing association of chronic cough with 443 TB and HIV/AIDs in our study settings was a major driver of stigma and social exclusion experienced 444 by people with CRD and their families. This highlights the negative central role of these chronic 445 stigmatizing diseases in framing perceptions and attitudes towards other illnesses in the community. 446 Context-appropriate social safety nets and systematic community health education and 447 sensitization would be required to address the economic and social impacts of CRD identified in this 448 study, as well as the broad causes and management of chronic cough in the communities. In 449 addition, provision of available, accessible and affordable care for CRD is necessary to break the 450 cycles of poverty, ill-health and stigma in these sub-Saharan African contexts 451 Declarations 452

Ethical considerations 453
The study was approved by the ethics committees of National Institute for Medical Research, 454 Tanzania