Abstract
Introduction
Leprosy, podoconiosis and lymphatic filariasis (LF) are three skin-related neglected tropical diseases. All three conditions can lead to temporary and permanent impairments. These impairments progressively worsen and are major determinants of stigma, discrimination and participation restrictions. Self-care is essential to prevent disabilities and chronic disease complications. Many persons with leprosy-related, LF-related and podoconiosis-related disabilities need to practice self-management routines their entire life. This is difficult without support and encouragement of others. The objective of this study was to assess the effectiveness of a family-based intervention in terms of physical outcomes related to prevention and self-management of disabilities due to leprosy, podoconiosis and LF and family quality of life and well-being compared with usual practice and care.
Methods and analysis
The study will use a cluster-randomised controlled trial design with two study arms. The project will be carried out in endemic districts in East and West Gojjam zones in the Amhara region in Ethiopia. Clusters consist of kebeles (lower administrative structures in the district) that have been merged, based on their geographical proximity and the number of cases in each kebele. A total of 630 participants will be included in the study. The intervention group will consist of 105 persons affected by leprosy, 105 persons affected by LF or podoconiosis, and 210 family members. The control group will consist of 105 persons affected by leprosy and 105 persons affected by LF or podoconiosis. The family-based intervention comprises an essential care package that consists of the following three main components: (1) self-management of disabilities, (2) economic empowerment and (3) psychosocial support. Participants in the control areas will receive usual practice and care. Data analysis includes, but is not limited to, calculating the percentage of change and corresponding 95% CI of physical impairment outcomes in each group, before and after the intervention is implemented, effect sizes, intention to treat and difference in difference analysis.
Ethics and dissemination
Ethical approval has been obtained from the Debre Markos University Health Sciences Institutional Research Ethics Review Committee. Results will be disseminated through peer-reviewed publications, conference presentations and workshops.
Trial registration number
PACTR202108907851342.
Keywords: Infectious diseases & infestations, EDUCATION & TRAINING (see Medical Education & Training), INFECTIOUS DISEASES, PREVENTIVE MEDICINE, PUBLIC HEALTH, WOUND MANAGEMENT
Strengths and limitations of this study.
This family-based intervention cluster-randomised controlled trial was preceded by a proof-of-concept study, in which the intervention was found feasible.
While self-management of disabilities is the main component of the family-based intervention, the essential care package goes beyond self-care and also includes economic empowerment and a psychosocial care component.
This study is led by and partly carried out by the Ethiopian National Association of Persons Affected by Leprosy, a large Ethiopian leprosy disabled persons’ organisation.
Inclusion of family members in self-care activities ensures sustainability of the intervention.
Because randomisation will be done at the level of kebeles, it will not be possible to conduct a blinded outcome assessment because research staff will be aware of the area they are in. It is not considered feasible to find people from outside the study areas to conduct the outcome assessment.
Introduction
Leprosy, podoconiosis and lymphatic filariasis (LF) are neglected tropical diseases (NTDs).1 NTDs are a group of communicable diseases that are among the most common conditions, particularly among the world’s poorest populations.2 3 These diseases predominate in rural and impoverished urban areas of low-income and middle-income countries.4 Worldwide, over one billion people have one or more NTDs.5 NTDs are ‘poverty promoting’ conditions; they cause suffering through acute illness, pain, long-term disability, early death and through mental and social consequences.2 4
Leprosy, podoconiosis and LF are three skin-related NTDs.1 All three conditions have skin manifestations such as patches, ulcers, wounds, nodules or localised swelling.6–9 They are caused by bacteria (leprosy), chronic exposure to red clay volcanic soil (podoconiosis) and nematode worms that are transmitted by mosquitoes (LF).7 8 10 Leprosy, podoconiosis and LF can lead to temporary and permanent impairments if not diagnosed and treated early.1 6 11 These impairments progressively worsen and are major determinants of stigma and participation restrictions.12–14
Social consequences of all three conditions may include reduced work and education opportunities, social isolation, exclusion and problems in interpersonal relationships, including marital problems.15–18 Psychological consequences may include feelings of shame, low self-esteem, mental distress, depression, anxiety, and decreased individual and family quality of life.15–18 In addition, these conditions may impose a social and economic burden on families.16 19 Family members may also experience stigma.16 20–23 Furthermore, costs for treatment and reduced ability to work may cause a financial burden for the entire family.16 19
Most impairments, such as wounds, swelling and contractures, are largely preventable.1 The most effective strategy for prevention of disabilities is early diagnosis and prompt treatment.24 Self-care is also an essential component of prevention of disabilities and for prevention of chronic disease complications.24–27 Relatively simple methods exist for self-management of impairments, such as daily washing of affected limbs, skin care, bandaging, exercises and the use of shoes.27 Most of these methods can be practised at home and are suitable for use across different skin-related NTDs.27–29 These self-care interventions have been found effective in, for example, reducing the incidence of acute dermatolymphangioadenitis in persons affected by podoconiosis and LF30 31 and in reducing ulcers among persons affected by leprosy.32 Because physical impairments are an important determinant of stigma, disease management is also an indirect intervention to reduce stigma.33
Many persons with leprosy-related, podoconiosis-related and LF-related disabilities need to practise self-management routines their entire life. This is difficult without support and encouragement of others. Family members can provide such support and encouragement. We recently conducted a proof-of-concept study in which we piloted a family-based intervention for prevention and self-management of disabilities due to leprosy, podoconiosis and LF in Ethiopia.34 This family-based intervention consisted of self-management of disabilities, awareness raising and economic empowerment, and was delivered during several monthly group meetings. Economic empowerment was an important component of the intervention, as income generation is essential for sustainable self-management and prevention of disabilities: without income, self-care items such as Vaseline and shoes cannot be bought. We found that the intervention had a positive effect on impairments and self-management of disabilities, family quality of life and stigma. However, sampling was not randomised, which means we could not determine the effectiveness of the intervention. To collect credible evidence for this new, previously piloted intervention, we aimed to conduct a similar study using a randomised controlled design.
Objectives
The primary objective of this study was to assess the effectiveness of a family-based intervention in terms of physical outcomes related to prevention and self-management of disabilities due to leprosy, podoconiosis or LF, and family quality of life and well-being compared with usual practice and care. Secondary objectives included the following: (1) to reduce the number of people who have an episode of depression, as measured with the Patient Health Questionnaire-9 (PHQ-9); (2) to reduce the level of stigma as measured with the SARI Stigma Scale (SSS), in-depth interviews and focus group discussions; (3) to improve social participation as measured with the Participation Scale (P-Scale); (4) to increase the number of people who have adequate knowledge of leprosy, LF and podoconiosis as measures with disease-specific Knowledge, Attitudes and Practices (KAP) measures; and (5) to empower people economically as measured by monthly household income, monthly financial contribution to the self-help group and in-depth interviews.
Methods and analysis
The protocol for this study is outlined further. This study protocol adheres to the Standard Protocol Items: Recommendations for Interventional Trials (SPIRIT) statement.35
Study design
The intervention consists of a cluster-randomised controlled trial (RCT), with two study arms. The two study arms consist of (1) the family-based intervention and (2) usual practice and care (control group).
Study setting
The project is carried out in endemic districts in East and West Gojjam zones in the Amhara region in Ethiopia (the proof-of-concept study was conducted in a different zone, the Awi zone). The Amhara region is the second largest state in population and is divided in 11 zones. All three conditions are endemic in the Amhara region. In 2019, Ethiopia had 3201 new patients with leprosy, 13% of the new patients had grade 2 disabilities.36 The prevalence of leprosy is highest in the Amhara, Afar and Oromiya regions.37 38 LF is endemic in the Amhara, Beneshangul-Gumuz, Southern Nations, Nationalities, and Peoples' Region (SNNPR) and Oromia regions. Three million people are estimated to be at risk of LF in the Amhara region.39 In addition, Ethiopia is estimated to have 25% (1 million cases) of the global burden of podoconiosis. Podoconiosis is spread out over one-fifth of the surface of Ethiopia, especially the Western part.37 40 41 The regions with the high prevalence of podoconiosis are Amhara, SNNPR, Oromiya and Beneshangul-Gumuz.40 41
East and West Gojjam zones are subdivided into 16 and 20 districts (woredas), respectively. The three districts selected for this study are Dega Damot and Dembecha districts (West Gojjam zone) and Enarge Enawga (East Gojjam zone). These districts have been selected based on their similarity in total population, sex ratio, number of urban/rural neighbourhoods (kebeles), number of hospitals, health centres and health posts, disease prevalence and lack of previous or ongoing leprosy, podoconiosis or LF-related work of other organisations (table 1). The latter to avoid possible contamination of the study results. The study is being conducted in real-world settings and populations.
Table 1.
Characteristics of the selected study areas
| Dega Damot district | Dembecha district | Enarge Enawga district | |
| Total population, n (%) | 181 325 (100) | 218 257 (100) | 172 939 (100) |
| Men | 89 756 (49.5) | 105 809 (48.5) | 86 297 (49.9) |
| Women | 91 156 (50.5) | 112 448 (51.5) | 86 642 (49.1) |
| Number of kebeles, n (%) | 36 (100) | 31 (100) | 35 (100) |
| Rural | 34 (94) | 27 (87) | 31 (89) |
| Urban | 2 (6) | 4 (13) | 4 (11) |
| Health facilities, n | |||
| Hospital | 1 | 1 | 1 |
| Health centre | 7 | 7 | 7 |
| Health post | 34 | 28 | 34 |
| Number of health extension workers working in the area | 88 | 60 | 76 |
| Percentage of total population that has podoconiosis (%) | >10 | 1–5 | >10 |
| Estimated number of persons with leprosy-related, podoconiosis-related or LF-related disabilities living in the area | Leprosy=132 Podoconiosis=352 |
Leprosy=135 Podoconiosis=1042 |
Leprosy=213 Podoconiosis or LF=797 |
| Geographical and background information |
|
|
|
| Previous or ongoing work with the target group in the area? | No | Yes, with persons affected by podoconiosis (no persons affected by podoconiosis will be included from this district). | No |
Data were collected from field census, health office reports and Molla et al and Berhe et al.50 51
LF, lymphatic filariasis.
Participants
People with leprosy-related impairments and people with LF-related or podoconiosis-related lymphoedema (‘persons affected’) will be included in this study. In addition, of each person affected, at least one adult family member will be included (eg, sibling, child, parent or grandparent of a person affected by leprosy, LF or podoconiosis).
People 15 years and above will be included in the study. All persons have to be residents of project areas of the study. All persons affected need to have leprosy-related, LF-related or podoconiosis-related impairments and have to be eligible to participate in self-care activities, as the focus is on skin and wound care of affected persons. Family members need to live in the same household as persons affected. People who are unable to coherently express themselves verbally (ie, are unable to understand and participate in an interview) will be excluded. In addition, persons affected who live alone will excluded.
Intervention
This RCT was preceded by a proof-of-concept study in which a family-based intervention was developed and found feasible.34 The family-based intervention consists of an essential care package that consists of the following three main components: (1) self-management of disabilities, (2) economic empowerment and (3) psychosocial support. All components of the intervention are family-based and family focused. Although not mentioned as a separate component, awareness raising of leprosy, LF and podoconiosis in the family and the community is an integral part of the intervention. The essential care package is described in more detail as follows:
Training sessions/group meetings for self-management and prevention of disabilities. Based on the proof-of-concept study, at least five group meetings will be held in a location that is most convenient for the participants. These sessions will be delivered in group format (several families participate with one person affected and one family member present per family) to introduce the family-based methods for self-management and prevention of disabilities. In the first session, basic training will be given to persons affected and their family members in using and giving psychosocial support, increasing prevention and self-management of disabilities skills, information about the disease, creating strategies to overcome barriers and facilitators to self-care. In the following training sessions, the research assistants support and guide all participating families (repeating the basic training given in the first session) and are available to clarify questions. During these meetings, physical impairment outcomes will routinely (monthly) be collected. Family members are encouraged to help their affected family member with self-care at home. (Each group will have approximately 20 participants; therefore, training for participants in the intervention group will not all be given at the same day/time.) We anticipate that the first group meeting will be held in February 2022. Group meetings will be conducted until September 2022.
Formation of self-help groups for economic empowerment. The project will facilitate the formation of self-help groups of affected persons; their family members are encouraged to join group meetings. The Ethiopian National Association of Persons Affected by Leprosy (ENAPAL), a large Ethiopian leprosy disabled persons’ organisation with a successful track record in establishing self-help groups, will coordinate and guide this part of the intervention. The facilitators of the project, trained by ENAPAL, will help to establish the self-help groups and will be present during the meetings but will not give guidance on the management of the groups. Management of the groups will be done by persons affected themselves; participants of the group will be asked to elect a ‘committee’ of persons affected. Each self-help group will collect a small contribution fee from its participants; these fees are used to provide loans for the participants of the self-help groups (microfinance). Self-help groups will also lobby for ‘benefits’, for example, the use of land, from the government. In addition, each self-help group participant and at least one of their family members will receive (one) vocational training. Income generation will benefit the whole family.
Psychosocial support will be part of the training sessions/group meetings for self-management and prevention of disabilities. Persons affected and their family members will be trained in using and giving psychosocial support.
The control group will undergo treatment as usual. Participants in the control areas will undergo the same basic training (one session) as the participants in the intervention group but will have no family members present during the training. When the intervention group has their additional four meetings (at least five meetings will be held), the participants in the control group will receive usual practice and care. In addition, they will receive information about existing mechanisms for economic empowerment (such as ‘funeral saving groups’ and other existing credit-saving initiatives).
Procedures
This study has two main phases. Each phase is briefly described as follows.
Phase I: preparatory phase. In this phase, a literature review will be conducted to guide the development of the psychosocial support component that will be added to the family-based intervention. In addition, the SSS, FQoL scale and P-Scale will be cross-culturally validated (the PHQ-9 has already been validated in Amharic42–44). We will assess conceptual, item, semantic, operational and measurement equivalence using a framework for cross-cultural equivalence testing based on the work of Herdman et al,45 Terwee et al46 and Stevelink and van Brakel.47 The KAP measure will be translated and pilot tested. A training workshop will be organised to train community health extension workers, local area health workers and the research team in research methods and family-based intervention. A list of persons affected registered in the community-level census that are eligible to participate in self-care activities will be prepared. Persons affected by leprosy, podoconiosis or LF and their family members will be recruited. A database will be established to monitor the routine intervention activities. Baseline data will be collected by the research assistants, and the results will be analysed by the researcher.
Phase II: implementation and evaluation of the family-based intervention. In this phase, the intervention will be implemented: at least five training sessions and family meetings will be held. This training is done by the researcher (who has extensive experience in providing training, self-care practices and the three conditions included in this study), with support from the research assistants and with at least one community health extension worker present at the meeting. Research assistants will receive a 4-day training on how to implement the intervention; this training is facilitated by the researcher and the project manager. In addition, each training session is carried out using standard operating procedures that have been developed using the WHO’s integrated morbidity management for LF and podoconiosis,48 the Ethiopian Ministry of Health’s LF and podoconiosis morbidity management and disability prevention guidelines and International Federation of Anti-Leprosy Associations (ILEP)’s guideline for prevention of disabilities in leprosy.49 As has been described in detail previously,34 participants in the intervention and control areas will receive basic tools to practise self-care (Vaseline, a bucket, shoes and soda). In this phase, the effectiveness and acceptability of the intervention will be evaluated (feasibility has already been established in the proof-of-concept study that was recently conducted34). This will be done by collecting the same information as in the baseline study (table 2), a few weeks and 1 year after implementation of the intervention. In addition, interviews will be conducted to collect most significant change stories and to assess the impact qualitatively. Because randomisation will be done at the level of kebeles, it will not be possible to conduct a blinded outcome assessment because research staff will be aware of the area they are in. It is not considered feasible to find people from outside the study areas to conduct the outcome assessment. All components of the study will be conducted in Amharic, the official language of Ethiopia and language spoken in the study areas.
Table 2.
Outcome measures
| Type of outcome | Specific outcome | Outcome measures |
| Implementation outcomes | Acceptability | Qualitative (IDI and FGD) |
| Disability management practices | Observations (field notes), qualitative (IDI and FGD) | |
| Economic empowerment | Registration of attendance of persons affected organisation group meetings, number of loans disbursed and total amount of money disbursed | |
| Effectiveness (persons affected level) | Physical impairment outcomes | For persons affected by leprosy:
For persons affected by podoconiosis and LF:
|
| Physical well-being | IDI | |
| Family quality of life | FQoL Scale, IDI | |
| Perceived, experienced and internalised stigma | SARI Stigma Scale | |
| Social participation | Participation Scale | |
| Mental well-being42–44 | Patient Health Questionnaire-9 | |
| Disease knowledge53 54 | Disease-specific KAP measure | |
| Attitudes towards the disease and persons affected by the disease | Qualitative (IDI and FGD) | |
| Economic empowerment | Monthly household income, monthly financial contribution to the self-help group, qualitative (IDI) | |
| Effectiveness (family member level) | Family quality of life | FQoL Scale, qualitative (IDI) |
| Perceived, experienced and internalised stigma | IDI | |
| Mental well-being42–44 | Patient Health Questionnaire-9 | |
| Disease knowledge53 54 | Disease specific KAP measure | |
| Attitudes towards (persons affected by) the disease | Qualitative (IDI and FGD) | |
| Economic empowerment | Monthly household income, monthly contribution to the self-help group, qualitative (IDI) | |
| Impact at community level | Most significant changes | Qualitative (IDI and FGD) |
| Impact assessment (to evaluate the change in the target population and communities) | Qualitative (IDI and FGD) |
FGD, focus group discussion; FQoL, Beach Centre Family Quality of Life; IDI, in-depth interview; KAP, Knowledge, Attitudes and Practices.
Outcomes
Table 2 details the outcomes measured during this study, including the methods that will be used to measure the outcomes. Physical impairment outcomes are the primary outcome measures. Acceptability, family quality of life, stigma, social participation, mental well-being, disease knowledge, attitudes and economic empowerment are secondary outcomes.
Participant timeline
The participant timeline, in line with SPIRIT recommendations, can be found in table 3.
Table 3.
Participant timeline
| Study period* | ||||||
| Enrolment | Preallocation | Allocation | Postallocation | |||
| Time point | T0 | Tx | T1 | T2 | ||
| Enrolment | ||||||
| Eligibility screen | X | |||||
| Informed consent | X | |||||
| Allocation | X | |||||
| Intervention | ||||||
| Group meetings | X | |||||
| Assessments | ||||||
| Questionnaires | ||||||
| SARI Stigma Scale | X | X | X | |||
| Beach Centre Family Quality of Life scale | X | X | X | |||
| Participation Scale | X | X | X | |||
| Patient Health Questionnaire-9 | X | X | X | |||
| Disease-specific Knowledge, Attitudes and Practices measure | X | X | X | |||
| Routine data | ||||||
| Physical impairment outcomes | X | X | X | X | ||
| Group meeting attendance | X | X | X | X | ||
| In-depth interviews | X | X | X | |||
| Focus group discussions | X | X | X | |||
*T0 denotes before the intervention/baseline. Tx denotes monthly monitoring during the intervention (routine data collection). T1 indicates 1-month postintervention. T2 indicates 1-year postintervention.
Sample size
A total of 630 participants, consisting of 420 persons affected and 210 family members, will be included in the study. It is difficult to distinguish LF and podoconiosis based on clinical features under field conditions, and the distinction between these conditions does not matter with regard to the outcomes of this study; therefore, persons affected by both these conditions are treated as one group. There will be one intervention and one control group for persons affected by leprosy, and one for persons affected by LF or podoconiosis. Family members are only included in the intervention group. The intervention group will consist of 105 persons affected by leprosy, 105 persons affected by LF or podoconiosis, and 210 family members. The control group will consist of 105 persons affected by leprosy and 105 persons affected by LF or podoconiosis. The sample size calculation is based on data from the proof-of-concept study.34 In the proof-of-concept study, 43% of the participants had leg impairments (wounds, nodules and/or infections) at intake. During the final assessment, the last session participants attended, and the number of participants with leg impairments had dropped to 21%. A sample size calculation for two proportions (proportion 1: 43%, proportion 2: 21%) with a significance of 0.05 and a power of 90% would give a total sample size of 92 participants in each group. We expect that the loss to follow-up will be no more than 15% (we do not expect a higher loss to follow-up, as participants will be followed up at home). Our sample size will therefore be 105 persons affected in each group. The kebeles have been selected in such a way that they are similar to each other; we therefore do not anticipate a cluster effect in the current outcomes.
Recruitment
Potential participants will be approached via community-level enumeration, healthcare settings, persons affected organisations, community leaders and by word of mouth. The recruitment period is 6 months, starting in October 2021. Once participants are enrolled, they will be followed up during the study period up to 12 months in the nearby health centre or health posts. In the case of loss to follow-up, participants will be visited in their home.
Allocation
The three districts will be randomly divided into clusters to implement either the family-based intervention or usual practice and care (control group). A complete enumeration of persons with the three diseases has been conducted in each district; kebeles (a lower administrative structure in the district) have been merged into ‘clusters’ based on their similarity, including their population characteristics, geographical proximity, the presence of a health centre and the number of cases in each kebele. Each cluster consists of three to five kebeles on average (ranging from two to seven), and all clusters have at least one health centre in the area. Sixteen clusters have been identified in the three study districts: Feresbet, Taeme, Dama Markos, Arefa, Damot Tsion, Sekela, Chat Warka (in Dega Damot district), Debre Work, Felege, Tenguma, Gedeb, Shifere, Metiya, Wonfit (in Enarge Enawga district), Dembecha town and Wad (in Dembecha district). Out of these 16 clusters, a total of 4 clusters for leprosy and 6 clusters for podoconiosis and LF will be randomly selected. The intervention and control areas will be randomly selected by putting the cluster names in a cup or box and randomly drawing names. We will ensure that the number of intervention and control areas (clusters) in each district is equal. A list will be prepared with all patients (leprosy, podoconiosis/LF) living in the project areas that are registered at community-level enumeration and that are eligible to participate in self-care activities. Persons affected to be included in the study will be selected by stratified systematic sampling with a random start from a list of persons affected registered at the primary healthcare centre. This is done by selecting the first person affected on the list at random (by throwing dice), and then selecting every Xth patient on the list, based on the total number needed. Four separate lists will be created: two for persons affected by leprosy (one intervention and one control) and two for persons affected by LF or podoconiosis (one intervention and one control).
Blinding
Due to the nature of the intervention, participants cannot be blinded.
Data management
Confidentiality and anonymity of data will be ensured in data collection, data storage, analysis and publication. Research assistants who will collect the data will be fully trained in proper data management, maintenance of confidentiality and ensuring privacy during data collection. All data will be collected in Ethiopia. Only data that have been fully anonymised will be shared with the international research team. The project leader of this study will take full responsibility for ensuring the appropriate storage and security of data. Data will be kept for 5 years and will be destroyed after this time frame when no longer required.
Data analysis
Quantitative data will be entered in a database created using EpiData software. Analyses will start once baseline data have been collected. Simple descriptive methods will be used to generate a demographic profile of the study sample. Differences between participants in the intervention and control groups, including demographic information and physical impairment outcomes, will be evaluated using the Mann-Whitney U test or t-test for continuous variables and the χ2 statistic for categorical variables. In addition, the mean with SD (or median with IQR, depending on the distribution of the data) of the total scores of the measures used will be calculated per participant group and per study area. The percentage change and corresponding 95% CI of physical impairment outcomes in each group, before and after the intervention is implemented and the statistical significance of this difference using a Z-test for differences between proportions will be calculated. Effect sizes will also be calculated. Stepwise multivariate regression with backward elimination will be done to examine what factors will have an independent effect on the outcomes. Data analysis will be done in the software packages Epi Info and SPSS Statistics version 27. We will also use intention to treat for categorical/nominal variables and difference in difference analysis for continues variables to evaluate the effectiveness of the intervention.
Qualitative data—the recordings of the in-depth interviews and focus group discussions—will be transcribed, translated to English and analysed using open, inductive coding and content analysis. Similar phrases with recurring themes will be coded in a qualitative software programme (MAXQDA) and clustered together in tables to identify connections.
Patient and public involvement
This research will be led by and partly carried out by ENAPAL (a leprosy disabled persons’ organisation). Persons affected by leprosy, LF and podoconiosis will assist the researchers in analysis of the data by helping to put issues in perspective and context. We will seek to employ and train persons affected as research assistants or at least those who have a family member affected by an NTD or with a disability.
Ethics and dissemination
Ethics
Ethical approval has been obtained from the Debre Markos University Health Sciences Institutional Research Ethics Review Committee (approval number HSC/R/C/Ser/Co/11/13). All participants will be fully informed about the nature and objective of the study and of confidentiality of the data prior to data collection. Written informed consent will be obtained from each participant prior to data collection. For participants who cannot read, an impartial witness will be present for the whole informed consent discussion. She or he will sign and date the consent form after the consent giver has done so. All people who are participating in the research will be provided with a participant information sheet. No incentives will be paid to participants.
Dissemination
A publication plan has been developed, which lists several planned articles for publication in scientific journals. All articles will be published in peer-reviewed, open access journals. The results of the study will also be shared through international conferences and at (working) meetings with international researchers and national policy makers and healthcare staff. A meeting will be organised at the end of the study to disseminate the results in the communities in the study areas. In addition, we aimed to share updates of the study through the ILEP newsletter and the Sasakawa Health Foundation newsletter.
bmjopen-2021-056620supp001.pdf (1.6MB, pdf)
Supplementary Material
Footnotes
Contributors: ATvN, MWA, TT and AS designed the study and were responsible for funding acquisition. MWA is the principal investigator of the study. MWA, NAM and TT are responsible for the implementation of the study in Ethiopia. MWA will lead data analysis with support from ATvN and NAM. ATvN drafted the manuscript. All authors read and approved the final version of the manuscript.
Funding: This work is supported by the Leprosy Research Initiative (grant number 708.20.17). The funders had no role in study design, decision to publish or preparation of the manuscript.
Competing interests: None declared.
Patient and public involvement: Patients and/or the public were not involved in the design, conduct, reporting or dissemination plans of this research.
Provenance and peer review: Not commissioned; externally peer reviewed.
Supplemental material: This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.
Ethics statements
Patient consent for publication
Not applicable.
References
- 1.Mitjà O, Marks M, Bertran L, et al. Integrated control and management of neglected tropical skin diseases. PLoS Negl Trop Dis 2017;11:e0005136. 10.1371/journal.pntd.0005136 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 2.Feasey N, Wansbrough-Jones M, Mabey DCW, et al. Neglected tropical diseases. Br Med Bull 2010;93:179–200. 10.1093/bmb/ldp046 [DOI] [PubMed] [Google Scholar]
- 3.Herricks JR, Hotez PJ, Wanga V, et al. The global burden of disease study 2013: what does it mean for the NTDs? PLoS Negl Trop Dis 2017;11:e0005424. 10.1371/journal.pntd.0005424 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 4.Hotez PJ, Molyneux DH, Fenwick A, et al. Incorporating a rapid-impact package for neglected tropical diseases with programs for HIV/AIDS, tuberculosis, and malaria. PLoS Med 2006;3:e102. 10.1371/journal.pmed.0030102 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 5.World Health Organization . Integrating neglected tropical diseases into global health and development: fourth WHO report on neglected tropical diseases. World Health Organization, 2017. [Google Scholar]
- 6.Suzuki K, Akama T, Kawashima A, et al. Current status of leprosy: epidemiology, basic science and clinical perspectives. J Dermatol 2012;39:121–9. 10.1111/j.1346-8138.2011.01370.x [DOI] [PubMed] [Google Scholar]
- 7.Davey G. Podoconiosis, non-filarial elephantiasis, and lymphology. Lymphology 2010;43:168. [PubMed] [Google Scholar]
- 8.Babu S, Nutman TB. Immunology of lymphatic filariasis. Parasite Immunol 2014;36:338–46. 10.1111/pim.12081 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 9.Chandler DJ, Grijsen ML, Fuller LC. With bare feet in the soil: Podoconiosis, a neglected cause of tropical lymphoedema. Dermatology 2021;237:1–12. 10.1159/000506045 [DOI] [PubMed] [Google Scholar]
- 10.Lastória JC, Abreu MAMMde, de AM. Leprosy: review of the epidemiological, clinical, and etiopathogenic aspects - part 1. An Bras Dermatol 2014;89:205–18. 10.1590/abd1806-4841.20142450 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 11.Lyons OTA, Modarai B. Lymphoedema. Surgery 2013;31:218–23. 10.1016/j.mpsur.2013.02.003 [DOI] [Google Scholar]
- 12.van Brakel WH, Sihombing B, Djarir H, et al. Disability in people affected by leprosy: the role of impairment, activity, social participation, stigma and discrimination. Glob Health Action 2012;5:18394. 10.3402/gha.v5i0.18394 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 13.Tora A, Franklin H, Deribe K, et al. Extent of podoconiosis-related stigma in Wolaita zone, southern Ethiopia: a cross-sectional study. Springerplus 2014;3:647. 10.1186/2193-1801-3-647 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 14.Zeldenryk LM, Gray M, Speare R, et al. The emerging story of disability associated with lymphatic filariasis: a critical review. PLoS Negl Trop Dis 2011;5:e1366. 10.1371/journal.pntd.0001366 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 15.Van't Noordende AT, Aycheh MW, Schippers AP. An exploration of family quality of life in persons with leprosy-, lymphatic filariasis- and podoconiosis-related disabilities and their family members in Ethiopia. Trans R Soc Trop Med Hyg 2020;114:1003–12. 10.1093/trstmh/traa090 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 16.van 't Noordende AT, Aycheh MW, Schippers A. The impact of leprosy, podoconiosis and lymphatic filariasis on family quality of life: A qualitative study in Northwest Ethiopia. PLoS Negl Trop Dis 2020;14:e0008173. 10.1371/journal.pntd.0008173 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 17.Molla YB, Tomczyk S, Amberbir T, et al. Patients’ perceptions of podoconiosis causes, prevention and consequences in East and West Gojam, Northern Ethiopia. BMC Public Health 2012;12:1–7. 10.1186/1471-2458-12-828 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 18.Krishna Kumari A, Harichandrakumar KT, Das LK, et al. Physical and psychosocial burden due to lymphatic filariasis as perceived by patients and medical experts. Trop Med Int Health 2005;10:567–73. 10.1111/j.1365-3156.2005.01426.x [DOI] [PubMed] [Google Scholar]
- 19.Awofeso N. Stigma and socio-economic reintegration of leprosy sufferers in Nigeria. Acta Leprol 1996;10:89–91. [PubMed] [Google Scholar]
- 20.Cassidy T, Worrell CM, Little K, et al. Experiences of a community-based lymphedema management program for lymphatic filariasis in Odisha state, India: an analysis of focus group discussions with patients, families, community members and program volunteers. PLoS Negl Trop Dis 2016;10:e0004424. 10.1371/journal.pntd.0004424 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 21.de Stigter DH, de Geus L, Heynders ML. Leprosy: between acceptance and segregation. community behaviour towards persons affected by leprosy in eastern Nepal. Lepr Rev 2000;71:492–8. 10.5935/0305-7518.20000051 [DOI] [PubMed] [Google Scholar]
- 22.Nicholls PG, Bakirtzief Z, Van Brakel WH, et al. Risk factors for participation restriction in leprosy and development of a screening tool to identify individuals at risk. Lepr Rev 2005;76:305–15. 10.47276/lr.76.4.305 [DOI] [PubMed] [Google Scholar]
- 23.Tekola F, Bull S, Farsides B, et al. Impact of social stigma on the process of obtaining informed consent for genetic research on Podoconiosis: a qualitative study. BMC Med Ethics 2009;10:13. 10.1186/1472-6939-10-13 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 24.Cross H. The prevention of disability as a consequence of leprosy. Lepr Rev 2015;86:208–12. 10.47276/lr.86.3.208 [DOI] [PubMed] [Google Scholar]
- 25.Narahari SR, Bose KS, Aggithaya MG, et al. Community level morbidity control of lymphoedema using self care and integrative treatment in two lymphatic filariasis endemic districts of South India: a non randomized interventional study. Trans R Soc Trop Med Hyg 2013;107:566–77. 10.1093/trstmh/trt054 [DOI] [PubMed] [Google Scholar]
- 26.Douglass J, Graves P, Gordon S. Self-Care for management of secondary lymphedema: a systematic review. PLoS Negl Trop Dis 2016;10:e0004740. 10.1371/journal.pntd.0004740 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 27.Chandler D, Fuller L. The skin—A common pathway for integrating diagnosis and management of NTDs. Trop Med Infect Dis 2018;3:101. 10.3390/tropicalmed3030101 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 28.Jansen B. Systematic review: skin and wound management interventions related to skin-neglected tropical diseases., 2017. Available: https://www.leprosy-information.org/resource/systematic-review-skin-and-wound-management-interventions-related-skin-neglected-tropical?aresult=0
- 29.Pryce J, Mableson HE, Choudhary R, et al. Assessing the feasibility of integration of self-care for filarial lymphoedema into existing community leprosy self-help groups in Nepal. BMC Public Health 2018;18:201. 10.1186/s12889-018-5099-0 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 30.Stocks ME, Freeman MC, Addiss DG. The effect of hygiene-based lymphedema management in lymphatic filariasis-endemic areas: a systematic review and meta-analysis. PLoS Negl Trop Dis 2015;9:e0004171. 10.1371/journal.pntd.0004171 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 31.Negussie H, Molla M, Ngari M, et al. Lymphoedema management to prevent acute dermatolymphangioadenitis in Podoconiosis in northern Ethiopia (GoLBeT): a pragmatic randomised controlled trial. Lancet Glob Health 2018;6:e795–803. 10.1016/S2214-109X(18)30124-4 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 32.Ebenso J, Muyiwa LT, Ebenso BE. Self care groups and ulcer prevention in Okegbala, Nigeria. Lepr Rev 2009;80:187–96. 10.47276/lr.80.2.187 [DOI] [PubMed] [Google Scholar]
- 33.Hofstraat K, van Brakel WH. Social stigma towards neglected tropical diseases: a systematic review. Int Health 2016;8 Suppl 1:i53–70. 10.1093/inthealth/ihv071 [DOI] [PubMed] [Google Scholar]
- 34.Van't Noordende AT, Wubie Aycheh M, Tadesse T, et al. A family-based intervention for prevention and self-management of disabilities due to leprosy, Podoconiosis and lymphatic filariasis in Ethiopia: a proof of concept study. PLoS Negl Trop Dis 2021;15:e0009167. 10.1371/journal.pntd.0009167 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 35.Chan A-W, Tetzlaff JM, Gøtzsche PC, et al. Spirit 2013 explanation and elaboration: guidance for protocols of clinical trials. BMJ 2013;346:e7586. 10.1136/bmj.e7586 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 36.World Health Organization . Weekly epidemiological record, 4 September 2020. Global leprosy (Hansen disease) update, 2019: time to step-up prevention initiatives. 95. WHO, 2020: 417–40. [Google Scholar]
- 37.Deribe K, Meribo K, Gebre T, et al. The burden of neglected tropical diseases in Ethiopia, and opportunities for integrated control and elimination. Parasit Vectors 2012;5:1–15. 10.1186/1756-3305-5-240 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 38.Berhe D, Haimanot RT, Tedla T, et al. Epidemiological pattern of leprosy in Ethiopia: a review of the control programmes. Lepr Rev 1990;61:258–66. 10.5935/0305-7518.19900027 [DOI] [PubMed] [Google Scholar]
- 39.Rebollo MP, Sime H, Assefa A, et al. Shrinking the lymphatic filariasis map of Ethiopia: reassessing the population at risk through nationwide mapping. PLoS Negl Trop Dis 2015;9:e0004172. 10.1371/journal.pntd.0004172 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 40.Deribe K, Cano J, Newport MJ, et al. Mapping and modelling the geographical distribution and environmental limits of Podoconiosis in Ethiopia. PLoS Negl Trop Dis 2015;9:e0003946. 10.1371/journal.pntd.0003946 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 41.Deribe K. Mapping the spatial distribution and epidemiology of Podoconiosis in Ethiopia: implications for disease control 2016.
- 42.Hanlon C, Medhin G, Selamu M, et al. Validity of brief screening questionnaires to detect depression in primary care in Ethiopia. J Affect Disord 2015;186:32–9. 10.1016/j.jad.2015.07.015 [DOI] [PubMed] [Google Scholar]
- 43.Gelaye B, Williams MA, Lemma S, et al. Validity of the patient health Questionnaire-9 for depression screening and diagnosis in East Africa. Psychiatry Res 2013;210:653–61. 10.1016/j.psychres.2013.07.015 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 44.Degefa M, Dubale B, Bayouh F, et al. Validation of the PHQ-9 depression scale in Ethiopian cancer patients attending the oncology clinic at Tikur Anbessa specialized Hospital. BMC Psychiatry 2020;20:1–7. 10.1186/s12888-020-02850-3 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 45.Herdman M, Fox-Rushby J, Badia X. A model of equivalence in the cultural adaptation of HRQoL instruments: the universalist approach. Qual Life Res 1998;7:323–35. 10.1023/A:1008846618880 [DOI] [PubMed] [Google Scholar]
- 46.Terwee CB, Bot SDM, de Boer MR, et al. Quality criteria were proposed for measurement properties of health status questionnaires. J Clin Epidemiol 2007;60:34–42. 10.1016/j.jclinepi.2006.03.012 [DOI] [PubMed] [Google Scholar]
- 47.Stevelink SAM, van Brakel WH. The cross-cultural equivalence of participation instruments: a systematic review. Disabil Rehabil 2013;35:1256–68. 10.3109/09638288.2012.731132 [DOI] [PubMed] [Google Scholar]
- 48.Deribe K, Kebede B, Tamiru M, et al. Integrated morbidity management for lymphatic filariasis and Podoconiosis, Ethiopia. Bull World Health Organ 2017;95:652–6. 10.2471/BLT.16.189399 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 49.ILEP . ILEP learning guide four: how to prevent disability in leprosy, 2006. Available: https://ilepfederation.org/wp-content/uploads/2020/02/LG4.pdf
- 50.Molla YB, Tomczyk S, Amberbir T, et al. Podoconiosis in East and West Gojam zones, Northern Ethiopia. PLoS Negl Trop Dis 2012;6:e1744. 10.1371/journal.pntd.0001744 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 51.Berhe B, Legese H, Mardu F, et al. Epidemiology and sex differences of Podoconiosis in Ethiopia: a systemic review and meta-analysis. Heliyon 2021;7:e05446. 10.1016/j.heliyon.2020.e05446 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 52.Brandsma JW, Van Brakel WH, Van BWH. Who disability grading: operational definitions. Lepr Rev 2003;74:366–73. 10.47276/lr.74.4.366 [DOI] [PubMed] [Google Scholar]
- 53.Van't Noordende AT, Lisam S, Ruthindartri P, et al. Leprosy perceptions and knowledge in endemic districts in India and Indonesia: differences and commonalities. PLoS Negl Trop Dis 2021;15:e0009031. 10.1371/journal.pntd.0009031 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 54.van 't Noordende AT, Korfage IJ, Lisam S, et al. The role of perceptions and knowledge of leprosy in the elimination of leprosy: a baseline study in Fatehpur district, Northern India. PLoS Negl Trop Dis 2019;13:e0007302. 10.1371/journal.pntd.0007302 [DOI] [PMC free article] [PubMed] [Google Scholar]
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