A Qualitative Study in Busoga, Uganda

BMJ Open: first published as 10.1136/bmjopen-2020-040992 on 23 February 2021. Downloaded from

PEER REVIEW HISTORY

BMJ Open publishes all reviews undertaken for accepted manuscripts. Reviewers are asked to complete a checklist review form (http://bmjopen.bmj.com/site/about/resources/checklist.pdf) and are provided with free text boxes to elaborate on their assessment. These free text comments are reproduced below.

ARTICLE DETAILS

TITLE (PROVISIONAL) Reactions to and explanations for the birth of a baby with albinism: A qualitative study in Busoga, Uganda. AUTHORS Taylor, Julie; Bradbury-Jones, C; Ogik, Peter; Kawuma, Fazira; Betts, Jane; Lund, Patricia

VERSION 1 – REVIEW

REVIEWER Professor Eleanor Ross Centre for Social Development in Africa University of Johannesburg South Africa REVIEW RETURNED 02-Jul-2020

GENERAL COMMENTS I applaud the authors on this beautifully written manuscript and their sensitivity in dealing with this important issue. Please include a section on limitations of the study. It would also be helpful to mention any efforts made to enhance trustworthiness of the data.

REVIEWER Giorgio Brocco Freie Universitaet Berlin REVIEW RETURNED 16-Jul-2020 http://bmjopen.bmj.com/

GENERAL COMMENTS The article, untitled “Reactions to and explanations for the birth of a baby with albinism: A qualitative study in Uganda”, seeks to explore reactions, explanations, and narratives bout the birth of children with albinism in the Busoga sub-region (kingdom) in the Eastern part of Uganda. The article is relevant in several aspects and deals with a topic that has galvanized lots of interest among scholars, journalists, politicians, NGO workers, and members of on September 26, 2021 by guest. Protected copyright. (inter)national institutions over the last years. The analysis provides the readers with significant research data and information on the various religious-moral reactions and explanations given for the births of children with albinism. However, specific issues have to be tackled to improve the article further. Hence, I consider the article as publishable on the condition of minor revisions. As it follows, I endeavor to indicate broad suggestions and queries and detailed amendments and additions to improve the manuscript. Therefore, I hope my comments can help the authors to revise the article. The first comment regards the article’s title. Since the research has been carried out in the Busoga region with its specific historical, cultural, and social configurations, it appears to be inaccurate to refer these birth narratives to the entire national territory. Hence, the exact region where the research was conducted should appear in the title.

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Could the authors provide more information on the social, economic, and political aspects of Uganda and the Busoga region? (P.6) The author should underline the role played by NGOs and governmental organizations in spreading biomedical and religious ideas about albinism. Furthermore, they should highlight their research positionality in the field in the sense that the opinions/ideas expressed by the research participants could be influenced by the presence of and recruitment process by NGO staff members and representatives. Why do the authors choose to use “witchcraft and sorcery” as terms for referring to traditional/local healing practices? Which is the interrelation between religious beliefs (e.g. Christian, Muslim, traditional religions) and local healing practices? I am of the opinion that the authors should integrate some research on healing practices/witchcraft carried out in Uganda with the anthropological studies conducted in Malawi, Tanzania, and other Southern African countries. This can help the reader to figure out the moral-religious constellations around the births of children with albinism in this specific region (p.7). Can the authors expand on the concept of personhood? In which ways is it relevant for the understanding of “unruly” births? In relation to this question, How is a “normal” child conceived in cultural and social terms in the Busoga region and Uganda at large? (p.8) Could the authors explain the concept of “animal totem” in relation to other moral-religious ideas? (p.9) How is the verb “produce” translated in the Lusonga language? See page 11. In referring to the hypo-pigmented skin of people with albinism the author should use the following adjectives: hypo-pigmented or lighter skin.

http://bmjopen.bmj.com/ VERSION 1 – AUTHOR RESPONSE

Reviewer 1 Comments Response I applaud the authors on this beautifully written Thank you very much for your kind comments. manuscript and their sensitivity in dealing with this important issue on September 26, 2021 by guest. Protected copyright.

Please include a section on limitations of the The limitations of the study are included in the study. bulleted section following the abstract (as per author instructions). We have expanded on these:

Although the unique context of the Busoga sub- region provides important insights, knowledge and practices are contextual and this limits generalisability.

Whilst participants appeared to speak freely and openly about their experiences, their ideas and

BMJ Open: first published as 10.1136/bmjopen-2020-040992 on 23 February 2021. Downloaded from

opinions could have been influenced by the albinism society and its representatives.

It would also be helpful to mention any efforts Thank you, this is important. Although this is made to enhance trustworthiness of the data. also covered in the accompanying COREQ checklist, we have added a further statement to this effect in the main text.

To enhance trustworthiness of the study, two researchers (JT, PL) undertook the analysis independently then met to agree coding trees and emergent themes. Two more analysts (CB- J, PO) ‘sense’ checked the themes and helped refine these. Analytic discussions informed the final interpretations and narrative.

Reviewer 2 Comments

The analysis provides the readers with Thank you for your kind comments. significant research data and information on the various religious-moral reactions and explanations given for the births of children with albinism.

The first comment regards the article’s title. We have amended the title to reflect this Since the research has been carried out in the important point: Busoga region with its specific historical, cultural, and social configurations, it appears to http://bmjopen.bmj.com/ be inaccurate to refer these birth narratives to the entire national territory. Hence, the exact Reactions to and explanations for the birth of a region where the research was conducted baby with albinism: A qualitative study in should appear in the title. Busoga, Uganda.

Could the authors provide more information on We have added more detail to this section to on September 26, 2021 by guest. Protected copyright. the social, economic, and political aspects of address this important point: Uganda and the Busoga region? (P.6) The author should underline the role played by NGOs and governmental organizations in spreading biomedical and religious ideas about The study was undertaken in the Busoga sub- albinism. region (kingdom) in the Eastern part of Uganda, around Lake Victoria with Jinja as the industrial and economic hub. Most of Busoga is very rural and marked by chronic poverty, with heavy reliance on fishing, subsistence farming and roadside trade. Christian and Islamic religions are mainstream, but parallel African beliefs and practices inform rites at birth, marriage and death. Almost 800 persons with albinism are registered with the local association, Source of

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the Nile Union of Persons with Albinism (SNUPA). SNUPA’s strong advocacy roots, government connections and wide networks along with a keenness to increase their evidence base made Busoga an ideal setting for conducting this research. There is scant information about albinism in the region and SNUPA is at the forefront of working with child protection and human rights organisations to debunk myths and persuade the government to take action.

Furthermore, they should highlight their This is a good point, thank you. We have added research positionality in the field in the sense this as a limitation to the study: that the opinions/ideas expressed by the research participants could be influenced by the presence of and recruitment process by NGO staff members and representatives. Whilst participants appeared to speak freely and openly about their experiences, their ideas and opinions could have been influenced by the albinism society and its representatives.

Why do the authors choose to use “witchcraft With respect, we do not think we have used the and sorcery” as terms for referring to words witchcraft or sorcery when referring to traditional/local healing practices? Which is the traditional healing practices. Rather these were interrelation between religious beliefs (e.g. words used by participants to describe Christian, Muslim, traditional religions) and local healing practices? I am of the opinion that the treatment/reaction/behaviours/attitudes to http://bmjopen.bmj.com/ authors should integrate some research on persons with albinism. There can be a spectrum healing practices/witchcraft carried out in of traditional practices, from using a few herbs Uganda with the anthropological studies to those where belief in ‘black’ magic is strong conducted in Malawi, Tanzania, and other (as in Uganda and explained in the section Southern African countries. This can help the before research questions). Indeed there is reader to figure out the moral-religious often a confused picture in between. If we have constellations around the births of children with mistakenly conflated the two then please do

albinism in this specific region (p.7). on September 26, 2021 by guest. Protected copyright. give a specific example so that we can amend.

We have referred to a number of studies in other countries, your own in Tanzania being particularly illuminative, but also in Malawi, South Africa, Zimbabwe, Zambia and others, as well as Uganda. This is not an anthropological paper and the word and reference limitations do not allow us to expand further here. However, we are mindful that there is more work to be done on integrating these strands and would welcome discussions about a joint future paper to do this.

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Can the authors expand on the concept of ‘Normal’ in this context and in many other personhood? In which ways is it relevant for the communities means non-disabled and non- understanding of “unruly” births? In relation to different. The birth of a boy is celebrated, that of this question, How is a “normal” child conceived a girl often less so. Within this hierarchy those in cultural and social terms in the Busoga region with a visible disability fare badly and people and Uganda at large? (p.8) with albinism worst of all. As we have explained, they are not seen as normal at birth.

On page 4 we explain:

Among populations such as in Uganda, the appearance of pale babies with albinism born to black parents is unusual, unexpected, and can cause alarm among all those at the birth, including the mother and attendants. Families may be stigmatised or vilified for having a child with albinism and mothers are often left to bring up multiple children on their own [7]. Imafidon [8] has argued that people with albinism are considered both physically and ontologically different in Black African communities, as less than a human being.

And on page 17 we explain:

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Within African communities, those who lack the expected features of personhood (e.g by forms of disability, mental ill health, albinism etc) are denied the intrinsic worth of being a person.

on September 26, 2021 by guest. Protected copyright.

Could the authors explain the concept of “animal In discussion with anthropological colleagues totem” in relation to other moral-religious ideas? we have learned that ‘totem’ was an incorrect (p.9) term in Africa. We have changed this to ‘familiar’ throughout. We have also added a sentence in the discussion:

those with albinism in Africa may be seen as spirits (or the reincarnation of spirits) as they are ‘white’ whereas ‘real’ people in African societies are black and this may be linked to the sighting of animal familiars also.

BMJ Open: first published as 10.1136/bmjopen-2020-040992 on 23 February 2021. Downloaded from

How is the verb “produce” translated in the Our Ugandan co-authors assure us that Lusoga language? See page 11. ‘produce’ means ‘to give birth’ and we have added this explanation where appropriate.

In referring to the hypo-pigmented skin of Thank you, we have amended this. people with albinism the author should use the following adjectives: hypo-pigmented or lighter skin.

Editorial Comments

- Please revise the ‘Strengths and limitations’ We have amended this section as advised. section of your manuscript (after the abstract). This section should contain five short bullet points, no longer than one sentence each, that relate specifically to the methods. The results of the study should not

Please state whether consent was verbal or Consent was both verbal and written and we written. added this appropriately.

Authors must include a statement in the Thank you for this comment. We would prefer to methods section of the manuscript under the use the heading ‘Community engagement and sub-heading 'Patient and Public Involvement'. involvement’ in preference to 'Patient and Public

Involvement' because it aligns with the term This should provide a brief response to the following questions: advocated by the WHO when researching with communities, rather than in a health context. How was the development of the research However, this was not allowed on resubmission. http://bmjopen.bmj.com/ question and outcome measures informed by We have added this section to the methods: patients’ priorities, experience, and preferences? How did you involve patients in the design of this study? Patient and public involvement Were patients involved in the recruitment to and conduct of the study? The SNUPA team included people with How will the results be disseminated to study albinism, disabled people and Ugandan on September 26, 2021 by guest. Protected copyright. participants? volunteers. SNUPA was central from inception, For randomised controlled trials, was the burden including developing the research questions and of the intervention assessed by patients themselves? design, undertaking data collection and Patient advisers should also be thanked in the assisting in the analysis. People with albinism contributorship statement/acknowledgements. were recruited through SNUPA’s networks and were invited to a stakeholder event to discuss and refine the findings at the end of the study. Educational and advocacy materials were distributed to all participants and the findings used by both SNUPA and their partner NGO Advantage Africa.

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We have also referred to the participants in the acknowledgements and to additional SNUPA advisers in the author contribution.

VERSION 2 – REVIEW

REVIEWER Prof. Eleanor Ross University of Johannesburg South Africa REVIEW RETURNED 02-Oct-2020

GENERAL COMMENTS Please include a brief mention of the study limitations.

REVIEWER Giorgio Brocco Freie Universitaet Berlin, Germany REVIEW RETURNED 16-Oct-2020

GENERAL COMMENTS The authors have done well in responding to my comments and concerns, and I hope that my suggestions helped them to improve their article further. The qualitative research on people with albinism in Busoga region, Uganda, is now far better situated and enhances both the empirical and theoretical dimensions of the research on mothering, local explanations, and health-related stigma, and social acceptance of children and parents with albinism. I have no further substantive issues. However, I think that a joint future paper on various aspects of witchcraft, sorcery, and traditional healing in various African contexts is necessary.

VERSION 2 – AUTHOR RESPONSE http://bmjopen.bmj.com/

Reviewer: 1 Dr. Eleanor Ross, University of Johannesburg

Comments to the Author: Please include a brief mention of the study limitations [in the discussion]. on September 26, 2021 by guest. Protected copyright.

Response:

Although the study limitations are described in the article summary following the abstract, we have added 100 words more on these in the discussion (p17-18), as follows:

A strength of this qualitative study is that it provides hitherto unreported birth stories and the reactions and explanations surrounding them. To that end, it has generated new knowledge in terms of understandings about albinism in Uganda. Until now, such insights have remained within the spheres of local communities. The sample size was unusually large for a qualitative study, which gives us confidence that a range of experiences have been captured. However, there were some challenges. Although the unique context of the Busoga sub-region provides important insights as already stated, we recognise that knowledge and practices are contextual and that this limits generalisability.

BMJ Open: first published as 10.1136/bmjopen-2020-040992 on 23 February 2021. Downloaded from

Reviewer: 2 Dr. Giorgio Brocco, Freie Universität Berlin Comments to the Author: The authors have done well in responding to my comments and concerns, and I hope that my suggestions helped them to improve their article further. The qualitative research on people with albinism in Busoga region, Uganda, is now far better situated and enhances both the empirical and theoretical dimensions of the research on mothering, local explanations, and health-related stigma, and social acceptance of children and parents with albinism. I have no further substantive issues. However, I think that a joint future paper on various aspects of witchcraft, sorcery, and traditional healing in various African contexts is necessary.

Response: Thank you very much for your valuable suggestions that have indeed helped improve and enhance our article. We agree that a future paper is necessary.