{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T05:12:28Z","timestamp":1772169148162,"version":"3.50.1"},"update-to":[{"DOI":"10.12688\/wellcomeopenres.14838.1","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2019,9,11]],"date-time":"2019-09-11T00:00:00Z","timestamp":1568160000000}}],"reference-count":36,"publisher":"F1000 Research Ltd","license":[{"start":{"date-parts":[[2019,9,11]],"date-time":"2019-09-11T00:00:00Z","timestamp":1568160000000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100007601","name":"Horizon 2020","doi-asserted-by":"publisher","award":["734584"],"award-info":[{"award-number":["734584"]}],"id":[{"id":"10.13039\/501100007601","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004440","name":"Wellcome Trust","doi-asserted-by":"publisher","award":["206016"],"award-info":[{"award-number":["206016"]}],"id":[{"id":"10.13039\/100004440","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["wellcomeopenresearch.org"],"crossmark-restriction":false},"short-container-title":["Wellcome Open Res"],"abstract":"<ns4:p>Global concern broke out in late 2015 as thousands of children in Brazil were born with microcephaly, which was quickly linked to congenital infection with Zika virus (ZIKV). ZIKV is now known to cause a wider spectrum of severe adverse outcomes\u2014congenital Zika syndrome (CZS)\u2014and also milder impairments. This study aimed to explore the social and economic impacts of CZS in Brazil. Data was collected through mixed methods across two settings: Recife City and Jaboat\u00e3o dos Guararapes in Pernambuco State (the epicentre of the epidemic), and the city of Rio de Janeiro (where reports of ZIKV infection and CZS were less frequent). Data was collected May 2017-January 2018. Ethical standards were adhered to throughout the research. In-depth qualitative interviews were conducted with: mothers and other carers of children with CZS (approximately 30 per setting), pregnant women (10-12 per setting), men and women of child-bearing age (16-20 per setting), and health professionals (10-12 per setting). Thematic analysis was undertaken independently by researchers from at least two research settings, and these were shared for feedback.<\/ns4:p>\n                  <ns4:p>A case-control study was undertaken to quantitatively explore social and economic differences between caregivers of a child with CZS (cases) and caregivers with an unaffected child (controls). We aimed to recruit 100 cases and 100 controls per setting, from existing studies. The primary caregiver, usually the mother, was interviewed using a structured questionnaire to collect information on: depression, anxiety, stress, social support, family quality of life, health care and social service use, and costs incurred by families. Multivariable logistic regression analyses were used to compare outcomes for cases and controls. Costs incurred as a result of CZS were estimated from the perspective of the health system, families and society. Modelling was undertaken to estimate the total economic burden of CZS from those three perspectives.<\/ns4:p>","DOI":"10.12688\/wellcomeopenres.14838.2","type":"journal-article","created":{"date-parts":[[2019,9,11]],"date-time":"2019-09-11T10:23:20Z","timestamp":1568197400000},"page":"127","update-policy":"https:\/\/doi.org\/10.12688\/wellcomeopenres.crossmark-policy","source":"Crossref","is-referenced-by-count":11,"title":["Social and economic impacts of congenital Zika syndrome in Brazil: Study protocol and rationale for a mixed-methods study"],"prefix":"10.12688","volume":"3","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8952-0023","authenticated-orcid":false,"given":"Hannah","family":"Kuper","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3600-7250","authenticated-orcid":false,"given":"Tereza 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status","group":{"name":"current-referee-status","label":"Current Referee Status"}},{"value":"10.21956\/wellcomeopenres.16167.r34013, Debora Diniz, Law School, University of Bras\u00edlia, Bras\u00edlia, Brazil, 07 Nov 2018, version 1, 1 approved, 1 approved with reservations","URL":"https:\/\/wellcomeopenresearch.org\/articles\/3-127\/v1#referee-response-34013","order":0,"name":"referee-response-34013","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"<b>Hannah Kuper<\/b>; \n<i>Posted: 14 Aug 2019<\/i>; Dear Dr Diniz, &nbsp; Thank you for the detailed and thoughtful comments on the manuscript, which we have attempted to address, as follows: 1.&nbsp;&nbsp;&nbsp;&nbsp; We have now clarified in the methods that in order to contrast the two regions, the quantitative analyses were stratified by region, with formal testing for the presence of effect modification. Data collection methods differed between the two sites, as you correctly noted, which is now clarified as a limitation in the discussion with consideration for how this issue was mitigated as far as possible and dealt with in the analysis and reporting of the results papers. &nbsp; 2.&nbsp;&nbsp;&nbsp;&nbsp; We have clarified in the text the interviewers were all female Brazilian social scientists from the local region. &nbsp; 3.&nbsp;&nbsp;&nbsp;&nbsp; We have emphasised that the major local\/regional response was the elimination of the vaccine, while national\/international efforts also focussed on vaccines, diagnosis, etc. &nbsp; 4.&nbsp;&nbsp;&nbsp;&nbsp; We have revised the text throughout to place greater emphasis on the social and economic impacts on women\/mothers, and have largely replaced \u201cfamily\u201d with \u201chousehold\u201d. &nbsp; We have now used the term CZS throughout, unless microcephaly was expressly meant, we tried to clarify in the text how these conditions were different. &nbsp; 5.&nbsp;&nbsp;&nbsp;&nbsp; We agree that a focus on medical costs alone is insufficient. Therefore, in our data collection we have included costs of direct costs (e.g. travel or medical costs), indirect costs (e.g. lost productivity) and coping costs (e.g. need to borrow money and pay interest), as now clarified in Table 1 and in the description of quantitative data collected. The economic modelling will use a time horizon of 10 years, since there are great uncertainties in the long-term costs and outcomes for these children (e.g. potential impact of educational exclusion or welfare changes). &nbsp; 6.&nbsp;&nbsp;&nbsp;&nbsp; As described in point 5, we have attempted to provide more information on how economic data was collected. &nbsp; 7.&nbsp;&nbsp;&nbsp;&nbsp; As clarified in point 1, we have tried to explain how we \u201ccontrast\u201d the two settings and take account for the differences in context of interviews in interpreting the findings. &nbsp; 8.&nbsp;&nbsp;&nbsp;&nbsp; The time of training of interviewers differed between the sites because of differences in the experience of those recruited. &nbsp; 9.&nbsp;&nbsp;&nbsp;&nbsp; We have included additional information on how we attempted to preserve the anonymity and protect women who may have made statements about abortion during the interview, including through anonymization and safe storage of data, restrictions on data sharing, and the inclusion of doctors in the team who can be gate-keepers for the data. We would, of course, value your expertise in navigating these difficult issues.  Best wishes  Hannah and colleagues","URL":"https:\/\/wellcomeopenresearch.org\/articles\/3-127\/v2#referee-comment-3926","order":1,"name":"referee-comment-3926","label":"Referee Comment","group":{"name":"article-reports","label":"Article Reports"}},{"value":"10.21956\/wellcomeopenres.16851.r36441, Anne\u00a0C. Wheeler, RTI International, Research Triangle Park, NC, USA, 22 Oct 2019, version 2, indexed","URL":"https:\/\/wellcomeopenresearch.org\/articles\/3-127\/v2#referee-response-36441","order":2,"name":"referee-response-36441","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"This study was supported by The Wellcome Trust and the Department for International Development (grant: 206016\/Z\/17\/Z). This study was also supported by a supplementary grant from the European Union\u2019s Horizon 2020 research and innovation programme, under Zika-PLAN grant agreement no. 734584. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.","order":3,"name":"grant-information","label":"Grant Information"},{"value":"This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.","order":0,"name":"copyright-info","label":"Copyright"}]}}