Utilising the ‘Getting to Outcomes®’ Framework in Community Engagement for Development and Implementation of Sickle Cell Disease Newborn Screening in Kaduna State, Nigeria

Background: Sickle Cell Disease (SCD) has been designated by WHO as a public health problem in sub-Saharan Africa, and the development of newborn screening (NBS) is crucial to the reduction of high SCD morbidity and mortality. Strategies from the field of implementation science can be useful for supporting the translation of NBS evidence from high income countries to the unique cultural context of sub-Saharan Africa. One such strategy is community engagement at all levels of the healthcare system, and a widely-used implementation science framework, “Getting to Outcomes®” (GTO), which incorporates continuous multilevel evaluation by stakeholders about the quality of the implementation. Objectives: (1) to obtain critical information on potential barriers to NBS in the disparate ethnic groups and settings (rural and urban) in the healthcare system of Kaduna State in Nigeria; and, (2) to assist in the readiness assessment of Kaduna in the implementation of a sustainable NBS programme for SCD. Methods: Needs assessment was conducted with stakeholder focus groups for two days in Kaduna state, Nigeria, in November 2017. Results: The two-day focus group workshop had a total of 52 participants. Asking and answering the 10 GTO accountability questions provided a structured format to understand strengths and weaknesses in implementation. For example, we found a major communication gap between policy-makers and user groups. Conclusion: In a two-day community engagement workshop, stakeholders worked successfully together to address SCD issues, to engage with each other, to share knowledge, and to prepare to build NBS for SCD in the existing healthcare system.


Introduction
Sickle Cell Disease (SCD) has been designated by the World Health Organisation (WHO) as a public health problem in sub-Saharan Africa [1][2][3]. It is projected that, unless specific action is taken, the burden of disease will continue to increase into 2050, especially in Nigeria and Democratic Republic of Congo, where this increase is estimated to be more than 100% [4]. The number of annual births with SCD is estimated to be 100,000 to 150,000 in Nigeria. Our pilot Newborn screening (NBS) study of infants up to six months old in an area within Kaduna State, Nigeria, reported an incidence of 1.7% [4,5], which suggests that over 4000 babies with SCD are born every year (based on 240,000 annual overall births per state). Consistent with WHO's call to action, national and regional policies for the management and control of SCD are required, especially in the view of limited resources across most of sub-Saharan Africa. SCD represents an urgent health burden, both in terms of mortality and morbidity. It is estimated that it accounts for 8-16% of under-five mortality in sub-Saharan Africa [6]. Mortality among children with SCD in Africa is estimated at 50% to 90% by 10 years of age, mostly from preventable infections [2].
Effective management of SCD should incorporate NBS with the prevention of infections (including pneumococcal septicaemia and malaria), parental education, and support at all levels of healthcare provision to enable the timely recognition of SCD complications and health maintenance. The development of NBS programmes in sub-Saharan Africa is crucial to the reduction of high infant mortality. These programmes must be guided by empirical evidence, often accumulated in high income countries, such as United States of America (USA) and United Kingdom (UK), and simultaneously fit within the unique cultural context of sub-Saharan Africa (which is very distinct from the setting of the original clinical trial). This often poses a challenge in implementation where the original trial does not fit with the local context. Strategies from the field of implementation science, defined as the "scientific study of methods to promote the systematic uptake of research findings and other evidence-based practices into routine practice, and, hence, to improve the quality and effectiveness of health services and care" [7] can be useful for supporting the translation of evidence from clinical trials to implementation in contexts vastly different from that originally employed in the clinical trial, such as Kaduna State in Africa. One such strategy is community engagement at all levels of the healthcare system [8].
Kaduna State in northern Nigeria was the country's old colonial capital, it is a microcosm of the entire country, and has a population of over eight million made up of over 60 different ethnic groups, with 23 local governments, three geopolitical (senatorial) zones, with over 30 health care facilities for secondary care, two academic institutions (tertiary care and undergraduate training), and five teaching hospitals (tertiary care) hospitals. The academic institutions are Ahmadu Bello University Teaching Hospital and Barau Dikko Teaching Hospital, Kaduna State University, and the other four tertiary care level hospitals are National Eye Centre, National Ear Care Centre, Federal Neuro-Psychiatric Hospital, and 44 Nigerian Armed Forces Reference Hospital. The State offers free healthcare for pregnant women and children up to five years of age. Kaduna State Primary Health Care Agency is led by an Executive Secretary to oversee primary care centres and clinics in conjunction with the local governments.
We embarked on community engagement as the initial step to informing the development and implementation of an NBS programme for SCD in Kaduna State. Community engagement has been broadly defined as involving communities in information giving, consultation, decision-making, planning, co-design, governance, and delivery of services [9]. This was an early phase of sustained engagement with a broad range of community representatives to be inclusive and aimed for equal partnership. Furthermore, the application of implementation science within health systems is of benefit to the development and implementation of health interventions. Despite favourable evidence in clinical trials, programmes often fail to reach their desired outcomes in the real world due to limitations outside the trial environment and challenges with implementation.
Implementation science provides strategies to help guide implementation, therefore improving access to evidence-based services that fit with the culture of the population in need. The first phase usually consists of descriptive, formative research to better understand the major implementation challenges and to design potential strategies to overcome these [10]. We employed a widely-used implementation science framework, "Getting to Outcomes ® " (GTO), which incorporates continuous multilevel evaluation by stakeholders about the quality of the implementation [11][12][13]. GTO is a 10-step system of accountability that guides the user through the process of planning, monitoring, and evaluating programmes. The continuous evaluation facilitates adaptation of the programme to local capacity and motivation for change, which maximizes the chances of programme success.
The objectives of the community engagement were two-fold. First, to obtain critical information pertaining to disparate ethnic groups and settings (rural and urban), including potential barriers to a successful NBS with the Kaduna State healthcare system and subsequent policy implementation. Second, to assist in the readiness assessment of Kaduna State in the implementation of a sustainable NBS programme for SCD.

Methods
Qualitative research methodology was employed in a two-day focus group workshop with an identical format over the two days in Kaduna. A representative group of participants were invited for each of the two focus group sessions. These comprised parents of children with SCD, adults with SCD, representatives of patient association and support groups, community leaders, health professionals, and policy-makers from the three health zones in Kaduna state, including nurses and midwives. Community health extensions workers from primary healthcare centres, doctors, and nurses from general and teaching hospitals were among the participants. In addition, five participants from the neighbouring Niger State were invited to the first focus group session to highlight the anticipated differences between states.
Focus group discussions were facilitated by a faculty of five international and local experts in SCD from the UK and Nigeria, including paediatricians, haematologists, a psychologist, and a professional in community engagement. The focus group format included brief introductory lectures on SCD and NBS. This was followed by a series of 10 questions based on the ten-point GTO framework for discussion. Each participant was given the opportunity and was encouraged to be candid with their responses and discussions in a relaxed and open atmosphere and speak in any language of their preference. Proceedings were transcribed by a professional scribe and audio-recorded. Subsequently, transcripts were produced from the audio recordings by two professionals that were experienced in transcribing (LG) and qualitative research (EG). Their combined report was reviewed by the facilitators of focus groups for accuracy and consistency (KAA, BO, and BI).

Results
There was a total of 52 participants for the two-day focus group workshop (Table 1). Discussions based on the GTO questions and additional issues are summarised by themes generated below. In addition, core themes identified within the GTO framework and categorised by the type of participant or institution are presented in Table 2.

Discussion
Readiness is part of GTO, but what we did in this focus group was broader than readiness alone. We organized the findings by GTO steps, which served to (1) understand differences in perspectives across the different levels (this is important for addressing potential barriers) and (2) to remain accountable for implementation. From an implementation standpoint, one of the challenges faced in health care settings is the transport of interventions from a research trial to naturalistic setting. There are many factors that get in the way of successful implementation in naturalistic settings, especially in complex settings, like the multilevel healthcare structure in Kaduna. Differences in the vision, needs, resources, and goals of different levels of the health system may get in the way of successful implementation. This complexity is compounded by differences in contextual factors between the setting of the original clinical trial of the intervention and the local context where the intervention is being implemented. Most likely adaptations are needed to achieve the similar outcomes of a well-funded clinical trial in a developing country. In order to identify which adaptations are needed, and at which level these adaptations are needed, community engagement at each healthcare level is needed.
SCD poses a major public health problem in Nigeria. Community engagement as a first step to developing and implementing a sustainable NBS programme was carried out by SCD experts from UK, USA, and Nigeria, working with a charity in Nigeria called the Sickle Cell Cohort Research (SCORE) Foundation. Focus group discussions employing an implementation science approach with patients, parents, community leaders, doctors, nurses, and community health workers allowed active participation and important information to be gathered about the difficulties and solutions for testing newborn babies in these communities, including cultural and religious beliefs.
This study employed a well-known implementation framework to guide community engagement. Through focus groups, we uncovered certain areas where potential barriers to implementation may exist and where certain adaptations may be needed to improve the chances of achieving programmatic success. For example, we found a major communication gap between policy-makers and user groups. There is an absence of patient-users consultation within the state policy framework and therefore the lack of opportunity to incorporate their views in service planning and implementation. Asking and answering the 10 GTO accountability questions provided a structured format to understand the strengths and weaknesses in the implementation setting. This led nicely to the development of plans that support quality implementation. In this way, the hospitals will be more prepared for implementation and increase their chances of programmatic success.
The goals and objectives were addressed. Outcomes include the opportunity for participants working together to address SCD issues, to network, and engage with each other. Shared knowledge by participants, greater awareness of what is in place albeit on a small scale. Some myths and misinformation were addressed. There is no doubt that the importance of NBS for SCD programme development and implementation in Kaduna State, Niger State and the entire country cannot be over emphasised. To ensure the sustainability of the programme, the government has to be fully committed to it by providing the legal framework, policies, and adequate funding. It is also important to note that issues such as lack of public awareness and concerns could be barriers to a successful programme. Therefore, it is necessary to educate the general public through media campaigns, and advocate in partnership with the support of religious and traditional leaders within.

Summary and Conclusions
The two-day workshop successfully set the stage for the development and implementation plan of the NBS programme for SCD communities. Recommendations for the next steps to developing a Kaduna State NBS for SCD programme were made to the State's Commissioner of Health, and subsequently an initial four-day training workshop was organised prior to step by step implementation: (i) Procurement of reagents (ii) collection of blood spots from one local government area (1/23) of the state to test robustness of specimen collection, transportation to the laboratory, analysis turnaround time; result disclosure to families, (iii) counselling to families; and, (iv) referral to treatment clinic. A number of key themes from this 'Getting To Outcomes' (10 steps) assessment process require urgent implementation by Kaduna State through the setting up of steering committee to address the issues that were raised regarding the Objectives of a SCD programme, Perceptions, and Implementation of NBS. For the State to adopt Community based approach by involving Volunteer Community Mobilisers (VCMs) and Traditional Birth Attendants (TBAs) for maximum benefit and to ensure that a robust monitoring and evaluation process is in place.