Summary of evidence
Our scoping review process identified 24 publications that have addressed maternal and congenital syphilis in Indigenous Peoples worldwide. We observed a substantial increase in publications on this topic, 50% of which were published in the last five years. This is in line with the persistence of a high burden of syphilis-related disease globally [
2], and with WHO goals for eliminating congenital syphilis [
1], although we did not find a specific mention of this issue in Indigenous Peoples in these publications.
The high proportion of studies conducted in Brazil (50%) together with the absence of studies in many countries that have a significant indigenous population and effective indigenous participation in decision-making processes in health and definition of research priorities are noteworthy. The high frequency of studies in Brazil seems to result from the promotion and encouragement of regional research through the implementation of a science and technology unit of the Ministry of Health – the Oswaldo Cruz Foundation (FIOCRUZ), in the state of Mato Grosso do Sul [
12,
20]. This institution offered a professional master’s course in border health surveillance in the region [
11,
21]. Another contributing factor was the conducting of extensive research to analyze the cost-effectiveness of implementing rapid tests for syphilis and HIV in indigenous populations in remote areas, where access to health and laboratory networks is minimal [
16,
17,
22,
23,
31].
The absence of studies in countries with a substantial indigenous population, such as China, India, Indonesia, Ethiopia, Philippines, Canada, Mexico, and New Zealand, might be due to low scientific interest in the subject, use of non-indexed descriptors, or nonspecific keywords in publications, and grey literature not identified in searches. Other possible explanations include concern about further stigmatizing this population, lack of prioritization of this as an issue, and limited capacity to self-conduct and report on the data.
According to the United Nations, even though the most accepted criterion for defining indigenousness is self-identification, there is no consensus on a single defining characteristic or term, given the enormous diversity of traditional peoples and societies. Although the use of the word “indigenous” is widespread, it has a negative connotation in some regions. In this case, it is usually exchanged for other words, such as tribes, first peoples/nations, aboriginals, and ethnic groups [
35], which makes it difficult to identify all relevant studies on a purely bibliographic basis. However, our review was exhaustive in including these terms in search strategies.
The studies which investigated data quality related to maternal and congenital syphilis in Indigenous Peoples (DQ) analyzed three quality dimensions: completeness, coverage, and consistency. The quality of the records was found to be poor, showing underreporting of cases and incompleteness of notifications, particularly for socioeconomic, treatment, and ethnicity variables, which can lead to information or selection biases. The poor data quality contributes to underestimating the burden of these diseases in Indigenous Peoples and reduces accuracy in estimating ethnic-racial inequities and identifying social determinants of syphilis; this in turn limits the usefulness of the data to inform social and health policies for disease control [
11,
15]. As in the report by Palma-Pinedo and colleagues [
14] specifically for syphilis in the Peruvian Amazon, Sousa and colleagues [
36] highlighted the poor quality of indigenous data in the health information systems in Brazil. This was reported to be due to the high turnover of the workforce, lack of training, work overload, and lack of integration between different information systems. Such evidence indicates the need for investment in the expansion of financial, material, and workforce resources, training for syphilis diagnosis, management and surveillance, awareness of health workers of intercultural work, and strengthening of surveillance systems to improve the quality of health care and information systems, including those specifically for syphilis.
The studies addressing the diagnosis, offer, access, and use of health services by Indigenous Peoples (DAH) highlighted existing barriers to health care, such as low prenatal care coverage and limited laboratory resources for diagnosis. These conditions are responsible for missed opportunities for diagnosis and treatment, resulting in under-detection and under-reporting of maternal and congenital syphilis. The intersection of several geopolitical and health care management levels and heterogeneous levels of health care organization, particularly in remote areas, are strong limitations for surveillance and timely diagnosis and treatment, creating further difficulties for the control and prevention of congenital syphilis. Syphilis control in these populations, particularly in remote areas [
17,
37], presupposes the expansion of access to health care through universal systems, with an emphasis on primary care, especially prenatal care and delivery, which is socio-culturally sensitive [
19]. It includes the offer of rapid testing for syphilis and other STIs [
16,
23], the expansion of screening for syphilis in the general population, in the most vulnerable groups and during pregnancy and childbirth [
16,
27], health worker training [
20,
30], intersectoral articulation and coordinated responses, with community participation [
24,
25,
29].
As for the frequency of maternal and congenital syphilis in Indigenous Peoples (DF), most studies reported the prevalence of syphilis based on seroprevalence surveys, and only three studies reported maternal syphilis detection rates or incidence of congenital syphilis. There was heterogeneity in the frequency of outcomes between different ethnic groups, territories, regions, and countries, with a higher burden of disease in comparison to their counterparts [
12,
31], as widely documented for other diseases and conditions [
4]. The location in regions with limited access to health care, on the international border, or in remote areas, proved to be a relevant context for the high frequency of maternal and congenital syphilis [
11,
22] a fact also reported by Hierink and colleagues [
37] about other infectious diseases in low- and middle-income countries. However, the comparison of frequencies between indigenous populations in remote and urban areas in the same region suggests that proximity to urban areas may be an additional risk for the transmission of syphilis in these populations [
33]. The results indicate that the life context of Indigenous Peoples plays a central role in the frequency of maternal and congenital syphilis, influencing the infectivity, transmission rate, and duration of the disease [
37,
38].
Studies on syphilis determinants in indigenous populations (DT) are scarce. All identified studies had a cross-sectional design and reported factors associated with maternal syphilis. No study investigated factors associated with congenital syphilis. In general, factors associated with maternal syphilis were similar to those described in the literature for the non-indigenous population [
28,
30,
32,
33]. Contextual factors of particular relevance to Amazonian Indigenous Peoples were highlighted, such as the high degree of intrusion in indigenous territories, including illegal mining or deforestation, and the increased mobility of the population to access resources in regional attraction centers [
31]. A recent analysis of the risk of the geographic spread of COVID-19 in Indigenous Peoples in Brazil, using proxy indicators of mobility and intrusion in indigenous territories, confirmed these context-related vulnerabilities to infectious diseases [
39,
40]. The lack of a significant association with other supposed risk variables is possibly attributed to the low power of the studies due to the relatively low number of events of interest in small populations.
Gestational or congenital syphilis in indigenous people (OCS) was significantly associated with the outcomes of prematurity, stillbirth, perinatal mortality, and low birth weight [
24,
26,
30,
32,
34]. It is noteworthy that indigenous populations, in general, constitute small population groups [
41], thus the absolute frequency of health outcomes related to syphilis tends to be neglected. The rarity of events of interest in health investigations often results in methodological challenges, given the low power of studies to investigate disease determinants.
Knowledge gaps and future research
The few existing publications on the frequency of maternal and congenital syphilis are mainly based on prevalence data and seem somewhat biased due to underreporting of the disease, lack of comprehensive data quality, and restriction of analysis to some countries in the Americas and Oceania. Future research should provide robust estimates of the frequency of maternal and congenital syphilis in Indigenous Peoples in a wide range of populations and contexts to better represent diversity, vulnerability, and inequalities. Performing routine analyses of data quality, estimating underreported cases, and generating possible correction factors at different geographic scales could support the monitoring of ethnic-racial trends in syphilis inequalities. The linkage of datasets addressing all phases of the health-disease process could improve data completeness and consistency, enabling innovative and robust analysis of determinants and impacts of public policies for controlling the disease. It is also necessary that health and surveillance systems be improved by continuously increasing the quality of health information systems data, availability of up-to-date and reliable sociodemographic data, and eliminating access barriers to universal, public healthcare provided by qualified professionals.
Theoretical models for analyzing the determinants of maternal and congenital syphilis in Indigenous Peoples must consider contextual variables that enable the identification of modifiable social determinants beyond those already well-established for the general population to support social and health policies implemented at the state level. The investigation of incident outcomes (maternal and congenital syphilis), possibly prioritizing analytical epidemiological designs, could lead to risk estimates and a better understanding of causality and consistently monitor the temporal trend of the disease. Research on syphilis outcomes in the conceptus is also scarce and deserves to be expanded into different contexts. Assessment of the impact of social and health policies on maternal and congenital syphilis and its outcomes in the conceptus is non-existent in the accessed literature and should be considered a hotspot for future research.