BackgroundThe vast majority of residents of high-income countries (>90%) reportedly have high access to safely managed drinking water, the highest level of service under the Sustainable Development Goal (SDG) framework. Owing perhaps to the widely held perception of near universal access to high-quality water services in these countries, the burden of waterborne disease in these contexts is understudied.MethodsWe conducted a systematic review of estimates of the disease burden attributed to drinking water in countries where >90% of the population has access to safely managed drinking water per official SDG monitoring. We searched PubMed, Web of Science, and SCOPUS for articles published before September 10, 2021.FindingsWe identified 24 studies reporting estimates for the disease burden attributable to microbial contaminants. Across these studies, the population-weighted average burden of gastrointestinal illness risks attributed to drinking water was about 3,529 annual cases per 100,000 people. Beyond exposure to infectious agents, we also identified 10 studies reporting disease burden, predominantly, cancer risks, associated with chemical contaminants. Across these studies, the pooled population-weighted average of excess cancer cases attributable to drinking water was 1.8 annual cancer cases per 100,000 people.InterpretationThese estimates exceed WHO-recommended normative targets for disease burden attributable to drinking water and highlight that there remains important preventable disease burden in these contexts. However, the available literature was scant and limited in geographic scope, disease outcomes, range of microbial and chemical contaminants, and inclusion of subpopulations that could most benefit from water infrastructure investments. These subpopulations include rural, low-income communities; Indigenous or Aboriginal peoples; and populations marginalized due to discrimination by race, ethnicity, or socioeconomic status. Studies quantifying drinking water-associated disease burden in countries with reportedly high access to safe drinking water, with a focus on specific subpopulations and promoting environmental justice, are needed.Research in contextEvidence before this studyAlthough the burden of disease associated with unsafe drinking water in low- and middle-income countries has been subject of much research, there is uncertainty about the magnitude and nature of this disease burden in high-income countries and countries where access to safely managed drinking water is >90%. To assess the magnitude of this burden in countries with high levels of access to high quality water, we conducted a systematic review of estimates of the disease burden attributed to drinking water in countries where >90% of the population has access to safely managed drinking water as defined by the WHO/UNICEF Joint Monitoring Programme for Water Supply, Sanitation and Hygiene (JMP). We searched PubMed, Web of Science, and SCOPUS for relevant articles published before September 10, 2021. We searched for articles including “burden of disease” OR “disease burden” OR “gastrointestinal illness” anywhere in the text. From among these articles, we selected those with the terms “drinking water” OR “tap water.” From this reduced set, we selected articles reporting on countries with >90% safe water access and including estimates of cases, hospitalizations, or deaths related to drinking water contamination. Additional articles were identified from consultations with subject-matter experts. Only articles in English were recovered, which may have biased our results towards specific countries.Added value of this studyTo our knowledge, this is the first global review of its kind to identify burden of waterborne disease estimates associated with microbiological contaminants as well as chemical contaminants found in drinking water, specifically in countries with reportedly high access (>90%) to safely managed drinking water. Despite the perception that high quality water is ubiquitous in these countries, we found that burden estimates in these countries exceeded WHO-recommended targets for disease burden attributable to unsafe drinking water. This review also highlighted the gaps in our understanding of waterborne disease burden across and within countries where access to high quality drinking water is common.Implications of all the available evidenceEven though the levels of access to safely managed drinking water are high in many high-income and some middle-income countries, there remains a potentially important preventable disease burden in these high-income countries. Our review suggests that further burden research expanding the scope of contaminants, disease outcomes, geographic area, and vulnerable subpopulations within countries is necessary. Currently, available evidence is insufficient to support rational decision-making and to prioritize public investment to protect the health of marginalized communities. These groups include rural, low-income communities; Indigenous or Aboriginal peoples; and populations marginalized due to discrimination by race, ethnicity, or socioeconomic status.