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This study aimed to systematically review the available evidence on dental, occlusion, and craniofacial anomalies in individuals with FA and to describe the occurrence of these anomalies in a cohort from two Brazilian referral centers.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Materials and Methods<\/jats:title>\n                    <jats:p>Electronic searches were conducted across six databases, supplemented by manual searches and gray literature. The Brazilian cohort included 46 patients diagnosed with FA.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      A total of 19 articles describing 158 cases of FA were analyzed. The estimated prevalence of dental\/craniofacial anomalies ranged from 13.3% to 71.4%. In our cohort, 93.5% of patients exhibited anomalies, primarily root abnormalities (69.6%), tooth rotation (54.3%), and tooth agenesis (26%). Males (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.031) and individuals with endocrine disorders (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.047) were more likely to experience alterations in eruption and\/or exfoliation. Anomalies in size and shape were significantly more prevalent among individuals who had undergone hematopoietic stem\u2010cell transplantation at age 14 or older (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.002).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>The high occurrence of dental\/craniofacial anomalies in individuals with FA suggests that these anomalies are part of the disease's phenotypic spectrum, emphasizing the need to expand and standardize the diagnostic criteria of the disease.<\/jats:p>\n                  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