Background: Iodine is needed for the production of thyroid hormones, which are essential for infant growth and development. Given that there are wide variations in breast milk iodine concentration (BMIC) and urinary iodine concentration (UIC), it is unclear if BMIC is associated with UIC in populations residing in iodine sufficient or deficient areas. Aim: To investigate if BMIC can be used as a biomarker for iodine status in lactating women and children <2 years of age. Methods: Electronic databases; PubMed, Web of Science and Scopus were searched until year 2021, for studies investigating the relationship between BMIC and UIC. Studies were reviewed for eligibility, according to inclusion and exclusion criteria, followed by data extraction, according to the PRISMA guidelines. Results: Overall, 51 studies met the criteria for inclusion in the systematic review. BMIC ranged from 18 to 1153 µg/L. In iodine-deficient and iodine-sufficient lactating women, BMIC ranged from 26 to 185 µg/L and 15 to 1006 µg/L, respectively. In most studies, the categorisation of iodine status assessed by median UIC was consistent with the categorisation of iodine status assessed by median BMIC cut off of ≥100 µg/L, to determine iodine sufficiency in lactating women and children <2 years of age. Conclusions: The systematic review indicated that BMIC is a promising biomarker of iodine status in lactating women and children <2 years of age. However, these data need to be interpreted cautiously, given the study limitations in the included studies. Future studies should consider investigating the optimal median BMIC, as there is a lack of high-quality observational and intervention studies in lactating women and infants.