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Introduction: Although the liver and lung are the most frequently affected organs in cystic echinococcosis, the cysts may develop in any viscera and tissues. Breast is a rare primary localization with few cases described in the literature. We present an updated and systematic review and discuss the possible mechanisms of spreading, diagnostic and treatment options., Materials and Methods: We performed a literature search in PUBMED using the key words ‘hydatid disease’, ‘cystic echinococcosis’ and ‘breast echinococcosis’ without time limitation. Only studies reporting breast cystic echinococcosis were included., Results: Overall, 121 cases with cystic echinococcosis and 2 with alveolar echinococcosis were reported. A total of 52 cases were included in the analysis. The mean size of cysts was 5.5 cm (range 1.7-12). The most common clinical presentation was painless lump presented from 4 months to 19 years before the final diagnosis. Most cases had isolated breast CE, few cases had synchronous localizations – femoral, thigh and lung, and previous liver CE. Most were active CL and CE1-2 cysts (72%). Ultrasound was used in 83%, followed by mammography (35%). Fine needle aspiration was reported in 27 cases with positive finding in 59%., Conclusions: In cases with cystic breast lesions from endemic regions we recommend the US as a gold standard. CT and MRT are more accurate but expensive tools without the potential to change the surgical tactic. In contrast to the other localizations of CE, complete excision of the cysts is the best diagnostic and treatment approach., (This is an open access article distributed under the terms of the Creative Commons Attribution License (CC BY 4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.)