Cerebral microbleeds (CMBs) are one kind of neuroimaging manifestations in cerebral small vessel disease, pathologically correlated with hemosiderin deposition. Susceptibility-weighted imaging can be used to assess CMBs. CMBs are increasingly recognized in the fields of cerebrovascular diseases and neurodegenerative diseases. However, some of rare diseases can also manifest CMBs. It is therefore necessary to reliably evaluate their presence and distribution in the brain. Understanding the uncommon causes of CMBs is not only helpful in diagnosis and prognosis of these rare diseases, but can also help in better understanding different pathophysiology involved in the development of CMBs. In this article, we described uncommon causes of CMBs.