In humans, the cytoplasmic FMR1 interacting protein (CYFIP) family is composed of CYFIP1 and CYFIP2. Despite their high similarity and shared interaction with many partners, CYFIP1 and CYFIP2 act at diferent points in cellular processes. CYFIP1 and CYFIP2 have diferent expression levels in human tissues, and knockout animals die at diferent time points of development. CYFIP1, similar to CYFIP2, acts in the WAVE regulatory complex (WRC) and plays a role in actin dynamics through the activation of the Arp2/3 complex and in a posttranscriptional regulatory complex with the fragile X mental retardation protein (FMRP). Previous reports have shown that CYFIP1 and CYFIP2 may play roles in posttranscriptional regulation in diferent ways. While CYFIP1 is involved in translation initiation via the 5′UTR, CYFIP2 may regulate mRNA expression via the 3′UTR. In addition, this CYFIP protein family is involved in neural development and maturation as well as in diferent neural disorders, such as intellectual disabilities, autistic spectrum disorders, and Alzheimer’s disease. In this review, we map diverse studies regarding the functions, regulation, and implications of CYFIP proteins in a series of molecular pathways. We also highlight mutations and their structural efects both in functional studies and in neural diseases.