Over the past two decades, many improvements have been made in the management of patients with leukemia. Research in this field most often focuses on the youngest and oldest patient age groups. However, the population of patients in between those age groups has received relatively little attention with few studies specifically focusing on them. This important ‘age gap' has demonstrated a unique, difficult-to-treat group of patients known as adolescents and young adults, or AYAs. Variably defined in the literature as patients from late teenage years to the age of up to 40 years, the AYA group of patients represents a vulnerable subset of patients now identified to require its own focus, development of therapeutic strategies and parallel emphasis on special support systems involving multidisciplinary psychosocial care. Despite the great advancements that have been realized for patients with chronic myeloid leukemia (CML), the AYA group has seldom been the focus of specific reports and studies, and the outcome appears to lag behind the general population. This review focuses on this subset of AYA patients with CML and summarizes the available data and recent developments, challenges and treatment options for this group of patients.