To consolidate the current understanding of dysregulation of the JAK/STAT pathway and therapeutic implications in CTCL, we conducted a literature review of the PubMed, Web of Science, and ClinicalTrials.gov databases with the following keywords: cutaneous T-cell lymphoma, mycosis fungoides, Sézary syndrome, pathogenesis, JAK/STAT pathway, therapeutic strategies, JAK/STAT inhibitors, clinical response, and adverse effects. The gene discussed is SOAT1; the disease is Sezary syndrome.