In recent years, studies have associated dupilumab with unmasking or progression of cutaneous T‐cell lymphoma (CTCL). The objective of this study was to synthesize reported cases of CTCL in the setting of dupilumab use. Two independent reviewers conducted a systematic review using PubMed and Embase databases. Twelve studies, of 27 total patients, were included. Dupilumab‐associated CTCL was diagnosed on average 7.8 months following dupilumab initiation. Of reports that included body surface area (BSA), 100% had a BSA >50% prior to diagnosis, and the majority of patients were diagnosed at later stages of disease: stage III (4/18) or IV (6/18). Post‐dupilumab biopsies revealed a greater density of cells and a predominant lichenoid pattern, compared to various histologic patterns and less cell density in pre‐dupilumab biopsies. The majority of TCR gene rearrangement studies were equivocal (20%) or negative (60%), posing a potential diagnostic pitfall. Limitations included lack of standardized data collection and reporting and a small number of studies. We suggest that earlier biopsies, three to four months after initiation of dupilumab, may be helpful for earlier recognition of CTCL in patients who are not improving, progressing, or have morphologic changes on dupilumab.