OBJECTIVES: A systematic literature review was undertaken to identify relevant epidemiology, economic burden, and economic evaluation data in cutaneous T-cell lymphomas (CTCL), particularly mycosis fungoides (MF) and Sézary syndrome (SS) subtypes.

METHODS: A search for studies reporting economic data for CTCL (including MF and SS; limited to English language) was conducted via OVID in five biomedical and economic databases: Embase, MEDLINE, EBM reviews, EconLit, and PsycINFO; key conference proceedings and regulatory websites were also searched. Abstracts and full-text publications were examined to identify studies reporting prevalence, incidence, and mortality data; classification and progression of any stage CTCL; economic evaluations; economic burden/costs associated with CTCL; and utility values for patients with CTCL, MF, and SS.

RESULTS: Of 1406 citations (from 1998-2017) retrieved, 26 studies were included in the qualitative analysis; 3 additional studies were identified from regulatory agencies and conferences. Of the 29 publications, 3 reported on cost-effectiveness models (CEM), 3 on budget impact models (BIM), 4 on cost of illness (COI), and 19 on epidemiology. In general, studies were heterogeneous, making comparisons difficult. In BIM studies, treatment costs associated with targeted cancer therapies were $460,000–$530,000/1 million covered lives and $0.46–$0.53 per patient per member. Across the 3 CEM studies, treatments included extracorporeal photopheresis (ECP; n=3 studies); interferon-α (IFN), bexarotene, denileukin diftitox, romidepsin, and vorinostat (n=2 each); and methotrexate (MTX) and pralatrexate (n=1 each). ECP, IFN, and MTX were deemed cost-effective in all studies in which they were evaluated. Epidemiology studies reported a very broad range of CTCL prevalence; rates for SS ranged from 1.9% to 9%. CTCL annual incidence rates ranged from 0.17-0.57/100,000.

CONCLUSIONS: Epidemiology and economic data for CTCL are limited and heterogeneous. Cost-effective treatments included ECP, IFN, and MTX. Costs and burden of illness were greater for patients with more severe disease. SPONSORSHIP: Kyowa Kirin