Economic and Health-Related Quality of Life Burden in Endogenous Cushing’s Syndrome: A Systematic Literature Review

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OBJECTIVES: Endogenous Cushing’s syndrome (CS) is a rare endocrine disorder characterized by chronic cortisol overproduction. Our objective was to summarize the economic and health-related (HR) quality of life (QoL) burden of CS and evaluate post-treatment burden.

METHODS: A systematic literature review (SLR) using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) checklist was conducted to identify peer-reviewed manuscripts and conference abstracts published in English from 2015 to December 4th, 2020, in PubMed, MEDLINE, and Embase.

RESULTS: Forty-five publications were included. Data were extracted from 37 primary studies; eight SLRs were included for reference only. Five studies (13%) reported on economic burden, 31 (84%) reported HRQoL using patient-reported outcome measures (PROMs), and one (3%) reported both. Overall annual direct healthcare costs for patients with CS ranged from $26,269 (2012) to $34,992 (2010). Additionally, one study reported that the total per patient per month cost for patients with Cushing’s disease was $3,232. Nine studies (24%) reported pre- and post-treatment burden using general or symptom-specific PROMs, with eight reporting pre- and post-surgery burden. Four of the eight studies had longitudinal follow-up for ≥1-year post-operatively. One study reported that 27% of patients experienced recurrence post-surgery with the majority recurring within 10 months. Another study found no statistically significant differences in HRQoL, measured using CushingQOL, between patients treated with 1-4 pituitary surgeries. This demonstrates that while surgery was a primary intervention, sustained remission or cure was not guaranteed among patients. Additionally, three studies reported statistically significant decreases in HRQoL in certain domains, including depression, anxiety, and fear following surgery.

CONCLUSIONS: Few studies address the economic burden of CS; however, the available data indicate patients experience a high direct economic burden. Patient wellbeing is impacted by a high HRQoL burden, which persists in key domains despite current treatment interventions including surgery.


Page Wilson G1, Okeyo J2, Ortiz N3, Oak B4, Zullo K5, Silber A6, Moloney S3, Geer EB7

1Columbia University Irving Medical Center, New York, NY, USA, 2Xeris Pharmaceuticals, Inc., Trevose, PA, USA, 3Strongbridge Biopharma plc, a wholly owned subsidiary of Xeris Biopharma Holdings, Inc., Trevose, PA, USA, 4Trinity Life Sciences, Waltham, MA, USA, 5Trinity Life Sciences, Princeton, NJ, USA, 6Trinity Partners, LLC, Waltham, MA, USA, 7Memorial Sloan Kettering Cancer Center, New York, NY, USA


ISPOR 2022

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