The Direct Healthcare Cost of Stargardt Disease: A Claims-Based Analysis

ABSTRACT

Purpose: Stargardt disease (SD) is the most common juvenile macular degeneration and a leading cause of uncorrectable childhood blindness. The progressive and incurable nature of this chronic condition entails a long-term financial burden on affected individuals. The economic costs of SD have not been characterized in detail, so we aimed to estimate the direct healthcare cost of SD.

Methods: Outpatient administrative claims data (2010–2014) for patients with SD were analyzed from the IBM® MarketScan® Commercial Claims and Encounters Database. Two comparison groups were selected: nonexudative age-related macular degeneration (AMD) and bilateral sensorineural hearing loss (SHL). Gross median payments per year of insurance coverage were calculated.

Results: A total of 472,428 patients were analyzed (5,015 SD, 369,750 SHL and 97,663 AMD patients respectively). The payment per year of insurance coverage for SD (median: 105.58 USD, IQR: 50.53 USD–218.71 USD) was higher than that of SHL (median: 51.01 USD, IQR: 25.66 USD–121.66 USD, p < .001) and AMD (median: 76.20 USD, IQR: 38.00 USD–164.86 USD, p < .001). When adjusted for age, sex, year of first service, and type of benefit plan, the annual payment for SD was 47.83 USD higher than SHL (p < .001) and 17.34 USD higher than AMD (p < .001).

Conclusions: There is a significant direct healthcare cost associated with SD. The annual per-patient cost of SD was higher than SHL, another condition that causes sensory impairment in people of all ages, and nonexudative AMD which causes a similar pattern of visual loss that typically begins later in life. The total lifetime per-patient cost of SD may exceed that of nonexudative AMD.

KEYWORDS:

• Inherited retinal diseases
• Visual rehabilitation
• Macular degeneration
• Orphan disease
• Low vision

Disclosure statement

None of the authors have any proprietary interests or conflicts of interest related to this submission

Dual publication

Apart from the preprint and conference proceedings listed below, this submission has not been published anywhere previously and is not simultaneously being considered for any other publication

Meeting presentation

Partial analyses were presented as a poster at the Association for Research in Vision and Ophthalmology Annual Meeting 2018 (proceedings: Aziz et al., Healthcare Costs of Stargardt Disease. Investigative Ophthalmology & Visual Science July 2018, Vol.59, 5221)

Preprint

This manuscript was posted as a preprint to the preprint server https://www.medrxiv.org/

(Link: https://doi.org/10.1101/2020.01.22.20017962)

Additional information

Funding

Foundation Fighting Blindness CD-RM-0918-0749-JHU (MSS), Joseph Albert Hekimian Fund (MSS), National Institute on Aging K01AG052640 (BKS), Research to Prevent Blindness (unrestricted grant to the Wilmer Eye Institute)