Economic Burden in Patients With Systemic Scleroderma Associated Interstitial Lung Disease: A Systematic Review

OBJECTIVES: Systemic scleroderma (SSc) is a multisystem autoimmune disease characterised by skin and internal organ fibrosis, often leading to interstitial lung disease (ILD) in ~50% patients contributing to significant morbidity and disability. We did a systematic review to assess the economic burden associated with SSc-ILD.

METHODS: A systematic search was conducted in Embase^® and MEDLINE to identify relevant studies published in last 10 years. Studies were included if they provided information regarding total, direct, indirect costs and healthcare resource use. The review was conducted following the PRISMA and Cochrane guidelines. The costs were adjusted to 2023 values using cumulative inflation rates in the respective geographics.

RESULTS: Overall, 12 studies evaluating 24,977 patients (female >75% and mean age range 46-71 years) with SSc-ILD were included. Four studies conducted in Europe (Denmark [1], France [1], multi-country [2]) estimated an annual mean direct cost ranging from €23,464-€116,668 and indirect cost ranging from €9,924-€37,251. Six studies conducted in North America (Canada [1], USA [5]) reported an annual mean direct cost ranging from $45,488-$109,819 and indirect cost ranging from $8,203-$14,349. In one study conducted in the UK the annual median total cost reported was £7,751 [IQR: 4,532-19,753] including a direct cost of £5,854 [IQR: 1,752-4,102]. A study conducted in Australia reported the annual median direct cost of A$6,475 [IQR: 7,701-52,647]. Hospitalisation was the main driver for the direct cost and productivity loss for the indirect cost. Hospitalisation was the most frequently reported healthcare resource used by 80% of the patients followed by high resolution computed tomography by 58% patients across the studies.

CONCLUSIONS:

SSc-ILD represents a significant economic burden on patients and healthcare systems globally and thus requiring effective management strategies. Future innovations should prioritise developing cost-effective therapies for the management of SSc-ILD.