Indirect costs associated with premature mortality among those with veno-occlusive disease/sinusoidal obstruction syndrome with multiorgan dysfunction post-hematopoietic stem-cell-transplant

Abstract

Purpose

The study objective was to develop an economic model to assess projected costs of lost productivity associated with premature deaths due to veno-occlusive disease (VOD)/ sinusoidal obstruction syndrome (SOS) with multiorgan dysfunction (MOD) among patients in the US who underwent hematopoietic stem-cell transplant (HSCT) in 2013.

Methods

Data sources included the US Census Bureau and Department of Health, epidemiologic research organizations, and medical research literature. The model considered only lost productivity associated with premature death, with lifetime salary assumed to reflect productivity. Average annual salary was assumed to be the same for HSCT survivors and the general population, with a working age range between 18 and 65 years. Key data inputs included number of HSCTs by graft type (allogeneic and autologous) performed in the US in 2013, HSCT-related mortality, mortality associated with VOD/SOS with MOD, and life-expectancy reduction for HSCT survivors vs the general population. Excess mortality equaled total deaths among patients with VOD/SOS and MOD minus deaths in these patients due to causes other than VOD/SOS with MOD.

Results

Among 18,284 patients who underwent HSCT in the US in 2013, the model estimated that 361 excess deaths due to VOD/SOS with MOD occurred (158 following allogeneic and 203 after autologous transplants). These deaths accounted for total lost work productivity of 5,990 years and $124,212,173 in lost wages, averaging 17 years and $343,791 per patient. A sensitivity analysis incorporating adjustment factors for epidemiologic and economic inputs calculated total financial loss of $84 million to $194 million.

Limitation

Estimates of post-HSCT VOD/SOS with MOD incidence and mortality were approximated, due to changing HSCT practices.

Conclusion

Premature death due to VOD/SOS with MOD imposes a substantial economic burden in this population in terms of lost productivity. Additional studies of this economic burden are warranted.

Keywords:

• veno-occlusive disease/sinusoidal obstruction syndrome
• stem-cell transplantation
• mortality
• lost productivity/income
• indirect costs
• economic burden

Acknowledgments

This research was funded by Jazz Pharmaceuticals Inc, the manufacturer of defibrotide. The authors thank Larry Deblinger and The Curry Rockefeller Group, LLC of Tarry-town, NY for providing medical writing support and editorial assistance in formatting, proofreading, and copy editing, and fact-checking, which was funded by Jazz Pharmaceuticals in accordance with Good Publication Practice (GPP3) guidelines (http://www.ismpp.org/gpp3). Jazz Pharmaceuticals also reviewed and edited the publication for scientific accuracy.

Author contributions

All authors were responsible for the study conception and design, involved in the collection and assembly of data, participated in study-data analysis, interpretation, and manuscript writing, and provided their final approval of this manuscript.

Disclosure

WT and ZYZ are consultants for Jazz Pharmaceuticals. KFV is an employee of Jazz Pharmaceuticals and holds stock and/ or stock options in Jazz Pharmaceuticals. The authors report no other conflicts of interest in this work.