Abstract
Blood stem cell gene therapy to treat hemoglobinopathies is beginning to transform health for small numbers of patients in the U.S. and Europe, where these conditions qualify as rare diseases. Yet hemoglobinopathies are common globally, disproportionately affecting low- and middle-income countries (LMICs), creating an ethical imperative to ensure access where disease burden is greatest. Gene therapy could have blockbuster drug potential if distributable to these regions, but cost is a major barrier. Cost-effectiveness analysis (CEA) models are seldom adapted to low-income settings, where limited data and resources constrain efforts to contextualize high-income evidence. Here, we present a novel framework to evaluate high-income country authorized gene therapies in LMIC contexts. Uganda, where sickle cell disease (SCD) imposes a major burden and no curative therapies are available, is the test case. We evaluate cost-effectiveness of gene therapy for adolescents and adults with SCD in Uganda, adapting U.S. evidence to local economic conditions. Using a three-state Markov model to estimate lifetime costs of standard-of-care in Uganda, two U.S.-based CEA models were adapted using scaling factors and applied to two authorized gene therapies for SCD, Lyfgenia™ (lovo-cel) and Casgevy® (exa-cel), assuming biologically consistent efficacy across populations. Incremental cost-effectiveness ratios (ICERs) were calculated from healthcare and societal perspectives, with internationally accepted gross domestic product-based thresholds. This study demonstrates that Casgevy could be cost-effective in Uganda at a scaled cost when societal benefits are considered. This framework enables CEAs for emerging therapies where local clinical trial data are limited, supporting local decision-makers, global funders, and manufacturers in advancing equitable access to transformative therapies in LMICs.
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Data availability
All data used to draw conclusions from this study are included in this manuscript.
Code availability
The analytical model and simulation code used to generate the results presented in this study are available from the corresponding author upon request.
Data availability
All data used in this study are publicly available. Sources include published literature, open-access economic evaluations, and publicly accessible databases as cited throughout the manuscript. No proprietary or patient-level data were used.
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Acknowledgements
We are grateful for the support of and contributions by the Washington Research Foundation (WRF). We are grateful to Thomas Gallagher (Sr. Scientific Writer at UMass Chan Medical School) for assistance in preparing this manuscript. Research reported in this publication was supported by the National Institutes of Health Office of the Director, Office of Research Infrastructure Programs (ORIP) award numbers P51OD010425 and U42OD011123. This research was supported by the Fred Hutchinson Cancer Research Center/University of Washington/Seattle Children’s Cancer Consortium (P30 CA015704). This study was supported in part by NIH grants R01AI167009 and R01AI158728 (MPIs: J. Adair and J. Taylor), grants from the Bill and Melinda Gates Foundation (INV002613 (PI: J. Adair) and INV03398 (PI: Dr. Boro Dropulić)) which supported L. Bayigga, and by the Washington Research Foundation (PI: J. Adair), which supported R. Kyeyune.
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FM-C, RH, AB, and JEA conceived ideas. FM-C, RH, and JEA wrote the paper. JEA and RH supervised this research, secured funding, and edited the paper. FM-C performed research. LB, RKB, and CK-M provided Ugandan context for this study and feedback on the study design. All authors read and approved the manuscript.
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JA and CK-M are co-founders of the Global Gene Therapy Initiative (GGTI). JA is a Fellow of the Ugandan National Academy of Sciences (FUNAS). Work conducted for this study was performed while JA was employed at the Fred Hutchinson Cancer Center. JA is now Professor and Vice Chair of the Department of Genetic and Cellular Medicine at the University of Massachusetts Chan Medical School. FM-C and RH have assigned an innovation related to this study (Scalable Methodology for Adapting High-Income Cost-Effectiveness Evidence to Low-Resource Healthcare Settings) to the University of Washington. At the time of manuscript submission, this innovation is not protected by a patent. None of the remaining authors declare any conflicts of interest.
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This study did not involve human participants, animals, or the collection of identifiable personal data. All analyses were conducted using publicly available, previously published data sources and simulation models. All methods were performed in accordance with the relevant guidelines and regulations. Because no primary data collection involving human participants or live vertebrates was performed, approval from an ethics committee was not required, and informed consent was not applicable. No identifiable images of human research participants are included in this article; therefore, written informed consent for publication of images was not required.
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Montano-Campos, J.F., Adair, J.E., Basu, A. et al. Cost-effectiveness of gene therapy for sickle cell disease in Uganda: tailoring high-income evidence to Uganda’s context. Gene Ther (2026). https://doi.org/10.1038/s41434-026-00598-1
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DOI: https://doi.org/10.1038/s41434-026-00598-1