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Cost-effectiveness of IVF with PGT-M/A to prevent transmission of spinal muscular atrophy in offspring of carrier couples

  • Genetics
  • Published:
Journal of Assisted Reproduction and Genetics Aims and scope Submit manuscript

Abstract

Purpose

To evaluate the cost-effectiveness of in-vitro fertilization with preimplantation genetic testing for aneuploidy and monogenic disorders (IVF with PGT-M/A) to prevent transmission of spinal muscular atrophy to offspring of carrier couples.

Methods

A decision-analytic model was created to compare the cost-effectiveness of IVF with PGT-M/A to unassisted conception with prenatal diagnostic testing and termination (if applicable). IVF with PGT-M/A costs were determined using a separate Markov state-transition model. IVF outcomes data was derived from 76 carriers of monogenic disorders who underwent IVF with PGT-M/A at a single academic REI center. Other probabilities, costs, and utilities were derived from the literature. Costs were modeled from healthcare perspective. Utilities were modeled from the parental perspective as quality-adjusted life-years (QALYs).

Results

The incremental cost-effectiveness ratio for IVF with PGT-M/A compared to unassisted conception is $22,050 per quality-adjusted life-year. The average cost of IVF with PGT-M/A is $41,002 (SD: $8,355). At willingness-to-pay thresholds of $50,000 and $100,000, IVF with PGT-M/A is cost-effective 93.3% and 99.5% of the time, respectively.

Conclusions

Compared to unassisted conception, IVF with PGT-M/A is cost-effective for preventing the transmission of spinal muscular atrophy to the offspring of carrier couples. These findings support insurance coverage of IVF with PGT-M/A for carriers of spinal muscular atrophy.

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Data Availability

The IVF outcome data used in this manuscript can be made available for review, without identifiers, upon request.

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Acknowledgements

We would like to acknowledge and thank Dr. Jeremy Goldhaber-Fiebert, PhD for his contributions to this work.

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Authors and Affiliations

  1. Department of Obstetrics & Gynecology, Stanford University School of Medicine, 453 Quarry Road MC #5317, Palo Alto, CA, 94304, USA

    Arian Khorshid

  2. Stanford Fertility and Reproductive Medicine Center, Sunnyvale, CA, USA

    Alleigh L. H. Boyd, Barry Behr, Qianying Zhao, Ruben Alvero & Brindha Bavan

Authors
  1. Arian Khorshid
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  2. Alleigh L. H. Boyd
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  3. Barry Behr
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  4. Qianying Zhao
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  5. Ruben Alvero
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  6. Brindha Bavan
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Contributions

All authors contributed to the study conception and design. Material preparation, data collection were performed by Arian Khorshid, Alleigh Boyd, and Qianying Zhao. Model design and analysis was performed by Arian Khorshid, Ruben Alvero, and Brindha Bavan. The first draft of the manuscript was written by Arian Khorshid, and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript. The principal investigator overseeing this work was Brindha Bavan.

Corresponding author

Correspondence to Arian Khorshid.

Ethics declarations

Brindha Bavan is a clinical research consultant for Stanford’s Atropos Health. All other authors have no relevant financial or non-financial interests to disclose.

This work was supported by the Stanford University Department of OBGYN

Data regarding any of the subjects in the study has not been previously published unless specified.

Data will be made available to the editors of the journal for review or query upon request.

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Supplementary Information

Below is the link to the electronic supplementary material.

10815_2023_2738_MOESM1_ESM.pdf

Supplementary Figure 1: Tornado diagram of one-way sensitivity analysis for variables used in IVF with PGT-M/A Cost Model. Overall cost is on the x-axis. The base-case and 95% confidence intervals are listed next to each variable. Variables without impact on overall cost were omitted from this diagram. (PDF 53 KB)

10815_2023_2738_MOESM2_ESM.pdf

Supplementary Figure 2: Tornado diagram of one-way sensitivity analysis for variables used in SMA Model. Incremental Cost-Effectiveness Ratio (ICER) is on the x-axis. The base-case and 95% confidence intervals are listed next to each variable. Variables without impact on overall cost were omitted from this diagram. (PDF 61 KB)

Supplementary file3 (DOCX 17 KB)

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Khorshid, A., Boyd, A.L.H., Behr, B. et al. Cost-effectiveness of IVF with PGT-M/A to prevent transmission of spinal muscular atrophy in offspring of carrier couples. J Assist Reprod Genet 40, 793–801 (2023). https://doi.org/10.1007/s10815-023-02738-7

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