Cost-effectiveness Analysis of Genotype-Specific Surveillance and Preventive Strategies for Gynecologic Cancers Among Women With Lynch Syndrome

Wright, Jason D.; Silver, Elisabeth R.; Tan, Sarah; Hur, Chin; Kastrinos, Fay

doi:10.1001/jamanetworkopen.2021.23616

Cited by 8 publications

(17 citation statements)

References 60 publications

(143 reference statements)

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“…Wright et al [ 52 ] evaluated the gene-specific cost-effectiveness of RRS in women with LS. They found that hysterectomy and ES at the age of 40 with DO at the age of 50 was an optimal strategy for MLH1 and MSH6 PV carriers, with ICURs of $33,269/QALY and $20,008/QALY, respectively.…”

Section: Resultsmentioning

confidence: 99%

“…For MSH2 PV carriers, the optimal strategy would be hysterectomy and ES at the age of 40 with DO at the age of 50, with utility scores for early menopause below 0.86, hysterectomy and BSO at the age of 40, with a utility score of 0.87–0.94, or hysterectomy and BSO at the age of 35, with a utility score greater than 0.95. If disutility of hysterectomy was taken into consideration, the optimal strategy would not change for MSH2 or MSH6 PV carriers, while hysterectomy and BSO at the age of 40 was favourable for MLH1 PV carriers; no intervention was optimal for PMS2 PV carriers, with a post-hysterectomy utility score less than 0.97 [ 52 ]. In PSA, the corresponding optimal strategy in the base case had a probability of 84.2% to be cost-effective for MLH1 , 71.0% for MSH6 , 86.2% for MSH2 and 91.6% for PMS2 .…”

Section: Resultsmentioning

confidence: 99%

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Cost-Effectiveness of Risk-Reducing Surgery for Breast and Ovarian Cancer Prevention: A Systematic Review

Xia

Oxley

Sideris

et al. 2022

Cancers

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Policymakers require robust cost-effectiveness evidence of risk-reducing-surgery (RRS) for decision making on resource allocation for breast cancer (BC)/ovarian cancer (OC)/endometrial cancer (EC) prevention. We aimed to summarise published data on the cost-effectiveness of risk-reducing mastectomy (RRM)/risk-reducing salpingo-oophorectomy (RRSO)/risk-reducing early salpingectomy and delayed oophorectomy (RRESDO) for BC/OC prevention in intermediate/high-risk populations; hysterectomy and bilateral salpingo-oophorectomy (BSO) in Lynch syndrome women; and opportunistic bilateral salpingectomy (OBS) for OC prevention in baseline-risk populations. Major databases were searched until December 2021 following a prospective protocol (PROSPERO-CRD42022338008). Data were qualitatively synthesised following a PICO framework. Twenty two studies were included, with a reporting quality varying from 53.6% to 82.1% of the items scored in the CHEERS checklist. The incremental cost-effectiveness ratio/incremental cost-utility ratio and cost thresholds were inflated and converted to US$2020, using the original currency consumer price index (CPI) and purchasing power parities (PPP), for comparison. Eight studies concluded that RRM and/or RRSO were cost-effective compared to surveillance/no surgery for BRCA1/2, while RRESDO was cost-effective compared to RRSO in one study. Three studies found that hysterectomy with BSO was cost-effective compared to surveillance in Lynch syndrome women. Two studies showed that RRSO was also cost-effective at ≥4%/≥5% lifetime OC risk for pre-/post-menopausal women, respectively. Seven studies demonstrated the cost-effectiveness of OBS at hysterectomy (n = 4), laparoscopic sterilisation (n = 4) or caesarean section (n = 2). This systematic review confirms that RRS is cost-effective, while the results are context-specific, given the diversity in the target populations, health systems and model assumptions, and sensitive to the disutility, age and uptake rates associated with RRS. Additionally, RRESDO/OBS were sensitive to the uncertainty concerning the effect sizes in terms of the OC-risk reduction and long-term health impact. Our findings are relevant for policymakers/service providers and the design of future research studies.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Cost-Effectiveness of Risk-Reducing Surgery for Breast and Ovarian Cancer Prevention: A Systematic Review

Xia

Oxley

Sideris

et al. 2022

Cancers

View full text Add to dashboard Cite

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“…Women with PMS2 pathogenic variants do not require BSO. While cost-effectiveness analysis of alternative strategies such as two-step surgery has been undertaken in Lynch Syndrome, there are no prospective trials evaluating this strategy and thus no available QOL outcomes [ 13 ]. Furthermore, women with PTEN pathogenic variants, a family history of EC, type 2 diabetes and/or obesity may benefit from RRH without BSO for increased EC (without OC) risk.…”

Section: Discussionmentioning

confidence: 99%

“…For women at substantially increased EC risk, such as those with Lynch syndrome, EC prevention is highly cost-effective for health systems [ 12 , 13 ] and recommended by international guidelines [ 14 ]. The most effective prevention is surgical removal of the uterus in the form of risk-reducing hysterectomy (RRH), which prevents 100% of EC cases [ 15 ].…”

Section: Introductionmentioning

confidence: 99%

“…This further allows for the calculation of quality adjusted life years (QALYs), and health-economic modelling of preventive strategies [ 28 ]. Previous health economic modelling studies on EC prevention have lacked high-quality data on utility scores of RRH, using estimates rather than values derived directly from patients who have undergone RRH, and results were sensitive to these (estimated) utility scores [ 13 , 29 ]. Therefore, determining the QoL after RRH remains a priority.…”

Section: Introductionmentioning

confidence: 99%

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Quality of Life after Risk-Reducing Hysterectomy for Endometrial Cancer Prevention: A Systematic Review

Oxley

Xiong

Xia

et al. 2022

Cancers

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Background: Risk-reducing hysterectomy (RRH) is the gold-standard prevention for endometrial cancer (EC). Knowledge of the impact on quality-of-life (QoL) is crucial for decision-making. This systematic review aims to summarise the evidence. Methods: We searched major databases until July 2022 (CRD42022347631). Given the paucity of data on RRH, we also included hysterectomy as treatment for benign disease. We used validated quality-assessment tools, and performed qualitative synthesis of QoL outcomes. Results: Four studies (64 patients) reported on RRH, 25 studies (1268 patients) on hysterectomy as treatment for uterine bleeding. There was moderate risk-of-bias in many studies. Following RRH, three qualitative studies found substantially lowered cancer-worry, with no decision-regret. Oophorectomy (for ovarian cancer prevention) severely impaired menopause-specific QoL and sexual-function, particularly without hormone-replacement. Quantitative studies supported these results, finding low distress and generally high satisfaction. Hysterectomy as treatment of bleeding improved QoL, resulted in high satisfaction, and no change or improvements in sexual and urinary function, although small numbers reported worsening. Conclusions: There is very limited evidence on QoL after RRH. Whilst there are benefits, most adverse consequences arise from oophorectomy. Benign hysterectomy allows for some limited comparison; however, more research is needed for outcomes in the population of women at increased EC-risk.

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Exploring Gene-Specific Guidelines for Risk Management of Gynecological Cancer in Lynch Syndrome

Petelin

Trainer

2021

JAMA Netw Open

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Lynch syndrome is a unifying term for a hereditary cancer syndrome associated with abrogation of the DNA mismatch repair (MMR) pathway. Heterozygous pathogenic variants (PVs) in the MMR genes MLH1, MSH2, MSH6, and PMS2 are associated with an increased risk of a spectrum of cancers, with colorectal cancer (CRC), endometrial cancer (EC), and ovarian cancer (OC) being the most common. Providing precise gene-specific cancer risks while minimizing ascertainment and selection bias is challenging but vital for guiding appropriate decision-making for patient care. 1 Improving the ratio of clinical benefit to potential harms is important given that many individuals identified with PVs do not have cancer or are at increased risk of a second metachronous cancer owing to improved survival stemming from advancements in early detection and cancer treatment. 2 Although colonoscopy screening guidelines are increasingly targeted to individuals' gene-specific CRC risk, current clinical guidelines for management of MMR-associated gynecological cancers are more conservative and based generally on the more highly penetrant gene risks associated with MSH2 and MLH1 (EC risk, 35%-47% to age 70 years; OC risk, 11%-17% to age 70 years). While screening for EC and OC is offered in some settings, there is insufficient evidence of clinical effectiveness, and

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Cost-effectiveness Analysis of Genotype-Specific Surveillance and Preventive Strategies for Gynecologic Cancers Among Women With Lynch Syndrome

Abstract: This economic evaluation analyzes the cost-effectiveness of screening and prevention strategies by genotype among women with Lynch syndrome.

Cited by 8 publications

References 60 publications

Cost-Effectiveness of Risk-Reducing Surgery for Breast and Ovarian Cancer Prevention: A Systematic Review

Cost-Effectiveness of Risk-Reducing Surgery for Breast and Ovarian Cancer Prevention: A Systematic Review

Quality of Life after Risk-Reducing Hysterectomy for Endometrial Cancer Prevention: A Systematic Review

Exploring Gene-Specific Guidelines for Risk Management of Gynecological Cancer in Lynch Syndrome

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