Prevalence and Prognosis of Lynch Syndrome and Sporadic Mismatch Repair Deficiency in Endometrial Cancer.


Journal

Journal of the National Cancer Institute
ISSN: 1460-2105
Titre abrégé: J Natl Cancer Inst
Pays: United States
ID NLM: 7503089

Informations de publication

Date de publication:
04 09 2021
Historique:
received: 30 11 2020
revised: 28 01 2021
accepted: 01 03 2021
pubmed: 12 3 2021
medline: 25 2 2022
entrez: 11 3 2021
Statut: ppublish

Résumé

Standard screening of endometrial cancer (EC) for Lynch syndrome (LS) is gaining traction; however, the prognostic impact of an underlying hereditary etiology is unknown. We established the prevalence, prognosis, and subsequent primary cancer incidence of patients with LS-associated EC in relation to sporadic mismatch repair deficient (MMRd)-EC in the large combined Post Operative Radiation Therapy in Endometrial Carcinoma-1, -2, and -3 trial cohort. After MMR-immunohistochemistry, MLH1-promoter methylation testing, and next-generation sequencing, tumors were classified into 3 groups according to the molecular cause of their MMRd-EC. Kaplan-Meier method, log-rank test, and Cox model were used for survival analysis. Competing risk analysis was used to estimate the subsequent cancer probability. All statistical tests were 2-sided. Among the 1336 ECs, 410 (30.7%) were MMRd. A total of 380 (92.7%) were fully triaged: 275 (72.4%) were MLH1-hypermethylated MMRd-ECs; 36 (9.5%) LS MMRd-ECs, and 69 (18.2%) MMRd-ECs due to other causes. Limiting screening of EC patients to 60 years or younger or to 70 years or younger would have resulted in missing 18 (50.0%) and 6 (16.7%) LS diagnoses, respectively. Five-year recurrence-free survival was 91.7% (95% confidence interval [CI] = 83.1% to 100%; hazard ratio = 0.45, 95% CI = 0.16 to 1.24, P = .12) for LS, 95.5% (95% CI = 90.7% to 100%; hazard ratio = 0.17, 95% CI = 0.05 to 0.55, P = .003) for "other" vs 78.6% (95% CI = 73.8% to 83.7%) for MLH1-hypermethylated MMRd-EC. The probability of subsequent LS-associated cancer at 10 years was 11.6% (95% CI = 0.0% to 24.7%), 1.5% (95% CI = 0.0% to 4.3%), and 7.0% (95% CI = 3.0% to 10.9%) within the LS, "other," and MLH1-hypermethylated MMRd-EC groups, respectively. The LS prevalence in the Post Operative Radiation Therapy in Endometrial Carcinoma trial population was 2.8% and among MMRd-ECs was 9.5%. Patients with LS-associated ECs showed a trend towards better recurrence-free survival and higher risk for second cancers compared with patients with MLH1-hypermethylated MMRd-EC.

Sections du résumé

BACKGROUND
Standard screening of endometrial cancer (EC) for Lynch syndrome (LS) is gaining traction; however, the prognostic impact of an underlying hereditary etiology is unknown. We established the prevalence, prognosis, and subsequent primary cancer incidence of patients with LS-associated EC in relation to sporadic mismatch repair deficient (MMRd)-EC in the large combined Post Operative Radiation Therapy in Endometrial Carcinoma-1, -2, and -3 trial cohort.
METHODS
After MMR-immunohistochemistry, MLH1-promoter methylation testing, and next-generation sequencing, tumors were classified into 3 groups according to the molecular cause of their MMRd-EC. Kaplan-Meier method, log-rank test, and Cox model were used for survival analysis. Competing risk analysis was used to estimate the subsequent cancer probability. All statistical tests were 2-sided.
RESULTS
Among the 1336 ECs, 410 (30.7%) were MMRd. A total of 380 (92.7%) were fully triaged: 275 (72.4%) were MLH1-hypermethylated MMRd-ECs; 36 (9.5%) LS MMRd-ECs, and 69 (18.2%) MMRd-ECs due to other causes. Limiting screening of EC patients to 60 years or younger or to 70 years or younger would have resulted in missing 18 (50.0%) and 6 (16.7%) LS diagnoses, respectively. Five-year recurrence-free survival was 91.7% (95% confidence interval [CI] = 83.1% to 100%; hazard ratio = 0.45, 95% CI = 0.16 to 1.24, P = .12) for LS, 95.5% (95% CI = 90.7% to 100%; hazard ratio = 0.17, 95% CI = 0.05 to 0.55, P = .003) for "other" vs 78.6% (95% CI = 73.8% to 83.7%) for MLH1-hypermethylated MMRd-EC. The probability of subsequent LS-associated cancer at 10 years was 11.6% (95% CI = 0.0% to 24.7%), 1.5% (95% CI = 0.0% to 4.3%), and 7.0% (95% CI = 3.0% to 10.9%) within the LS, "other," and MLH1-hypermethylated MMRd-EC groups, respectively.
CONCLUSIONS
The LS prevalence in the Post Operative Radiation Therapy in Endometrial Carcinoma trial population was 2.8% and among MMRd-ECs was 9.5%. Patients with LS-associated ECs showed a trend towards better recurrence-free survival and higher risk for second cancers compared with patients with MLH1-hypermethylated MMRd-EC.

Identifiants

pubmed: 33693762
pii: 6161779
doi: 10.1093/jnci/djab029
pmc: PMC8418420
doi:

Substances chimiques

MutL Protein Homolog 1 EC 3.6.1.3

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

1212-1220

Informations de copyright

© The Author(s) 2021. Published by Oxford University Press.

Auteurs

Cathalijne C B Post (CCB)

Department of Radiation Oncology, Leiden University Medical Center, Leiden, the Netherlands.

Ellen Stelloo (E)

Department of Pathology, Leiden University Medical Center, Leiden, the Netherlands.

Vincent T H B M Smit (VTHBM)

Department of Pathology, Leiden University Medical Center, Leiden, the Netherlands.

Dina Ruano (D)

Department of Pathology, Leiden University Medical Center, Leiden, the Netherlands.

Carli M Tops (CM)

Department of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.

Lisa Vermij (L)

Department of Pathology, Leiden University Medical Center, Leiden, the Netherlands.

Tessa A Rutten (TA)

Department of Pathology, Leiden University Medical Center, Leiden, the Netherlands.

Ina M Jürgenliemk-Schulz (IM)

Department of Radiation Oncology, University Medical Center Utrecht, Utrecht, the Netherlands.

Ludy C H W Lutgens (LCHW)

Department of Radiation Oncology, MAASTRO Clinic, Maastricht, the Netherlands.

Jan J Jobsen (JJ)

Department of Radiation Oncology, Medical Spectrum Twente, Enschede, the Netherlands.

Remi A Nout (RA)

Department of Radiation Oncology, Leiden University Medical Center, Leiden, the Netherlands.

Emma J Crosbie (EJ)

Division of Cancer Sciences, University of Manchester, St Mary's Hospital, Manchester, UK.
Department of Obstetrics and Gynaecology, Manchester University NHS Foundation Trust, Manchester, UK.

Melanie E Powell (ME)

Department of Clinical Oncology, Barts Health NHS Trust, London, UK.

Linda Mileshkin (L)

Department of Medical Oncology, Peter MacCallum Cancer Centre, Melbourne, Australia.

Alexandra Leary (A)

Department of Medical Oncology, Gustave Roussy Cancer Center-INSERM U981, Université Paris Saclay, Villejuif, France.

Paul Bessette (P)

Department of Obstetrics and Gynecology, University of Sherbrooke, Sherbrooke, Quebec, Canada.

Hein Putter (H)

Department of Biostatistics, Leiden University Medical Center, Leiden, the Netherlands.

Stephanie M de Boer (SM)

Department of Radiation Oncology, Leiden University Medical Center, Leiden, the Netherlands.

Nanda Horeweg (N)

Department of Radiation Oncology, Leiden University Medical Center, Leiden, the Netherlands.

Maartje Nielsen (M)

Department of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.

Tom van Wezel (TV)

Department of Pathology, Leiden University Medical Center, Leiden, the Netherlands.

Tjalling Bosse (T)

Department of Pathology, Leiden University Medical Center, Leiden, the Netherlands.

Carien L Creutzberg (CL)

Department of Radiation Oncology, Leiden University Medical Center, Leiden, the Netherlands.

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