Cancer predisposition syndromes as a risk factor for early second primary neoplasms after childhood cancer - A national cohort study.

Waespe, Nicolas; Belle, Fabiën N.; Redmond, Shelagh; Schindera, Christina; Spycher, Ben D.; Rössler, Jochen; Ansari, Marc; Kuehni, Claudia E. (2021). Cancer predisposition syndromes as a risk factor for early second primary neoplasms after childhood cancer - A national cohort study. European journal of cancer, 145, pp. 71-80. Elsevier 10.1016/j.ejca.2020.11.042

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BACKGROUND

Childhood cancer patients are at increased risk of second primary neoplasms (SPNs). We assessed incidence and risk factors for early SPNs with a focus on cancer predisposition syndromes (CPSs).

PATIENTS AND METHODS

This cohort study used data from the Swiss Childhood Cancer Registry. We included patients with first primary neoplasms (FPNs) diagnosed before age 21 years from 1986 to 2015 and identified SPNs occurring before age 21. We calculated standardised incidence ratios (SIRs) and absolute excess risks (AERs) using Swiss population cancer incidence data, and cumulative incidence of SPNs. We calculated hazard ratios (HRs) of risk factors for SPNs using Fine and Gray competing risk regression.

RESULTS

Among 8074 childhood cancer patients, 304 (4%) were diagnosed with a CPS and 94 (1%) developed early SPNs. The incidence of SPNs was more than 10-fold higher in childhood cancer patients than the incidence of neoplasms in the general population (SIR = 10.6, 95% confidence interval [CI]: 8.7-13.1) and the AER was 179/100,000 person-years (CI: 139-219). Cumulative incidence of SPNs 20 years after FPN diagnosis was 23% in patients with CPSs (CI: 12-41%) and 2.7% in those without (CI: 2.0-3.6%). Risk factors for SPNs were CPSs (HR = 7.8, CI: 4.8-12.7), chemotherapy (HR = 2.2, CI: 1.1-4.6), radiotherapy (HR = 1.9, CI = 1.2-2.9), haematopoietic stem cell transplantation (HR = 1.8, CI: 1-3.3), and older age (15-20 years) at FPN diagnosis (HR = 1.9, CI: 1.1-3.2).

CONCLUSION

CPSs are associated with a high risk of SPNs before age 21 years. Identification of CPSs is important for appropriate cancer surveillance and targeted screening.

Item Type:

Journal Article (Original Article)

Division/Institute:

04 Faculty of Medicine > Department of Gynaecology, Paediatrics and Endocrinology (DFKE) > Clinic of Paediatric Medicine
04 Faculty of Medicine > Department of Gynaecology, Paediatrics and Endocrinology (DFKE) > Clinic of Paediatric Medicine > Paediatric Haematology/Oncology
04 Faculty of Medicine > Pre-clinic Human Medicine > Institute of Social and Preventive Medicine (ISPM)
04 Faculty of Medicine > Pre-clinic Human Medicine > BioMedical Research (DBMR) > DBMR Forschung Mu35 > Forschungsgruppe Hämatologie / Onkologie (Pädiatrie)
04 Faculty of Medicine > Pre-clinic Human Medicine > BioMedical Research (DBMR) > DBMR Forschung Mu35 > Forschungsgruppe Hämatologie / Onkologie (Pädiatrie)

UniBE Contributor:

Wäspe Laredo, Nicolas Thomas; Belle, Fabien Naomi; Redmond, Shelagh; Schindera, Christina; Spycher, Ben; Rössler, Jochen Karl and Kühni, Claudia

Subjects:

600 Technology > 610 Medicine & health
300 Social sciences, sociology & anthropology > 360 Social problems & social services

ISSN:

0959-8049

Publisher:

Elsevier

Funders:

[4] Swiss National Science Foundation ; [189] Swiss Cancer Research = Krebsforschung Schweiz ; [193] Swiss Cancer League = Krebsliga Schweiz

Language:

English

Submitter:

Doris Kopp Heim

Date Deposited:

14 Jan 2021 15:39

Last Modified:

13 Mar 2021 20:20

Publisher DOI:

10.1016/j.ejca.2020.11.042

PubMed ID:

33423008

Uncontrolled Keywords:

Adolescent medicine Cancer survivors Early detection of cancer Genetic counselling Genetic testing Hereditary neoplastic syndromes Neoplasms Pediatrics Second primary neoplasms Swiss childhood cancer registry

BORIS DOI:

10.48350/151289

URI:

https://boris.unibe.ch/id/eprint/151289

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