Second and subsequent tumours among 1927 retinoblastoma patients diagnosed in Britain 1951-2004

A MacCarthy; A M Bayne; P A Brownbill; K J Bunch; N L Diggens; G J Draper; M M Hawkins; H C Jenkinson; J E Kingston; C A Stiller; T J Vincent; M F G Murphy

doi:10.1038/bjc.2013.228

Second and subsequent tumours among 1927 retinoblastoma patients diagnosed in Britain 1951-2004

A MacCarthy, A M Bayne, P A Brownbill, K J Bunch, N L Diggens, G J Draper, M M Hawkins, H C Jenkinson, J E Kingston, C A Stiller, T J Vincent, M F G Murphy

Applied Health Research

Research output: Contribution to journal › Article › peer-review

70 Citations (Scopus)

Abstract

BACKGROUND: Retinoblastoma is an eye tumour of childhood that occurs in heritable and non-heritable forms. In the heritable form, there is a predisposition to the development of non-ocular subsequent primary tumours (SPTs).

METHODS: This study included 1927 retinoblastoma patients diagnosed in Britain from 1951 to 2004. Ascertainment was through the (UK) National Registry of Childhood Tumours; cases were followed-up for the occurrence of SPTs. Standardised incidence ratios (SIRs) were calculated.

RESULTS: We identified 169 SPTs in 152 patients. The SIR analysis included 145 SPTs with cancer registrations from the years 1971 to 2009. These tumours occurred in 132 patients: 112 of the 781 heritable and 20 of the 1075 (presumed) non-heritable cases under surveillance at the start of this period developed at least one registered SPT. The SIRs for all tumours combined were 13.7 (95% confidence interval 11.3-16.5) in heritable cases and 1.5 (0.9-2.3) in non-heritable cases. The main types of SPT in the heritable cases were leiomyosarcoma, (31 cases; SIR 1018.7 (692.2-1446.0)), osteosarcoma (26 cases; SIR 444.6 (290.4-651.4)), and skin melanoma (12 cases; SIR 18.6 (9.6-32.4)).

CONCLUSION: The risk of SPTs in heritable retinoblastoma is extremely high. This has important implications for the clinical follow-up and counselling of survivors and their families.

Original language	English
Pages (from-to)	2455-63
Number of pages	9
Journal	British Journal of Cancer
Volume	108
Issue number	12
DOIs	https://doi.org/10.1038/bjc.2013.228
Publication status	Published - 25 Jun 2013

Keywords

Adolescent
Adult
Child
Child, Preschool
Female
Genetic Predisposition to Disease
Humans
Incidence
Infant
Infant, Newborn
Male
Middle Aged
Neoplasms, Second Primary
Registries
Retinal Neoplasms
Retinoblastoma
Survivors
Time Factors
United Kingdom
Young Adult
Journal Article
Research Support, Non-U.S. Gov't

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

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10.1038/bjc.2013.228

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MacCarthy, A., Bayne, A. M., Brownbill, P. A., Bunch, K. J., Diggens, N. L., Draper, G. J., Hawkins, M. M., Jenkinson, H. C., Kingston, J. E., Stiller, C. A., Vincent, T. J., & Murphy, M. F. G. (2013). Second and subsequent tumours among 1927 retinoblastoma patients diagnosed in Britain 1951-2004. British Journal of Cancer, 108(12), 2455-63. https://doi.org/10.1038/bjc.2013.228

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title = "Second and subsequent tumours among 1927 retinoblastoma patients diagnosed in Britain 1951-2004",

abstract = "BACKGROUND: Retinoblastoma is an eye tumour of childhood that occurs in heritable and non-heritable forms. In the heritable form, there is a predisposition to the development of non-ocular subsequent primary tumours (SPTs).METHODS: This study included 1927 retinoblastoma patients diagnosed in Britain from 1951 to 2004. Ascertainment was through the (UK) National Registry of Childhood Tumours; cases were followed-up for the occurrence of SPTs. Standardised incidence ratios (SIRs) were calculated.RESULTS: We identified 169 SPTs in 152 patients. The SIR analysis included 145 SPTs with cancer registrations from the years 1971 to 2009. These tumours occurred in 132 patients: 112 of the 781 heritable and 20 of the 1075 (presumed) non-heritable cases under surveillance at the start of this period developed at least one registered SPT. The SIRs for all tumours combined were 13.7 (95% confidence interval 11.3-16.5) in heritable cases and 1.5 (0.9-2.3) in non-heritable cases. The main types of SPT in the heritable cases were leiomyosarcoma, (31 cases; SIR 1018.7 (692.2-1446.0)), osteosarcoma (26 cases; SIR 444.6 (290.4-651.4)), and skin melanoma (12 cases; SIR 18.6 (9.6-32.4)).CONCLUSION: The risk of SPTs in heritable retinoblastoma is extremely high. This has important implications for the clinical follow-up and counselling of survivors and their families.",

keywords = "Adolescent, Adult, Child, Child, Preschool, Female, Genetic Predisposition to Disease, Humans, Incidence, Infant, Infant, Newborn, Male, Middle Aged, Neoplasms, Second Primary, Registries, Retinal Neoplasms, Retinoblastoma, Survivors, Time Factors, United Kingdom, Young Adult, Journal Article, Research Support, Non-U.S. Gov't",

author = "A MacCarthy and Bayne, {A M} and Brownbill, {P A} and Bunch, {K J} and Diggens, {N L} and Draper, {G J} and Hawkins, {M M} and Jenkinson, {H C} and Kingston, {J E} and Stiller, {C A} and Vincent, {T J} and Murphy, {M F G}",

year = "2013",

month = jun,

day = "25",

doi = "10.1038/bjc.2013.228",

language = "English",

volume = "108",

pages = "2455--63",

journal = "British Journal of Cancer",

issn = "0007-0920",

publisher = "Cancer Research UK",

number = "12",

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TY - JOUR

T1 - Second and subsequent tumours among 1927 retinoblastoma patients diagnosed in Britain 1951-2004

AU - MacCarthy, A

AU - Bayne, A M

AU - Brownbill, P A

AU - Bunch, K J

AU - Diggens, N L

AU - Draper, G J

AU - Hawkins, M M

AU - Jenkinson, H C

AU - Kingston, J E

AU - Stiller, C A

AU - Vincent, T J

AU - Murphy, M F G

PY - 2013/6/25

Y1 - 2013/6/25

N2 - BACKGROUND: Retinoblastoma is an eye tumour of childhood that occurs in heritable and non-heritable forms. In the heritable form, there is a predisposition to the development of non-ocular subsequent primary tumours (SPTs).METHODS: This study included 1927 retinoblastoma patients diagnosed in Britain from 1951 to 2004. Ascertainment was through the (UK) National Registry of Childhood Tumours; cases were followed-up for the occurrence of SPTs. Standardised incidence ratios (SIRs) were calculated.RESULTS: We identified 169 SPTs in 152 patients. The SIR analysis included 145 SPTs with cancer registrations from the years 1971 to 2009. These tumours occurred in 132 patients: 112 of the 781 heritable and 20 of the 1075 (presumed) non-heritable cases under surveillance at the start of this period developed at least one registered SPT. The SIRs for all tumours combined were 13.7 (95% confidence interval 11.3-16.5) in heritable cases and 1.5 (0.9-2.3) in non-heritable cases. The main types of SPT in the heritable cases were leiomyosarcoma, (31 cases; SIR 1018.7 (692.2-1446.0)), osteosarcoma (26 cases; SIR 444.6 (290.4-651.4)), and skin melanoma (12 cases; SIR 18.6 (9.6-32.4)).CONCLUSION: The risk of SPTs in heritable retinoblastoma is extremely high. This has important implications for the clinical follow-up and counselling of survivors and their families.

AB - BACKGROUND: Retinoblastoma is an eye tumour of childhood that occurs in heritable and non-heritable forms. In the heritable form, there is a predisposition to the development of non-ocular subsequent primary tumours (SPTs).METHODS: This study included 1927 retinoblastoma patients diagnosed in Britain from 1951 to 2004. Ascertainment was through the (UK) National Registry of Childhood Tumours; cases were followed-up for the occurrence of SPTs. Standardised incidence ratios (SIRs) were calculated.RESULTS: We identified 169 SPTs in 152 patients. The SIR analysis included 145 SPTs with cancer registrations from the years 1971 to 2009. These tumours occurred in 132 patients: 112 of the 781 heritable and 20 of the 1075 (presumed) non-heritable cases under surveillance at the start of this period developed at least one registered SPT. The SIRs for all tumours combined were 13.7 (95% confidence interval 11.3-16.5) in heritable cases and 1.5 (0.9-2.3) in non-heritable cases. The main types of SPT in the heritable cases were leiomyosarcoma, (31 cases; SIR 1018.7 (692.2-1446.0)), osteosarcoma (26 cases; SIR 444.6 (290.4-651.4)), and skin melanoma (12 cases; SIR 18.6 (9.6-32.4)).CONCLUSION: The risk of SPTs in heritable retinoblastoma is extremely high. This has important implications for the clinical follow-up and counselling of survivors and their families.

KW - Adolescent

KW - Adult

KW - Child

KW - Child, Preschool

KW - Female

KW - Genetic Predisposition to Disease

KW - Humans

KW - Incidence

KW - Infant

KW - Infant, Newborn

KW - Male

KW - Middle Aged

KW - Neoplasms, Second Primary

KW - Registries

KW - Retinal Neoplasms

KW - Retinoblastoma

KW - Survivors

KW - Time Factors

KW - United Kingdom

KW - Young Adult

KW - Journal Article

KW - Research Support, Non-U.S. Gov't

U2 - 10.1038/bjc.2013.228

DO - 10.1038/bjc.2013.228

M3 - Article

C2 - 23674091

SN - 0007-0920

VL - 108

SP - 2455

EP - 2463

JO - British Journal of Cancer

JF - British Journal of Cancer

IS - 12

ER -

Second and subsequent tumours among 1927 retinoblastoma patients diagnosed in Britain 1951-2004

Abstract

Keywords

UN SDGs

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