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Titolo:
Screening for Wilms tumor and hepatoblastoma in children with Beckwith-Wiedemann syndromes: A cost-effective model
Autore:
McNeil, DE; Brown, M; Ching, A; DeBaun, MR;
Indirizzi:
NCI, Genet Epidemiol Branch, Div Genet Epidemiol, NIH,EPS, Bethesda, MD 20892 USA NCI Bethesda MD USA 20892 enet Epidemiol, NIH,EPS, Bethesda, MD 20892 USA NCI, Appl Res Branch, DCPC, NIH, Bethesda, MD 20892 USA NCI Bethesda MD USA 20892 l Res Branch, DCPC, NIH, Bethesda, MD 20892 USA Washington Univ, Sch Med, Dept Pediat, St Louis, MO 63110 USA Washington Univ St Louis MO USA 63110 Dept Pediat, St Louis, MO 63110 USA
Titolo Testata:
MEDICAL AND PEDIATRIC ONCOLOGY
fascicolo: 4, volume: 37, anno: 2001,
pagine: 349 - 356
SICI:
0098-1532(200110)37:4<349:SFWTAH>2.0.ZU;2-6
Fonte:
ISI
Lingua:
ENG
Soggetto:
HEMIHYPERTROPHY; CANCER; ANIRIDIA; RISK; LIFE; CT;
Keywords:
Beckwith-Wiedemann syndrome; cost-effectiveness analysis; Wilms tumor; cancer screening;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Clinical Medicine
Citazioni:
21
Recensione:
Indirizzi per estratti:
Indirizzo: DeBaun, MR NCI, Genet Epidemiol Branch, Div Genet Epidemiol, NIH,EPS, Room7125,6120 Execut Blvd,MSC 7236, Bethesda, MD 20892 USA NCI Room 7125,6120 Execut Blvd,MSC 7236 Bethesda MD USA 20892 A
Citazione:
D.E. McNeil et al., "Screening for Wilms tumor and hepatoblastoma in children with Beckwith-Wiedemann syndromes: A cost-effective model", MED PED ONC, 37(4), 2001, pp. 349-356

Abstract

Background. We undertook a cost-benefit analysis of screening for Wilms tumor and hepatoblastoma in children with Beckwith-Wiedemann syndrome (BWS), a known cancer predisposition syndrome. The purpose of this analysis was twofold: first, to assess whether screening in children with BWS has the potential to be cost-effective; second, if screening appears to be cost-effective, to determine which parameters would be most important to assess if a screening trial were initiated. Procedures. We used data from the BWS registry at the National Cancer Institute, the National Wilms Tumor Study (NWTS), and large published series to model events for two hypothetical cohorts of 1,000 infants born with BWS. One hypothetical cohort was screened for cancer until a predetermined age, representing the base case. The other cohort was unscreened. For our base case, we assumed: (a) sonography examinations three times yearly (triannually) from birth until 7 years of age; (b) screening would result in one stage shift downward at diagnosis for Wilms tumor and hepatoblastoma; (c) 100% sensitivity and 95% specificity for detecting clinical stage I Wilms tumor and hepatoblastoma; (d) a 3% discount rate; (e)a false positive result cost of $402. We estimated mortality rates based on published Wilms tumor and hepatoblastoma stage specific survival. Results. Using the base case, screening a child with BWS from birth until 4 years of age results in a cost per life year saved of $9,642 while continuing until 7 years of age results in a cost per life-year saved of $14,740. When variables such as cost of screening examination, discount rate, and effectiveness of screening were varied based on high and low estimates, the incremental cost per life-year saved for screening up until age four remained comparable to acceptable population based cancer screening ranges (<$50,000 per life year saved). Conclusions. Under our model's assumptions, abdominal sonography examinations in children with BWS represent a reasonable strategy tor a cancer screening program. A cancer screening trial is warranted to determine if, when, and how often children with BWS should be screened and to determine cost-effectiveness in clinical practice.

ASDD Area Sistemi Dipartimentali e Documentali, Università di Bologna, Catalogo delle riviste ed altri periodici
Documento generato il 20/01/21 alle ore 11:33:21