Radiographic screening of infants and young children with genetic predisposition for rare malignancies: DICER1 mutations and pleuropulmonary blastoma

AJR. American Journal of Roentgenology
Divya G SabapathyAnanth V Annapragada

Abstract

The purpose of this study was to compare the risks of radiation in screening strategies using chest radiographs and CT to detect a rare cancer in a genetically predisposed population against the risks of undetected disease. A decision analytic model of diagnostic imaging screening strategies was built to predict outcomes and cumulative radiation doses for children with DICER1 mutations screened for pleuropulmonary blastoma. Screening strategies compared were chest radiographs followed by chest CT for a positive radiographic result and CT alone. Screening frequencies ranged from once in 3 years to once every 3 months. BEIR VII (model VII proposed by the Committee on the Biological Effects of Ionizing Radiation) risk tables were used to predict excess cancer mortality for each strategy, and the corresponding loss of life expectancy was calculated using Surveillance Epidemiologic and End Results (SEER) statistics. Loss of life expectancy owing to undetected progressive pleuropulmonary blastoma was estimated on the basis of data from the International Pleuropulmonary Blastoma Registry. Sensitivity analysis was performed for all model parameters. Loss of life expectancy owing to undetected disease in an unscreened population exceede...Continue Reading

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Citations

Oct 21, 2016·Pediatric Blood & Cancer·David J LiuDouglas Strother
Nov 11, 2016·Familial Cancer·Eduardo J Bardón-CanchoCarmen Garrido-Colino
Sep 30, 2017·Pediatric Blood & Cancer·Kalene van EngelenHarriet Druker
Nov 14, 2018·Radiographics : a Review Publication of the Radiological Society of North America, Inc·John P LichtenbergerEllen M Chung
Oct 30, 2015·Clinical Dysmorphology·Teck Wah TingSaumya S Jamuar
Oct 26, 2018·Case Reports in Obstetrics and Gynecology·B WormaldD P J Barton
Oct 18, 2019·Pediatric Radiology·R Paul GuillermanJohn R Priest
Jul 28, 2019·Children·Samantha KnightAndrew J Murphy

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