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Cancers 2019, 11(1), 86; https://doi.org/10.3390/cancers11010086

Longitudinal Monitoring of Alpha-Fetoprotein by Dried Blood Spot for Hepatoblastoma Screening in Beckwith–Wiedemann Syndrome

1
Department of Public Health and Pediatric Sciences, University of Torino, 10126 Torino, Italy
2
Neonatal Intensive Care Unit, Department of Obstetrics and Gynecology, S.Anna Hospital, Città della Salute e della Scienza, 10126 Torino, Italy
3
Division of Human Genetics, the Children’s Hospital of Philadelphia and Department of Pediatrics, The Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA 19104, USA
4
Pediatric Onco-Hematology, Stem Cell Transplantation and Cellular Therapy Division, Regina Margherita Children’s Hospital, Città della Salute e della Scienza, 10126 Torino, Italy
*
Author to whom correspondence should be addressed.
Received: 11 November 2018 / Revised: 21 December 2018 / Accepted: 11 January 2019 / Published: 14 January 2019
(This article belongs to the Special Issue Hepatoblastoma and Pediatric Liver Tumors)
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Abstract

Background: Hepatoblastoma screening in the Beckwith–Wiedemann spectrum (BWSp) is currently based on measuring a specific serum marker alpha-fetoprotein (αFP) every three months until the fourth birthday. Frequent blood draws can be a burden for patients and their families. Methods: We have developed a less invasive alternative testing method based on measuring αFPs from dried blood spots (DBS). The method was validated with 259 simultaneous plasma and DBS αFP measurements in 171 children (132 controls and 39 patients with BWSp). Results: The DBS and plasma measurements overlapped across the wide range of αFP concentrations independent of patient age (p < 0.0001), demonstrating the utility of this method for longitudinal monitoring. Occasional differences between measurements by the two techniques fell within standard laboratory error and would not alter clinical management. Conclusions: This novel method shows consistent overlap with the traditional blood draws, thereby demonstrating its utility for hepatoblastoma screening in this setting and alleviating the burden of frequent blood draws. This also may help increase patient compliance and reduce costs of health care screening. The DBS-based method for the measurement of cancer biomarkers may also be applied to several other chronic diseases with increased risks of αFP-producing liver tumors. View Full-Text
Keywords: alpha-fetoprotein; Beckwith–Wiedemann syndrome; dried blood spot; hepatoblastoma; screening alpha-fetoprotein; Beckwith–Wiedemann syndrome; dried blood spot; hepatoblastoma; screening
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This is an open access article distributed under the Creative Commons Attribution License which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited (CC BY 4.0).

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Mussa, A.; Ciuffreda, V.P.; Sauro, P.; Pagliardini, V.; Pagliardini, S.; Carli, D.; Kalish, J.M.; Fagioli, F.; Pavanello, E.; Ferrero, G.B. Longitudinal Monitoring of Alpha-Fetoprotein by Dried Blood Spot for Hepatoblastoma Screening in Beckwith–Wiedemann Syndrome. Cancers 2019, 11, 86.

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