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. 2019 Jan 14;11(1):86.
doi: 10.3390/cancers11010086.

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

Alessandro Mussa  1   2 , Valentina Pia Ciuffreda  3 , Pina Sauro  4 , Veronica Pagliardini  5 , Severo Pagliardini  6 , Diana Carli  7 , Jennifer M Kalish  8 , Franca Fagioli  9   10 , Enza Pavanello  11 , Giovanni Battista Ferrero  12
Affiliations

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

Alessandro Mussa et al. Cancers (Basel). .

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.

Keywords: Beckwith–Wiedemann syndrome; alpha-fetoprotein; dried blood spot; hepatoblastoma; screening.

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Conflict of interest statement

The authors declare no conflict of interest.

Figures

Figure 1
Figure 1
Correlation between α-fetoprotein (αPF) measured on plasma and on dried blood spot (DBS) (r2 = 0.999, p < 0.001).
Figure 2
Figure 2
The longitudinal evaluation of alpha-fetoprotein (αFP) plasmatic concentration on dried blood spots (DBS, closed circles) overlapped that on standard laboratory method (open circles) in the 12 patients affected by cancer-predisposition syndromes with αFP concentrations >10 U/mL.
See this image and copyright information in PMC

References

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