Duncan NRI Faculty and Staff Publications

Language

English

Publication Date

6-1-2022

Journal

Prenatal Diagnosis

DOI

10.1002/pd.6144

PMID

35394072

PMCID

PMC9995893

PubMedCentral® Posted Date

6-1-2023

PubMedCentral® Full Text Version

Author MSS

Abstract

The disease burden of de novo mutations (DNMs) has been evidenced only recently when the common application of next-generation sequencing technologies enabled their reliable and affordable detection through family-based clinical exome or genome sequencing. Implementation of exome sequencing into prenatal diagnostics revealed that up to 63% of pathogenic or likely pathogenic variants associated with fetal structural anomalies are apparently de novo, primarily for autosomal dominant disorders. Apparent DNMs have been considered to primarily occur as germline or zygotic events, with consequently negligible recurrence risks. However, there is now evidence that a considerable proportion of them are in fact inherited from a parent mosaic for the variant. Here, we review the burden of DNMs in prenatal diagnostics and the influence of parental mosaicism on the interpretation of apparent DNMs and discuss the challenges with detecting and quantifying parental mosaicism and its effect on recurrence risk. We also describe new bioinformatic and technological tools developed to assess mosaicism and discuss how they improve the accuracy of reproductive risk counseling when parental mosaicism is detected.

Keywords

Counseling, Female, Humans, Mosaicism, Mutation, Parents, Pregnancy, Pregnancy Trimester, First, Ultrasonography, Prenatal

Published Open-Access

yes

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