Cargando…

A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series

Fetal Nuchal fluid collections can manifest with two distinct presentations attributable to the same phenotypic spectrum: increased nuchal translucency (iNT) and cystic hygroma. The prenatal detection of these findings should prompt an accurate assessment through genetic counseling and testing, incl...

Descripción completa

Detalles Bibliográficos
Autores principales: Mastromoro, Gioia, Guadagnolo, Daniele, Khaleghi Hashemian, Nader, Bernardini, Laura, Giancotti, Antonella, Piacentini, Gerardo, De Luca, Alessandro, Pizzuti, Antonio
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9818917/
https://www.ncbi.nlm.nih.gov/pubmed/36611340
http://dx.doi.org/10.3390/diagnostics13010048
_version_ 1784865103259107328
author Mastromoro, Gioia
Guadagnolo, Daniele
Khaleghi Hashemian, Nader
Bernardini, Laura
Giancotti, Antonella
Piacentini, Gerardo
De Luca, Alessandro
Pizzuti, Antonio
author_facet Mastromoro, Gioia
Guadagnolo, Daniele
Khaleghi Hashemian, Nader
Bernardini, Laura
Giancotti, Antonella
Piacentini, Gerardo
De Luca, Alessandro
Pizzuti, Antonio
author_sort Mastromoro, Gioia
collection PubMed
description Fetal Nuchal fluid collections can manifest with two distinct presentations attributable to the same phenotypic spectrum: increased nuchal translucency (iNT) and cystic hygroma. The prenatal detection of these findings should prompt an accurate assessment through genetic counseling and testing, including karyotype, chromosomal microarray analysis (CMA) and multigene RASopathy panel. We performed a systematic review of the literature and meta-analysis, to calculate diagnostic yields of genetic testing in fetuses with iNT and cystic hygroma. We compared the results with a cohort of 96 fetuses with these isolated findings. Fetuses with isolated NT ≥ 2.5 mm showed karyotype anomalies in 22.76% of cases and CMA presented an incremental detection rate of 2.35%. Fetuses with isolated NT ≥ 3 mm presented aneuploidies in 14.36% of cases and CMA had an incremental detection rate of 3.89%. When the isolated NT measured at least 3.5 mm the diagnostic yield of karyotyping was 34.35%, the incremental CMA detection rate was 4.1%, the incremental diagnostic rate of the RASopathy panel was 1.44% and it was 2.44% for exome sequencing. Interestingly, CMA presents a considerable diagnostic yield in the group of fetuses with NT ≥ 3.5 mm. Similarly, exome sequencing appears to show promising results and could be considered after a negative CMA result.
format Online
Article
Text
id pubmed-9818917
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher MDPI
record_format MEDLINE/PubMed
spelling pubmed-98189172023-01-07 A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series Mastromoro, Gioia Guadagnolo, Daniele Khaleghi Hashemian, Nader Bernardini, Laura Giancotti, Antonella Piacentini, Gerardo De Luca, Alessandro Pizzuti, Antonio Diagnostics (Basel) Systematic Review Fetal Nuchal fluid collections can manifest with two distinct presentations attributable to the same phenotypic spectrum: increased nuchal translucency (iNT) and cystic hygroma. The prenatal detection of these findings should prompt an accurate assessment through genetic counseling and testing, including karyotype, chromosomal microarray analysis (CMA) and multigene RASopathy panel. We performed a systematic review of the literature and meta-analysis, to calculate diagnostic yields of genetic testing in fetuses with iNT and cystic hygroma. We compared the results with a cohort of 96 fetuses with these isolated findings. Fetuses with isolated NT ≥ 2.5 mm showed karyotype anomalies in 22.76% of cases and CMA presented an incremental detection rate of 2.35%. Fetuses with isolated NT ≥ 3 mm presented aneuploidies in 14.36% of cases and CMA had an incremental detection rate of 3.89%. When the isolated NT measured at least 3.5 mm the diagnostic yield of karyotyping was 34.35%, the incremental CMA detection rate was 4.1%, the incremental diagnostic rate of the RASopathy panel was 1.44% and it was 2.44% for exome sequencing. Interestingly, CMA presents a considerable diagnostic yield in the group of fetuses with NT ≥ 3.5 mm. Similarly, exome sequencing appears to show promising results and could be considered after a negative CMA result. MDPI 2022-12-23 /pmc/articles/PMC9818917/ /pubmed/36611340 http://dx.doi.org/10.3390/diagnostics13010048 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Systematic Review
Mastromoro, Gioia
Guadagnolo, Daniele
Khaleghi Hashemian, Nader
Bernardini, Laura
Giancotti, Antonella
Piacentini, Gerardo
De Luca, Alessandro
Pizzuti, Antonio
A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series
title A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series
title_full A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series
title_fullStr A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series
title_full_unstemmed A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series
title_short A Pain in the Neck: Lessons Learnt from Genetic Testing in Fetuses Detected with Nuchal Fluid Collections, Increased Nuchal Translucency versus Cystic Hygroma—Systematic Review of the Literature, Meta-Analysis and Case Series
title_sort pain in the neck: lessons learnt from genetic testing in fetuses detected with nuchal fluid collections, increased nuchal translucency versus cystic hygroma—systematic review of the literature, meta-analysis and case series
topic Systematic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9818917/
https://www.ncbi.nlm.nih.gov/pubmed/36611340
http://dx.doi.org/10.3390/diagnostics13010048
work_keys_str_mv AT mastromorogioia apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT guadagnolodaniele apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT khaleghihashemiannader apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT bernardinilaura apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT giancottiantonella apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT piacentinigerardo apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT delucaalessandro apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT pizzutiantonio apaininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT mastromorogioia paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT guadagnolodaniele paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT khaleghihashemiannader paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT bernardinilaura paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT giancottiantonella paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT piacentinigerardo paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT delucaalessandro paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries
AT pizzutiantonio paininthenecklessonslearntfromgenetictestinginfetusesdetectedwithnuchalfluidcollectionsincreasednuchaltranslucencyversuscystichygromasystematicreviewoftheliteraturemetaanalysisandcaseseries