External hydrocephalus as a prenatal feature of noonan syndrome.


Journal

Annals of human genetics
ISSN: 1469-1809
Titre abrégé: Ann Hum Genet
Pays: England
ID NLM: 0416661

Informations de publication

Date de publication:
11 2021
Historique:
revised: 28 03 2021
received: 30 01 2021
accepted: 22 05 2021
pubmed: 3 6 2021
medline: 15 12 2021
entrez: 2 6 2021
Statut: ppublish

Résumé

Brain malformations have been reported in RASopathies, including postnatal external hydrocephalus, a nonobstructive form of cerebrospinal fluid accumulation around the brain. It was described in a few patients with mutations of other genes than PTPN11, such as SOS1 and SHOC2 and never in prenatal diagnosis. The aim of this case report is to describe the prenatal presentation of a fetus with Noonan syndrome (NS) and external hydrocephalus. We report on a Noonan syndrome fetus with a de novo pathogenic PTPN11 c.923A>G p.Asn308Ser mutation, showing external hydrocephalus, an extremely rare fetal finding, corpus callosum, and cerebellar vermis under the 10th centile, plus a typical NS cardiopathy. This is the first case of Noonan syndrome prenatal diagnosis in a fetus presenting with external hydrocephalus. Following pathophysiological considerations, we suggest to consider NS in the differential diagnosis of external hydrocephalus, investigating other evocative findings and considering molecular screening for mutations in NS-related genes.

Identifiants

pubmed: 34075583
doi: 10.1111/ahg.12436
doi:

Substances chimiques

PTPN11 protein, human EC 3.1.3.48
Protein Tyrosine Phosphatase, Non-Receptor Type 11 EC 3.1.3.48

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

249-252

Informations de copyright

© 2021 John Wiley & Sons Ltd/University College London.

Références

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Auteurs

Gioia Mastromoro (G)

Department of Experimental Medicine, Sapienza University of Rome, Policlinico Umberto I Hospital, Rome, Italy.

Alessandro De Luca (A)

Ospedale Casa Sollievo della Sofferenza, Mendel Institute, San Giovanni Rotondo, Italy.

Enrica Marchionni (E)

Department of Experimental Medicine, Sapienza University of Rome, Policlinico Umberto I Hospital, Rome, Italy.

Antonella Spagnuolo (A)

Centro Sant'Anna ASL Roma1, Rome, Italy.

Flavia Ventriglia (F)

Department of Pediatrics, Sapienza University of Rome, Policlinico Umberto I Hospital, Rome, Italy.

Lucia Manganaro (L)

Department of Radiological Science, Sapienza University of Rome, Policlinico Umberto I Hospital, Rome, Italy.

Antonio Pizzuti (A)

Department of Experimental Medicine, Sapienza University of Rome, Policlinico Umberto I Hospital, Rome, Italy.
Ospedale Casa Sollievo della Sofferenza, Mendel Institute, San Giovanni Rotondo, Italy.

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