Two cases of 16q12.1q21 deletions and refinement of the critical region.


Journal

European journal of medical genetics
ISSN: 1878-0849
Titre abrégé: Eur J Med Genet
Pays: Netherlands
ID NLM: 101247089

Informations de publication

Date de publication:
Jun 2020
Historique:
received: 16 07 2019
revised: 27 01 2020
accepted: 07 02 2020
pubmed: 12 2 2020
medline: 30 12 2020
entrez: 12 2 2020
Statut: ppublish

Résumé

Interstitial deletions of 16q chromosome including 16q12.1q21 region are very rare, with only three cases reported to date. Main clinical features include dysmorphisms, short stature, microcephaly, eye abnormalities, epilepsy, development delay, intellectual disability, and autism spectrum disorder. We report two independent subjects with 16q12.1q21 deletion syndrome presenting with dysmorphic facial features, developmental delay, strabismus, and aggressive behavior. A minimal region of overlap spanning 1.7 Mb on chromosome 16, including IRX5, GNAO1, and NUDT21 genes was shared among these two cases and those previously reported. This minimal region of overlap suggests the potential pathogenic role of these genes, previously implicated in diseases of the central nervous system.

Identifiants

pubmed: 32045705
pii: S1769-7212(19)30485-9
doi: 10.1016/j.ejmg.2020.103878
pii:
doi:

Substances chimiques

Cleavage And Polyadenylation Specificity Factor 0
GNAO1 protein, human 0
Homeodomain Proteins 0
IRX5 protein, human 0
Nudt21 protein, human 0
Transcription Factors 0
GTP-Binding Protein alpha Subunits, Gi-Go EC 3.6.5.1

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

103878

Informations de copyright

Copyright © 2020 Elsevier Masson SAS. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of competing interest The authors declare no conflicts of interest and they received no primary funding for this research.

Auteurs

Diletta Apuzzo (D)

Department of Translational Medicine, Federico II University, Naples, Italy.

Gerarda Cappuccio (G)

Department of Translational Medicine, Federico II University, Naples, Italy; Telethon Institute of Genetics and Medicine, Pozzuoli, Naples, Italy.

Taneli Vaisanen (T)

Department of Clinical Genetics, Turku University Hospital, Turku, Finland.

Marianna Alagia (M)

Department of Translational Medicine, Federico II University, Naples, Italy.

Piero Pignataro (P)

Department of Molecular Medicine and Medical Biotechnology, Federico II University, Naples, Italy.

Rita Genesio (R)

Department of Molecular Medicine and Medical Biotechnology, Federico II University, Naples, Italy.

Nicola Brunetti-Pierri (N)

Department of Translational Medicine, Federico II University, Naples, Italy; Telethon Institute of Genetics and Medicine, Pozzuoli, Naples, Italy. Electronic address: brunetti@tigem.it.

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Classifications MeSH