Expanding Phenotype of Poirier-Bienvenu Syndrome: New Evidence from an Italian Multicentrical Cohort of Patients.


Journal

Genes
ISSN: 2073-4425
Titre abrégé: Genes (Basel)
Pays: Switzerland
ID NLM: 101551097

Informations de publication

Date de publication:
30 01 2022
Historique:
received: 31 12 2021
revised: 19 01 2022
accepted: 23 01 2022
entrez: 25 2 2022
pubmed: 26 2 2022
medline: 22 4 2022
Statut: epublish

Résumé

Poirier-Bienvenu Neurodevelopmental Syndrome (POBINDS) is a rare disease linked to mutations of the Our multicentric, retrospective study recruited nine patients with POBINDS, detected using next-generation sequencing panels and whole-exome sequencing. Clinical, laboratory, and neuroimaging data were reported for each patient in order to assess the severity of phenotype, and eventually, a correlation with the type of We reported nine unrelated patients with heterozygous de novo mutations of the Although it was not possible to assess a genotype-phenotype correlation in our patients, our research further expands the phenotype spectrum of POBINDS patients, identifying new mutations occurring in the

Sections du résumé

BACKGROUND
Poirier-Bienvenu Neurodevelopmental Syndrome (POBINDS) is a rare disease linked to mutations of the
METHODS
Our multicentric, retrospective study recruited nine patients with POBINDS, detected using next-generation sequencing panels and whole-exome sequencing. Clinical, laboratory, and neuroimaging data were reported for each patient in order to assess the severity of phenotype, and eventually, a correlation with the type of
RESULTS
We reported nine unrelated patients with heterozygous de novo mutations of the
CONCLUSION
Although it was not possible to assess a genotype-phenotype correlation in our patients, our research further expands the phenotype spectrum of POBINDS patients, identifying new mutations occurring in the

Identifiants

pubmed: 35205321
pii: genes13020276
doi: 10.3390/genes13020276
pmc: PMC8872204
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Références

Nat Commun. 2018 Feb 26;9(1):838
pubmed: 29483533
Am J Med Genet A. 2021 Feb;185(2):539-543
pubmed: 33166063
Prog Neurobiol. 2000 Feb;60(3):211-46
pubmed: 10658642
Hum Mutat. 2017 Aug;38(8):932-941
pubmed: 28585349
J Hum Genet. 2019 Apr;64(4):313-322
pubmed: 30655572
Signal Transduct Target Ther. 2021 May 17;6(1):183
pubmed: 33994545
Epilepsia. 2021 Jul;62(7):e103-e109
pubmed: 34041744
Neurogenetics. 2021 Oct;22(4):323-332
pubmed: 34370157
Int J Mol Sci. 2019 Nov 26;20(23):
pubmed: 31779225
Bioinformatics. 2019 Jun 1;35(11):1978-1980
pubmed: 30376034
Seizure. 2021 Dec;93:133-139
pubmed: 34740143
J Biol Chem. 2006 Jul 7;281(27):18394-400
pubmed: 16672224
Biomed Rep. 2017 Feb;6(2):127-133
pubmed: 28357063
Hum Mutat. 2017 Nov;38(11):1611-1612
pubmed: 28762608
Sci Rep. 2019 Nov 29;9(1):17909
pubmed: 31784560
Blood. 2010 Oct 7;116(14):2513-21
pubmed: 20576813
Neurosci Lett. 2018 Feb 22;667:4-9
pubmed: 28499889

Auteurs

Alessandro Orsini (A)

Pediatric Neurology, Pediatric Department, Santa Chiara University Hospital, Azienda Ospedaliero Universitaria Pisana, 56126 Pisa, Italy.

Andrea Santangelo (A)

Pediatric Neurology, Pediatric Department, Santa Chiara University Hospital, Azienda Ospedaliero Universitaria Pisana, 56126 Pisa, Italy.

Francesca Bravin (F)

Pediatric Neurology, Pediatric Department, Santa Chiara University Hospital, Azienda Ospedaliero Universitaria Pisana, 56126 Pisa, Italy.

Alice Bonuccelli (A)

Pediatric Neurology, Pediatric Department, Santa Chiara University Hospital, Azienda Ospedaliero Universitaria Pisana, 56126 Pisa, Italy.

Diego Peroni (D)

Pediatric Neurology, Pediatric Department, Santa Chiara University Hospital, Azienda Ospedaliero Universitaria Pisana, 56126 Pisa, Italy.
Department of Clinical and Experimental Medicine, University of Pisa, 56126 Pisa, Italy.

Roberta Battini (R)

Department of Clinical and Experimental Medicine, University of Pisa, 56126 Pisa, Italy.
Stella Maris Foundation, IRCCS, 56126 Calambrone, Italy.

Thomas Foiadelli (T)

Pediatric Clinic, Department of Clinical-Surgical, Diagnostic and Pediatric Sciences, IRCCS Policlinico San Matteo Foundation-University of Pavia, 27100 Pavia, Italy.

Veronica Bertini (V)

Cytogenetics Unit, Santa Chiara University Hospital, Azienda Ospedaliero Universitaria Pisana, 56126 Pisa, Italy.

Angelo Valetto (A)

Cytogenetics Unit, Santa Chiara University Hospital, Azienda Ospedaliero Universitaria Pisana, 56126 Pisa, Italy.

Michele Iacomino (M)

Unit of Medical Genetics, IRCCS Istituto Giannina Gaslini, 16147 Genova, Italy.

Vincenzo Nigro (V)

Medical Genetics, Department of Biochemistry, Biophysics and General Pathology University of Campania, Luigi Vanvitelli, 81100 Caserta, Italy.

Anna Laura Torella (AL)

Medical Genetics, Department of Biochemistry, Biophysics and General Pathology University of Campania, Luigi Vanvitelli, 81100 Caserta, Italy.

Marcello Scala (M)

Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, 16147 Genoa, Italy.
Paediatric Neurology and Muscular Disease Unit, IRCCS Istituto Giannina Gaslini, 16147 Genova, Italy.

Valeria Capra (V)

Unit of Medical Genetics, IRCCS Istituto Giannina Gaslini, 16147 Genova, Italy.

Maria Stella Vari (MS)

Paediatric Neurology and Muscular Disease Unit, IRCCS Istituto Giannina Gaslini, 16147 Genova, Italy.

Anna Fetta (A)

IRCCS Istituto delle Scienze Neurologiche di Bologna, UOC Neuropsichiatria dell'età Pediatrica, 40138 Bologna, Italy.

Veronica Di Pisa (V)

IRCCS Istituto delle Scienze Neurologiche di Bologna, UOC Neuropsichiatria dell'età Pediatrica, 40138 Bologna, Italy.

Francesca Montanari (F)

U.O. Genetica Medica, IRCCS Azienda Ospedaliero-Universitaria di Bologna, 40138 Bologna, Italy.

Roberta Epifanio (R)

Clinical Neurophysiology Unit, Scientific Institute IRCCS E. Medea, 23842 Bosisio Parini, Italy.

Paolo Bonanni (P)

Epilepsy and Clinical Neurophysiology Unit, IRCCS E. Medea Scientific Institute, 31015 Conegliano, Italy.

Roberto Giorda (R)

Molecular Biology Laboratory, IRCCS E. Medea Scientific Institute, 23842 Bosisio Parini, Italy.

Francesca Operto (F)

Child Neuropsychiatry Unit, Department of Medicine, Surgery and Dentistry, University of Salerno, 84084 Fisciano, Italy.

Grazia Pastorino (G)

Child Neuropsychiatry Unit, Department of Medicine, Surgery and Dentistry, University of Salerno, 84084 Fisciano, Italy.

Esra Sarigecili (E)

Department of Pediatric Neurology, Gazi University Faculty of Medicine, 06500 Ankara, Turkey.

Esra Sardaroglu (E)

Department of Pediatric Neurology, Gazi University Faculty of Medicine, 06500 Ankara, Turkey.

Cetin Okuyaz (C)

Department of Pediatric Neurology, Mersin University, Mersin 33110, Turkey.

Sevgan Bozdogan (S)

Department of Medical Genetics, Balcali Clinics and Hospital, Faculty of Medicine, Cukurova University, Adana 01330, Turkey.

Luciana Musante (L)

Medical Genetics Unit, Institute for Maternal and Child Health-IRCCS "Burlo Garofolo", 34137 Trieste, Italy.

Flavio Faletra (F)

Medical Genetics Unit, Institute for Maternal and Child Health-IRCCS "Burlo Garofolo", 34137 Trieste, Italy.

Caterina Zanus (C)

Child Neuropsychiatry Unit, Institute for Maternal and Child Health-IRCCS "Burlo Garofolo", 34137 Trieste, Italy.

Alessandro Ferretti (A)

Rare and Complex Epilepsy Unit, Department of Neuroscience, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.

Federico Vigevano (F)

Neuroscience Department, Bambino Gesù Children's Hospital, IRCCS, Full Member of European Reference Network EPICARE, 00165 Rome, Italy.

Pasquale Striano (P)

Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, 16147 Genoa, Italy.
Paediatric Neurology and Muscular Disease Unit, IRCCS Istituto Giannina Gaslini, 16147 Genova, Italy.

Duccio Maria Cordelli (DM)

IRCCS Istituto delle Scienze Neurologiche di Bologna, UOC Neuropsichiatria dell'età Pediatrica, 40138 Bologna, Italy.

Articles similaires

[Redispensing of expensive oral anticancer medicines: a practical application].

Lisanne N van Merendonk, Kübra Akgöl, Bastiaan Nuijen
1.00
Humans Antineoplastic Agents Administration, Oral Drug Costs Counterfeit Drugs

Smoking Cessation and Incident Cardiovascular Disease.

Jun Hwan Cho, Seung Yong Shin, Hoseob Kim et al.
1.00
Humans Male Smoking Cessation Cardiovascular Diseases Female
Humans United States Aged Cross-Sectional Studies Medicare Part C
1.00
Humans Yoga Low Back Pain Female Male

Classifications MeSH