12q21 Interstitial Deletions: Seven New Syndromic Cases Detected by Array-CGH and Review of the Literature.

12q21 deletion array-CGH congenital anomalies copy number variants (CNVs) developmental delay/intellectual disability (DD/ID) dysmorphisms genetic counseling loss of function patient management variation intolerant genes

Journal

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

Informations de publication

Date de publication:
27 04 2022
Historique:
received: 11 03 2022
revised: 22 04 2022
accepted: 24 04 2022
entrez: 28 5 2022
pubmed: 29 5 2022
medline: 1 6 2022
Statut: epublish

Résumé

Interstitial deletions of the long arm of chromosome 12 are rare, with a dozen patients carrying a deletion in 12q21 being reported. Recently a critical region (CR) has been delimited and could be responsible for the more commonly described clinical features, such as developmental delay/intellectual disability, congenital genitourinary and brain malformations. Other, less frequent, clinical signs do not seem to be correlated to the proposed CR. We present seven new patients harboring non-recurrent deletions ranging from 1 to 18.5 Mb differentially scattered across 12q21. Alongside more common clinical signs, some patients have rarer features such as heart defects, hearing loss, hypotonia and dysmorphisms. The correlation of haploinsufficiency of genes outside the CR to specific signs contributes to our knowledge of the effect of the deletion of this gene-poor region of chromosome 12q. This work underlines the still important role of copy number variations in the diagnostic setting of syndromic patients and the positive reflection on management and family genetic counseling.

Identifiants

pubmed: 35627165
pii: genes13050780
doi: 10.3390/genes13050780
pmc: PMC9141874
pii:
doi:

Types de publication

Journal Article Review Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

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Auteurs

Maria Paola Recalcati (MP)

Laboratorio di Citogenetica e Genetica Molecolare, IRCCS Istituto Auxologico Italiano, 20095 Cusano Milanino, Italy.

Ilaria Catusi (I)

Laboratorio di Citogenetica e Genetica Molecolare, IRCCS Istituto Auxologico Italiano, 20095 Cusano Milanino, Italy.

Maria Garzo (M)

Laboratorio di Citogenetica e Genetica Molecolare, IRCCS Istituto Auxologico Italiano, 20095 Cusano Milanino, Italy.

Serena Redaelli (S)

Dipartimento di Chirurgia e Medicina Traslazionale, Università di Milano-Bicocca, 20126 Milan, Italy.

Marta Massimello (M)

Ospedale San Gerardo, Unità di Genetica Pediatrica, Fondazione Monza e Brianza per il Bambino e la Sua Mamma (MBBM), 20900 Monza, Italy.

Silvia Beatrice Maitz (SB)

Ospedale San Gerardo, Unità di Genetica Pediatrica, Fondazione Monza e Brianza per il Bambino e la Sua Mamma (MBBM), 20900 Monza, Italy.

Mattia Gentile (M)

UOC Genetica Medica, PO Di Venere, ASL Bari, 70131 Bari, Italy.

Emanuela Ponzi (E)

UOC Genetica Medica, PO Di Venere, ASL Bari, 70131 Bari, Italy.

Paola Orsini (P)

UOC Genetica Medica, PO Di Venere, ASL Bari, 70131 Bari, Italy.

Anna Zilio (A)

U.O.S. Laboratorio di Genetica, Azienda ULSS 8 Berica, 36100 Vicenza, Italy.

Annamaria Montaldi (A)

U.O.S. Laboratorio di Genetica, Azienda ULSS 8 Berica, 36100 Vicenza, Italy.

Annapaola Calò (A)

U.O.S. Laboratorio di Genetica, Azienda ULSS 8 Berica, 36100 Vicenza, Italy.

Anna Paola Capra (AP)

Dipartimento di Scienze Biomediche, Odontoiatriche e Delle Immagini Morfologiche e Funzionali, Università di Messina, 98125 Messina, Italy.

Silvana Briuglia (S)

Dipartimento di Scienze Biomediche, Odontoiatriche e Delle Immagini Morfologiche e Funzionali, Università di Messina, 98125 Messina, Italy.

Maria Angela La Rosa (MA)

Dipartimento di Scienze Biomediche, Odontoiatriche e Delle Immagini Morfologiche e Funzionali, Università di Messina, 98125 Messina, Italy.

Lucia Grillo (L)

Research Unit of Rare Diseases and Neurodevelopmental Disorders, Oasi Research Institute-IRCCS, 94018 Troina, Italy.

Corrado Romano (C)

Research Unit of Rare Diseases and Neurodevelopmental Disorders, Oasi Research Institute-IRCCS, 94018 Troina, Italy.
Medical Genetics, Section of Medical Biochemistry, Department of Biomedical and Biotechnological Sciences, University of Catania, 95124 Catania, Italy.

Sebastiano Bianca (S)

Genetica Medica, ARNAS Garibaldi, 95123 Catania, Italy.

Michela Malacarne (M)

IRCCS Istituto Giannina Gaslini, UOC Laboratorio di Genetica Umana, 16147 Genova, Italy.

Martina Busè (M)

U.O.C. Genetica Medica AOOR Villa Sofia-Cervello, 90146 Palermo, Italy.

Maria Piccione (M)

Dipartimento di Scienze della Promozione della Salute, Materno-Infantile, Medicina Interna e Specialistica d'Eccellenza, "G. D'Alessandro" (PROMISE), Università Degli Studi di Palermo, 90144 Palermo, Italy.

Lidia Larizza (L)

Laboratorio di Citogenetica e Genetica Molecolare, IRCCS Istituto Auxologico Italiano, 20095 Cusano Milanino, Italy.

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Classifications MeSH