Mosaic Variegated Aneuploidy syndrome 2 caused by biallelic variants in CEP57, two new cases and review of the phenotype.


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:
Nov 2021
Historique:
received: 06 04 2021
revised: 01 09 2021
accepted: 04 09 2021
pubmed: 10 9 2021
medline: 13 1 2022
entrez: 9 9 2021
Statut: ppublish

Résumé

Mosaic Variegated Aneuploidy Syndrome 2 (MVA2; MIM 614114) is a rare autosomal recessive disorder, characterized by mosaic aneuploidies involving multiple chromosomes and tissues, caused by biallelic pathogenic variants in the CEP57 gene. Only 10 patients have been reported to date. We report two additional non related cases born to Moroccan consanguineous parents, carrying the previously described c.915_925dup11 CEP57 homozygous variant. Common features of these 12 cases include growth retardation, typically of prenatal onset, distinctive facial features, endocrine, cardiovascular and skeletal, abnormalities while malignancies have not been reported. This report describes the phenotypical spectrum of MVA2.

Identifiants

pubmed: 34500087
pii: S1769-7212(21)00204-4
doi: 10.1016/j.ejmg.2021.104338
pii:
doi:

Substances chimiques

CEP57 protein, human 0
Microtubule-Associated Proteins 0
Nuclear Proteins 0

Types de publication

Case Reports Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

104338

Informations de copyright

Copyright © 2021 Elsevier Masson SAS. All rights reserved.

Auteurs

Fernando Santos-Simarro (F)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: fernando.santos@salud.madrid.org.

Marta Pacio (M)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: martapaciomiguez@gmail.com.

Anna María Cueto-González (AM)

Departamento de Genética Clinical y Molecular,Grupo de Genética Médica, Hospital Universitari Vall d'Hebron, Barcelona, Spain; Institut de Recerca (VHIR), Hospital Universitari, Vall d'Hebron, Barcelona, Spain; European Reference Network, ERN ITHACA, Spain; European Reference Network, ERN CRANIO, Spain. Electronic address: amcueto@vhebron.net.

Elena Mansilla (E)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: elbuzonelena@gmail.com.

María Irene Valenzuela-Palafoll (MI)

Departamento de Genética Clinical y Molecular,Grupo de Genética Médica, Hospital Universitari Vall d'Hebron, Barcelona, Spain; Institut de Recerca (VHIR), Hospital Universitari, Vall d'Hebron, Barcelona, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: irenevalenzuelapalafoll@gmail.com.

Fermina López-Grondona (F)

Departamento de Genética Clinical y Molecular,Grupo de Genética Médica, Hospital Universitari Vall d'Hebron, Barcelona, Spain; Institut de Recerca (VHIR), Hospital Universitari, Vall d'Hebron, Barcelona, Spain. Electronic address: ferminalopez@gmail.com.

María Dolores Lledín (MD)

Servicio de Hepatología Pediátrica, Hospital Universitario La Paz, Madrid, Spain. Electronic address: dolores.lledin@salud.madrid.org.

Cristina Schuffelmann (C)

Servicio de Cuidados Intensivos Pediátricos, Hospital Universitario La Paz, Madrid, Spain. Electronic address: cristina.schuffelmann@salud.madrid.org.

Ángela Del Pozo (Á)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain. Electronic address: ingemm.adelpozo@gmail.com.

Mario Solis (M)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain. Electronic address: ingemm.msolis@gmail.com.

Patricia Vallcorba (P)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: isabel.vallcorba@salud.madrid.org.

Pablo Lapunzina (P)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: plapunzina@gmail.com.

Juan José Menéndez Suso (JJ)

Servicio de Cuidados Intensivos Pediátricos, Hospital Universitario La Paz, Madrid, Spain. Electronic address: juanjomen@yahoo.es.

Sofia M Siccha (SM)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain. Electronic address: siccha.sofiam@gmail.com.

Juan Manuel Montejo (JM)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain. Electronic address: juanmanuel.montejo@salud.madrid.org.

Rocío Mena (R)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain. Electronic address: mariarocio.mena@salud.madrid.org.

Carmen Jiménez-Rodríguez (C)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain. Electronic address: crodriguezj@salud.madrid.org.

Sixto García-Miñaúr (S)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: sixto.garciamin@gmail.com.

María Palomares-Bralo (M)

Instituto de Genética Medica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Madrid, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER, U753), ISCIII, Madrid, Spain; European Reference Network, ERN ITHACA, Spain. Electronic address: mpalomares.ingemm@gmail.com.

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