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PMID: 23756437 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Severe forms of Baraitser-Winter syndrome are caused by ACTB mutations rather than ACTG1 mutations.

European journal of human genetics : EJHG ·Vol. 22 ·No. 2 ·2014-02-00 ·Pages 179-83

Di Donato N, Rump A, Koenig R, Der Kaloustian VM, Halal F, Sonntag K, Krause C, Hackmann K, Hahn G, Schrock E, Verloes A

Abstract

ACTB and ACTG1 mutations have recently been reported to cause Baraitser-Winter syndrome (BRWS) - a rare condition characterized by ptosis, colobomata, neuronal migration disorder, distinct facial anomalies and intellectual disability. One of the patients carrying an ACTB mutation was previously diagnosed with Fryns-Aftimos syndrome (FAS), which is a rare and severe, multiple congenital anomaly (MCA) syndrome whose symptoms partially overlap with that of BRWS. However, several patients with Fryns-Aftimos were considered not to fit into the ACTB and ACTG1 spectrum because of their severe impairment and additional malformations. We report on three patients who had been diagnosed with FAS. All three patients carry a mutation in the ACTB gene. On the basis of the ACTB mutations and analysis of the clinical findings, we reclassify the diagnosis of these patients as severe BRWS. We suggest that mutations in ACTB cause a distinctly more severe phenotype than ACTG1 mutations, despite the structural similarity of beta- and gamma-actins and their overlapping expression pattern. We expand the spectrum of BRWS and confirm that FAS is not a separate entity but an early and severe manifestation of BRWS.

MeSH Terms
Abnormalities, Multiple/classification,genetics,pathology Actins/genetics Adolescent Child DNA Mutational Analysis Female Genetic Association Studies Humans Male Mutation, Missense Phenotype Severity of Illness Index Syndrome Young Adult
Chemicals
ACTG1 protein, human Actins
Authors & Affiliations
11 authors, click to expand affiliations / ORCID
Di Donato N
Institute for Clinical Genetics, Faculty of Medicine Carl Gustav Carus TU Dresden, Dresden, Germany.
Rump A
Institute for Clinical Genetics, Faculty of Medicine Carl Gustav Carus TU Dresden, Dresden, Germany.
Koenig R
Institute of Human Genetics, Johann Wolfgang Goethe University Hospital, Frankfurt/Main, Germany.
Der Kaloustian V M
Departments of Pediatrics and Human Genetics, McGill University, Montreal, PQ, Canada.
Halal F
Department of Medical Genetics, Montreal Children's Hospital, McGill University Health Centre, Montreal, PQ, Canada.
Sonntag K
Institute for Clinical Genetics, Faculty of Medicine Carl Gustav Carus TU Dresden, Dresden, Germany.
Krause C
Institute for Clinical Genetics, Faculty of Medicine Carl Gustav Carus TU Dresden, Dresden, Germany.
Hackmann K
Institute for Clinical Genetics, Faculty of Medicine Carl Gustav Carus TU Dresden, Dresden, Germany.
Hahn G
Institute of Diagnostic Radiology, Department of Pediatric Radiology, Faculty of Medicine Carl Gustav Carus TU Dresden, Dresden, Germany.
Schrock E
Institute for Clinical Genetics, Faculty of Medicine Carl Gustav Carus TU Dresden, Dresden, Germany.
Verloes A
Department of Genetics, Robert-Debré Hospital, Paris, France.
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Article Info
Journal
European journal of human genetics : EJHG
Abbr.
Eur J Hum Genet
ISSN
1476-5438
Published
2014-02-00
Epub
2013-00-12
Pages
179-83
Language
English
Region
England
NLM ID
9302235
PMCID
PMC3895648
Subset
IM
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