Home LiteratureArticle Details
PMID: 26126960 Published · ppublish English Case Reports Journal Article

ALG3-CDG: Report of two siblings with antenatal features carrying homozygous p.Gly96Arg mutation.

American journal of medical genetics. Part A ·Vol. 167A ·No. 11 ·2015-11-00 ·Pages 2748-54

Lepais L, Cheillan D, Frachon SC, Hays S, Matthijs G, Panagiotakaki E, Abel C, Edery P, Rossi M

Abstract

Congenital disorders of glycosylation (CDG) are a group of inborn errors of metabolism presenting with heterogeneous multisystemic clinical manifestations. To date, more than 60 different types of CDG have been reported. ALG3-CDG is very rare, with only nine patients described so far. We report two affected siblings presenting prenatally with skeletal abnormalities associated with dysmorphic features, cerebellar vermis hypoplasia, corpus callosum agenesis, hepatic fibrosis and poor prognosis. This is the first detailed report of an affected fetus including clinical, radiographic and pathological findings. The patients showed some clinical features previously unreported in ALG3-CDG, such as bone dysplasia, cataract, corneal opacities, and pons hypoplasia. Both patients were homozygous for the previously unreported p.Gly96Arg mutation of the ALG3 gene. One patient showed chondrodysplasia punctata (CDP), which has not been previously reported in CDG. An exhaustive genetic and metabolic assessment, performed in order to rule out other possible causes of CDP, showed abnormally raised levels of anti-nuclear antibodies in the mother who, nevertheless, did not show any clinical sign of autoimmune disease during a 7 years follow-up. We speculate that the observed CDP may be explained by the maternal anti-nuclear antibodies; alternatively, a possible link to the underlying metabolic disorder cannot be ruled out. In conclusion, we report the clinical, pathological, biochemical and molecular characterization of two further patients affected by ALG3-CDG, expanding the phenotypic spectrum of this very rare disease.

Keywords
ALG3 CDG EEG chondrodysplasia punctata glycosylation prenatal
MeSH Terms
Amino Acid Substitution/genetics Blotting, Western Brain/abnormalities Congenital Disorders of Glycosylation/diagnostic imaging,genetics Fatal Outcome Female Homozygote Humans Infant, Newborn Male Mutation/genetics Pregnancy Radiography Siblings Transferrin/metabolism
Chemicals
Transferrin
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Lepais Laureline
Centre de Référence des Anomalies du Développement, Service de Génétique, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
Cheillan David
Service des Maladies Héréditaires du Métabolisme et Dépistage Néonatal, Centre de Biologie et de Pathologie Est, Hospices Civils de Lyon, Bron, France. | INSERM U1060/Université Lyon-1, Lyon, France.
Frachon Sophie Collardeau
Service d'Anatomie Pathologique, Centre de Biologie et de Pathologie Est, Hospices Civils de Lyon, Bron, France. | Université Lyon 1, Lyon, France.
Hays Stéphane
Service de Réanimation Néonatale et Néonatologie, Hôpital de la Croix Rousse, Hospices Civils de Lyon, Lyon, France.
Matthijs Gert
Center for Human Genetics, UZ Gasthuisberg, Leuven, Belgium.
Panagiotakaki Eleni
Service Epilepsie, Sommeil, Explorations Fonctionnelles Neuropédiatriques (ESEFNP), Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
Abel Carine
Centre de Référence des Anomalies du Développement, Service de Génétique, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
Edery Patrick
Centre de Référence des Anomalies du Développement, Service de Génétique, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France. | Université Lyon 1, Lyon, France. | INSERM U1028, CNRS UMR5292, CRNL TIGER Team, Bron, France.
Rossi Massimiliano
Centre de Référence des Anomalies du Développement, Service de Génétique, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France. | INSERM U1028, CNRS UMR5292, CRNL TIGER Team, Bron, France.
Article Info
Journal
American journal of medical genetics. Part A
Abbr.
Am J Med Genet A
ISSN
1552-4833
Published
2015-11-00
Epub
2015-00-30
Pages
2748-54
Language
English
Region
United States
NLM ID
101235741
Subset
IM
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: product@genelibs.com