Literature DB >> 33186761

Novel neuroclinical findings of autosomal recessive primary microcephaly 15 in a consanguineous Iranian family.

Ehsan Razmara1, Homeyra Azimi2, Ali Reza Tavasoli3, Elnaz Fallahi4, Sadaf Valeh Sheida5, Milad Eidi1, Amirreza Bitaraf5, Zahra Farjami6, Mohammad Ali Daneshmand7, Masoud Garshasbi8.   

Abstract

Major facilitator superfamily domain-containing 2A (MFSD2A) is required for brain uptake of Docosahexaenoic acid and Lysophosphatidylcholine, both are essential for the normal neural development and function. Mutations in MFSD2A dysregulate the activity of this transporter in brain endothelial cells and can lead to microcephaly. In this study, we describe an 11-year-old male who is affected by autosomal recessive primary microcephaly 15. This patient also shows severe intellectual disability, recurrent respiratory and renal infections, low birth weight, and developmental delay. After doing clinical and neuroimaging evaluations, due to heterogeneity of neurogenetic disorders, no narrow clinical diagnosis was possible, therefore, we utilized targeted-exome sequencing to identify any causative genetic factors. This revealed a homozygous in-frame deletion (NM_001136493.1: c.241_243del; p.(Val81del)) in the MFSD2A gene as the most likely disease-susceptibility variant which was confirmed by Sanger sequencing. Neuroimaging revealed lateral ventricular asymmetry, corpus callosum hypoplasia, type B of cisterna magna, and widening of Sylvian fissures. All of these novel phenotypes are associated with autosomal recessive primary microcephaly-15 (MCPH15). According to the genotype-phenotype data, p.(Val81del) can be considered a likely pathogenic variant leading to non-lethal microcephaly. However, further cumulative data and molecular approaches are required to accurately identify genotype-phenotype correlations in MFSD2A.
Copyright © 2020 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Autosomal recessive primary microcephaly 15; In-frame variant; Intellectual disability; MFSD2A; Whole-exome sequencing

Mesh:

Substances:

Year:  2020        PMID: 33186761     DOI: 10.1016/j.ejmg.2020.104096

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  4 in total

1.  Symptomatic care of late-onset Alexander disease presenting with area postrema-like syndrome with prednisolone; a case report.

Authors:  Safoura Zardadi; Ehsan Razmara; Maryam Rasoulinezhad; Meisam Babaei; Mohammad Reza Ashrafi; Neda Pak; Masoud Garshasbi; Ali Reza Tavasoli
Journal:  BMC Pediatr       Date:  2022-07-13       Impact factor: 2.567

2.  Novel manifestations of Warburg micro syndrome type 1 caused by a new splicing variant of RAB3GAP1: a case report.

Authors:  Raziyeh Khalesi; Ehsan Razmara; Golareh Asgaritarghi; Ali Reza Tavasoli; Yasser Riazalhosseini; Daniel Auld; Masoud Garshasbi
Journal:  BMC Neurol       Date:  2021-04-28       Impact factor: 2.903

Review 3.  Lipid Transport and Metabolism at the Blood-Brain Interface: Implications in Health and Disease.

Authors:  Fabien Pifferi; Benoit Laurent; Mélanie Plourde
Journal:  Front Physiol       Date:  2021-03-30       Impact factor: 4.566

4.  Structural basis of omega-3 fatty acid transport across the blood-brain barrier.

Authors:  Rosemary J Cater; Geok Lin Chua; Satchal K Erramilli; James E Keener; Brendon C Choy; Piotr Tokarz; Cheen Fei Chin; Debra Q Y Quek; Brian Kloss; Joseph G Pepe; Giacomo Parisi; Bernice H Wong; Oliver B Clarke; Michael T Marty; Anthony A Kossiakoff; George Khelashvili; David L Silver; Filippo Mancia
Journal:  Nature       Date:  2021-06-16       Impact factor: 49.962

  4 in total

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