Literature DB >> 29307700

Spinal motor neuron involvement in a patient with homozygous PRUNE mutation.

Michele Iacomino1, Chiara Fiorillo2, Annalaura Torella3, Mariasavina Severino4, Paolo Broda5, Catia Romano6, Raffaele Falsaperla6, Giulia Pozzolini7, Carlo Minetti5, Pasquale Striano5, Vincenzo Nigro3, Federico Zara7.   

Abstract

In the last few years, whole exome sequencing (WES) allowed the identification of PRUNE mutations in patients featuring a complex neurological phenotype characterized by severe neurodevelopmental delay, microcephaly, epilepsy, optic atrophy, and brain or cerebellar atrophy. We describe an additional patient with homozygous PRUNE mutation who presented with spinal muscular atrophy phenotype, in addition to the already known brain developmental disorder. This novel feature expands the clinical consequences of PRUNE mutations and allow to converge PRUNE syndrome with previous descriptions of neurodevelopmental/neurodegenerative disorders linked to altered microtubule dynamics.
Copyright © 2017 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.

Entities:  

Keywords:  Brain development; Exome sequencing; Motor neuron; Muscle biopsy; PRUNE

Mesh:

Substances:

Year:  2017        PMID: 29307700     DOI: 10.1016/j.ejpn.2017.12.005

Source DB:  PubMed          Journal:  Eur J Paediatr Neurol        ISSN: 1090-3798            Impact factor:   3.140


  2 in total

1.  Neurodevelopmental disorder with microcephaly, hypotonia, and variable brain anomalies in a consanguineous Iranian family is associated with a homozygous start loss variant in the PRUNE1 gene.

Authors:  Mehdi Agha Gholizadeh; Mina Mohammadi-Sarband; Fatemeh Fardanesh; Masoud Garshasbi
Journal:  BMC Med Genomics       Date:  2022-04-04       Impact factor: 3.063

2.  NMIHBA results from hypomorphic PRUNE1 variants that lack short-chain exopolyphosphatase activity.

Authors:  Harikiran Nistala; John Dronzek; Claudia Gonzaga-Jauregui; Shek Man Chim; Saathyaki Rajamani; Samer Nuwayhid; Dennis Delgado; Elizabeth Burke; Ender Karaca; Matthew C Franklin; Prasad Sarangapani; Michael Podgorski; Yajun Tang; Melissa G Dominguez; Marjorie Withers; Ron A Deckelbaum; Christopher J Scheonherr; William A Gahl; May C Malicdan; Brian Zambrowicz; Nicholas W Gale; Richard A Gibbs; Wendy K Chung; James R Lupski; Aris N Economides
Journal:  Hum Mol Genet       Date:  2021-01-06       Impact factor: 6.150

  2 in total

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