Literature DB >> 9134193

A syndrome of autosomal recessive pontocerebellar hypoplasia with white matter abnormalities and protracted course in two brothers.

A Malandrini1, S Palmeri, M Villanova, E Parrotta, F Sicurelli, D Amato, D DeFalco, G C Guazzi.   

Abstract

We describe two brothers with an autosomal recessive syndrome characterized by neonatal onset, severe impairment of cognitive and motor functions, abnormal ocular movements and slight dystonic postures. Brain MR and CT scan showed a reduction in size of the cerebellum and to a lesser extent pons, accompanied by cerebral and cerebellar white matter abnormalities. These data suggest that they have a particular phenotype of pontocerebellar hypoplasia. Extensive laboratory investigation excluded known metabolic causes of pontocerebellar hypoplasia. We discuss the nosological status of pontocerebellar hypoplasia in relation to other early-onset pontocerebellar disorders.

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Year:  1997        PMID: 9134193     DOI: 10.1016/s0387-7604(96)00563-3

Source DB:  PubMed          Journal:  Brain Dev        ISSN: 0387-7604            Impact factor:   1.961


  2 in total

1.  Pontocerebellar hypoplasia type III (CLAM): extended phenotype and novel molecular findings.

Authors:  Burak Durmaz; Bernd Wollnik; Ozgur Cogulu; Yun Li; Hasan Tekgul; Filiz Hazan; Ferda Ozkinay
Journal:  J Neurol       Date:  2009-03-14       Impact factor: 4.849

2.  A homozygote variant in the tRNA splicing endonuclease subunit 54 causes pontocerebellar hypoplasia in a consanguineous Iranian family.

Authors:  Afrooz Sepahvand; Ehsan Razmara; Fatemeh Bitarafan; Mohammad Galehdari; Ali Reza Tavasoli; Navid Almadani; Masoud Garshasbi
Journal:  Mol Genet Genomic Med       Date:  2020-07-22       Impact factor: 2.183

  2 in total

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