Literature DB >> 8912329

Congenital pontocerebellar atrophy in three patients: clinical, radiologic and etiologic considerations.

N Zelnik1, W B Dobyns, S L Forem, E H Kolodny.   

Abstract

We report three patients with severe pontocerebellar atrophy (PCA) associated with a variable degree of cerebral atrophy. The clinical features consisted of progressive microcephaly, central hypotonia, visual impairment, abnormal eye movements and delayed psychomotor development. These are similar but not identical to the features of pontocerebellar hypoplasia type 2 described by Barth. The picture also differs from the classical form of autosomal dominant olivopontocerebellar atrophy. While in two patients the disease seemed to be genetic with highly suspicious autosomal recessive inheritance, the etiology in the third patient was probably nongenetic. We suggest that PCA is a morphologic entity with distinct radiologic features but variable clinical, pathophysiologic and etiologic backgrounds.

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Year:  1996        PMID: 8912329     DOI: 10.1007/s002340050334

Source DB:  PubMed          Journal:  Neuroradiology        ISSN: 0028-3940            Impact factor:   2.804


  5 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.  Disruption of cerebellar development: potential complication of extreme prematurity.

Authors:  Agnes Messerschmidt; Peter C Brugger; Eugen Boltshauser; Gerlinde Zoder; Walter Sterniste; Robert Birnbacher; Daniela Prayer
Journal:  AJNR Am J Neuroradiol       Date:  2005-08       Impact factor: 3.825

Review 3.  MRI of the fetal posterior fossa.

Authors:  Catherine Adamsbaum; Marie Laure Moutard; Christine André; Valérie Merzoug; Solène Ferey; Marie Pierre Quéré; Fanny Lewin; Catherine Fallet-Bianco
Journal:  Pediatr Radiol       Date:  2004-11-23

4.  Molecular and neuroimaging findings in pontocerebellar hypoplasia type 2 (PCH2): is prenatal diagnosis possible?

Authors:  John M Graham; Andrew H Spencer; Inessa Grinberg; Charles E Niesen; Lawrence D Platt; Marcel Maya; Yasmin Namavar; Frank Baas; William B Dobyns
Journal:  Am J Med Genet A       Date:  2010-09       Impact factor: 2.802

5.  Further delineation of pontocerebellar hypoplasia type 6 due to mutations in the gene encoding mitochondrial arginyl-tRNA synthetase, RARS2.

Authors:  Emma Glamuzina; Ruth Brown; Kieran Hogarth; Dawn Saunders; Isabelle Russell-Eggitt; Matthew Pitt; Carlos de Sousa; Shamima Rahman; Garry Brown; Stephanie Grunewald
Journal:  J Inherit Metab Dis       Date:  2011-11-16       Impact factor: 4.982

  5 in total

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