Literature DB >> 12571786

Microlissencephaly in microcephalic osteodysplastic primordial dwarfism: a case report and review of the literature.

L Klinge1, J Schaper, D Wieczorek, T Voit.   

Abstract

Microcephalic osteodysplastic primordial dwarfism (MOPD) is defined as a syndrome presenting with intrauterine and postnatal growth retardation, typical facial appearance, skeletal dysplasia and brain abnormalities. Autosomal-recessive inheritance is suspected. Sharing clinical manifestations, the former type III has been accepted to be the same entity as type I. We present the case of a male infant with MOPD I and micrencephaly with simplified gyral pattern to a degree defining it as microlissencephaly (MLIS). The brain abnormalities in MOPD I have not yet been classified. Reviewing the literature, we conclude that microlissencephaly appears to be the distinct developmental brain abnormality in MOPD I. Conversely, osteodysplastic changes have to be taken into consideration in the differential diagnosis of microlissencephaly. In addition, our patient suffered from acute lymphatic leukaemia which has not previously been described in association with MOPD I.

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Year:  2002        PMID: 12571786     DOI: 10.1055/s-2002-37086

Source DB:  PubMed          Journal:  Neuropediatrics        ISSN: 0174-304X            Impact factor:   1.947


  3 in total

1.  Microcephalic osteodysplastic primordial dwarfism type I with biallelic mutations in the RNU4ATAC gene.

Authors:  R Nagy; H Wang; B Albrecht; D Wieczorek; G Gillessen-Kaesbach; E Haan; P Meinecke; A de la Chapelle; J A Westman
Journal:  Clin Genet       Date:  2011-08-28       Impact factor: 4.438

2.  Lissencephaly: Expanded imaging and clinical classification.

Authors:  Nataliya Di Donato; Sara Chiari; Ghayda M Mirzaa; Kimberly Aldinger; Elena Parrini; Carissa Olds; A James Barkovich; Renzo Guerrini; William B Dobyns
Journal:  Am J Med Genet A       Date:  2017-04-25       Impact factor: 2.802

3.  A homozygous mutation in the stem II domain of RNU4ATAC causes typical Roifman syndrome.

Authors:  Yael Dinur Schejter; Adi Ovadia; Roumiana Alexandrova; Bhooma Thiruvahindrapuram; Sergio L Pereira; David E Manson; Ajoy Vincent; Daniele Merico; Chaim M Roifman
Journal:  NPJ Genom Med       Date:  2017-07-10       Impact factor: 8.617

  3 in total

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