Literature DB >> 484596

The Bowen-Conradi syndrome -- a highly lethal autosomal recessive syndrome of microcephaly, micrognathia, low birth weight, and joint deformities.

A G Hunter, S J Woerner, L D Montalvo-Hicks, S B Fowlow, R H Haslam, P J Metcalf, R B Lowry.   

Abstract

This paper describes six Hutterite children from five families who appear to have been affected by the same syndrome that was described in two brothers by Bowen and Conradi [1]. Our additional cases confirm that the major features of the syndrome include porportionate intrauterine growth retardation, microcephaly, micrognathia, a prominent nose, rocker-bottom feet, joint limitation, and failure to thrive, with death within the first year of life. Bowen-Conradi syndrome is an autosomal recessive trait and pedigree records show that all six families now known are related to each other through two couples born in the late 1700s but that there are additional earlier possible sources of the responsible gene. The differential diagnosis of this syndrome is discussed.

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Year:  1979        PMID: 484596     DOI: 10.1002/ajmg.1320030305

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  7 in total

1.  Lethal olivopontoneocerebellar hypoplasia with dysmorphic features in sibs.

Authors:  I D Young; P A McKeever; M V Squier; J Grant
Journal:  J Med Genet       Date:  1992-10       Impact factor: 6.318

2.  The syndrome of multiple ankyloses and facial anomalies. A neuropathologic analysis.

Authors:  R S Williams; L B Holmes
Journal:  Acta Neuropathol       Date:  1980       Impact factor: 17.088

3.  Antenatal ultrasonography findings and magnetic resonance imaging in a case of Pena-Shokeir phenotype.

Authors:  Xuan-Hong Tomai; Thanh-Xuan Jasmine; Thanh-Hai Phan
Journal:  Ultrasound       Date:  2017-01-10

Review 4.  Human diseases of the SSU processome.

Authors:  Samuel B Sondalle; Susan J Baserga
Journal:  Biochim Biophys Acta       Date:  2013-11-12

5.  Structural insight into the functional mechanism of Nep1/Emg1 N1-specific pseudouridine methyltransferase in ribosome biogenesis.

Authors:  Seth R Thomas; Christopher A Keller; Agnieszka Szyk; Joe R Cannon; Nicole A Laronde-Leblanc
Journal:  Nucleic Acids Res       Date:  2010-11-17       Impact factor: 16.971

6.  Effects of the Bowen-Conradi syndrome mutation in EMG1 on its nuclear import, stability and nucleolar recruitment.

Authors:  Ahmed S Warda; Bernard Freytag; Sara Haag; Katherine E Sloan; Dirk Görlich; Markus T Bohnsack
Journal:  Hum Mol Genet       Date:  2016-12-15       Impact factor: 6.150

7.  Mutation of EMG1 causing Bowen-Conradi syndrome results in reduced cell proliferation rates concomitant with G2/M arrest and 18S rRNA processing delay.

Authors:  Joy Armistead; Richard Hemming; Nehal Patel; Barbara Triggs-Raine
Journal:  BBA Clin       Date:  2014-05-29
  7 in total

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