Literature DB >> 9279757

Counselling dilemmas associated with the molecular characterisation of two Angelman syndrome families.

H L Gilbert1, J L Buxton, C T Chan, T McKay, S Cottrell, S Ramsden, R M Winter, M E Pembrey, S Malcolm.   

Abstract

We report the molecular characterisation of two families with Angelman syndrome referred for prenatal diagnosis, in which atypical molecular findings resulted in counselling dilemmas. The first is a familial case of Angelman syndrome in which the two affected children have mutations which affect the imprinting mechanism, as shown by the presence of paternal DNA methylation patterns at D15S63 and SNRPN and biparental inheritance of 15q11-q13 markers. DNA prepared from a 21 week fetal blood sample detected a fetus with normal maternal and paternal DNA methylation patterns at D15S63, but inheritance of the same maternal chromosome 15q11-q13 as the two affected sibs. This is probably a result of germline mosaicism in the mother. The second is a case of Angelman syndrome with an atypical deletion of 15q11-q13, which involves both unusual proximal and distal breakpoints. The deletion was characterised in order to assess the risk of Angelman syndrome in a second pregnancy in the mother of this child.

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Year:  1997        PMID: 9279757      PMCID: PMC1051027          DOI: 10.1136/jmg.34.8.651

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  36 in total

1.  Report of the first international workshop on human chromosome 15 mapping.

Authors:  T A Donlon
Journal:  Cytogenet Cell Genet       Date:  1992

2.  Genetic imprinting suggested by maternal heterodisomy in nondeletion Prader-Willi syndrome.

Authors:  R D Nicholls; J H Knoll; M G Butler; S Karam; M Lalande
Journal:  Nature       Date:  1989-11-16       Impact factor: 49.962

3.  Angelman syndrome.

Authors:  J Clayton-Smith; M E Pembrey
Journal:  J Med Genet       Date:  1992-06       Impact factor: 6.318

4.  The frequency of uniparental disomy in Prader-Willi syndrome. Implications for molecular diagnosis.

Authors:  M J Mascari; W Gottlieb; P K Rogan; M G Butler; D A Waller; J A Armour; A J Jeffreys; R L Ladda; R D Nicholls
Journal:  N Engl J Med       Date:  1992-06-11       Impact factor: 91.245

5.  Uniparental paternal disomy in Angelman's syndrome.

Authors:  S Malcolm; J Clayton-Smith; M Nichols; S Robb; T Webb; J A Armour; A J Jeffreys; M E Pembrey
Journal:  Lancet       Date:  1991-03-23       Impact factor: 79.321

6.  Analysis of parent of origin specific DNA methylation at SNRPN and PW71 in tissues: implication for prenatal diagnosis.

Authors:  T Kubota; S Aradhya; M Macha; A C Smith; L C Surh; J Satish; M S Verp; H L Nee; A Johnson; S L Christan; D H Ledbetter
Journal:  J Med Genet       Date:  1996-12       Impact factor: 6.318

7.  Dinucleotide repeat polymorphism of D15S10 in the Prader-Willi chromosome region (PWCR).

Authors:  R Lindeman; S Kouts; T Woodage; A Smith; R J Trent
Journal:  Nucleic Acids Res       Date:  1991-10-11       Impact factor: 16.971

8.  Chromosome 15 uniparental disomy is not frequent in Angelman syndrome.

Authors:  J H Knoll; K A Glatt; R D Nicholls; S Malcolm; M Lalande
Journal:  Am J Hum Genet       Date:  1991-01       Impact factor: 11.025

9.  A DNA methylation imprint, determined by the sex of the parent, distinguishes the Angelman and Prader-Willi syndromes.

Authors:  D J Driscoll; M F Waters; C A Williams; R T Zori; C C Glenn; K M Avidano; R D Nicholls
Journal:  Genomics       Date:  1992-08       Impact factor: 5.736

10.  Further evidence for dominant inheritance at the chromosome 15q11-13 locus in familial Angelman syndrome.

Authors:  J Clayton-Smith; T Webb; S A Robb; I Dijkstra; P Willems; S Lam; X J Cheng; M E Pembrey; S Malcolm
Journal:  Am J Med Genet       Date:  1992-09-15
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