Literature DB >> 9222958

A new X linked recessive syndrome of mental retardation and mild dysmorphism maps to Xq28.

G S Pai1, B Hane, M Joseph, R Nelson, L S Hammond, J F Arena, H A Lubs, R E Stevenson, C E Schwartz.   

Abstract

Efforts to understand the genetic basis of mental retardation are greatly assisted by the identification of families with multiple relatives with mental retardation that clinical geneticists encounter in the routine practice of their profession. Here we describe a linkage study of a four generation family in which X linked recessive mental retardation (XLMR) is associated with minor dysmorphism and premature death of the affected males. Microsatellite based polymorphic loci evenly spaced over the entire X chromosome were used initially to detect linkage to Xq28. Further analysis identified a haplotype of Xq28 markers bounded proximally by locus DXS1113 and distally by DXS1108 that cosegregated with XLMR in this family. Two point lod scores > 3.0 provided strong evidence that the gene locus responsible for XLMR in this family is within this 7 Mb region of Xq28. The minor anomalies noted in some affected males were not distinctive enough to suggest a unique syndrome. None of our patients had features of the Waisman-Laxova syndrome or the PPM-X syndrome. The possibility of allelism with any of the five other non-specific XLMR syndromes (MRX3, MRX16, MRX25, MRX28, and MRX41) mapped to Xq28 could not be excluded. While the recognition of a gene responsible for this disorder needs much additional work, multiple female relatives at risk in this family benefit immediately from knowing their genotype and heterozygotes will have the opportunity to undergo prenatal diagnosis.

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Year:  1997        PMID: 9222958      PMCID: PMC1050990          DOI: 10.1136/jmg.34.7.529

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


  19 in total

1.  Linkage to Xq28 in a family with nonspecific X-linked mental retardation.

Authors:  A M Nordström; M Penttinen; H von Koskull
Journal:  Hum Genet       Date:  1992-11       Impact factor: 4.132

2.  PPM-X: a new X-linked mental retardation syndrome with psychosis, pyramidal signs, and macroorchidism maps to Xq28.

Authors:  S Lindsay; M Splitt; S Edney; T P Berney; S J Knight; K E Davies; O O'Brien; M Gale; J Burn
Journal:  Am J Hum Genet       Date:  1996-06       Impact factor: 11.025

3.  Localisation of the MRX3 gene for non-specific X linked mental retardation.

Authors:  A Gedeon; B Kerr; J Mulley; G Turner
Journal:  J Med Genet       Date:  1991-06       Impact factor: 6.318

4.  A gene for nonspecific X-linked mental retardation (MRX41) is located in the distal segment of Xq28.

Authors:  B C Hamel; H Kremer; E Wesby-van Swaay; B van den Helm; A P Smits; B A Oostra; H H Ropers; E C Mariman
Journal:  Am J Med Genet       Date:  1996-07-12

5.  The clinical spectrum of mutations in L1, a neuronal cell adhesion molecule.

Authors:  E Fransen; L Vits; G Van Camp; P J Willems
Journal:  Am J Med Genet       Date:  1996-07-12

6.  +2.71 LOD score at zero recombination is not sufficient for establishing linkage between X-linked mental retardation and X-chromosome markers.

Authors:  R Robledo; P Melis; M Siniscalco; J Marchi; F Laficara; A Rinaldi; M Rocchi; G Filippi
Journal:  Am J Med Genet       Date:  1996-07-12

7.  Identification of FMR2, a novel gene associated with the FRAXE CCG repeat and CpG island.

Authors:  Y Gu; Y Shen; R A Gibbs; D L Nelson
Journal:  Nat Genet       Date:  1996-05       Impact factor: 38.330

8.  Waisman syndrome, a human X-linked recessive basal ganglia disorder with mental retardation: localization to Xq27.3-qter.

Authors:  R G Gregg; A B Metzenberg; K Hogan; G Sekhon; R Laxova
Journal:  Genomics       Date:  1991-04       Impact factor: 5.736

9.  Regional localization of two MRX genes to Xq28 (MRX28) and to Xp11.4-Xp22.12 (MRX33).

Authors:  E Holinski-Feder; A Golla; I Rost; H Seidel; O Rittinger; A Meindl
Journal:  Am J Med Genet       Date:  1996-07-12

10.  Overlapping submicroscopic deletions in Xq28 in two unrelated boys with developmental disorders: identification of a gene near FRAXE.

Authors:  A K Gedeon; M Meinänen; L C Adès; H Kääriäinen; J Gécz; E Baker; G R Sutherland; J C Mulley
Journal:  Am J Hum Genet       Date:  1995-04       Impact factor: 11.025

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  4 in total

Review 1.  Demystified ... FISH.

Authors:  J J Waters; A L Barlow; C P Gould
Journal:  Mol Pathol       Date:  1998-04

2.  Xq28 duplications including MECP2 in five females: Expanding the phenotype to severe mental retardation.

Authors:  E K Bijlsma; A Collins; F T Papa; M I Tejada; P Wheeler; E A J Peeters; A C J Gijsbers; J M van de Kamp; M Kriek; M Losekoot; A J Broekma; J A Crolla; M Pollazzon; M Mucciolo; E Katzaki; V Disciglio; M I Ferreri; A Marozza; M A Mencarelli; C Castagnini; L Dosa; F Ariani; F Mari; R Canitano; G Hayek; M P Botella; B Gener; M Mínguez; A Renieri; C A L Ruivenkamp
Journal:  Eur J Med Genet       Date:  2012-03-29       Impact factor: 2.708

3.  Clinical and molecular genetic characterization of familial MECP2 duplication syndrome in a Chinese family.

Authors:  Xiaoyan Li; Hua Xie; Qian Chen; Xiongying Yu; Zhaoshi Yi; Erzhen Li; Ting Zhang; Jian Wang; Jianmin Zhong; Xiaoli Chen
Journal:  BMC Med Genet       Date:  2017-11-15       Impact factor: 2.103

Review 4.  A brief history of MECP2 duplication syndrome: 20-years of clinical understanding.

Authors:  Daniel Ta; Jenny Downs; Gareth Baynam; Andrew Wilson; Peter Richmond; Helen Leonard
Journal:  Orphanet J Rare Dis       Date:  2022-03-21       Impact factor: 4.123

  4 in total

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