Literature DB >> 11149619

A boy with a submicroscopic 22qter deletion, general overgrowth and features suggestive of FG syndrome.

B B de Vries1, M Bitner-Glindzicz, S J Knight, J Tyson, K D MacDermont, J Flint, S Malcolm, R M Winter.   

Abstract

Over recent years, submicroscopic subtelomeric rearrangements have been shown to be a significant cause of mental retardation and, therefore, such abnormalities should be considered in every child with moderate to severe retardation with additional features suggestive of a chromosomal abnormality. The FG syndrome is an X-linked recessive mental retardation syndrome with congenital hypotonia, relative macrocephaly, a characteristic facies and constipation. We describe a severely mentally retarded boy with a history of severe constipation, truncal hypotonia, facial dysmorphism, fetal pads, and joint laxity, leading to an initial diagnosis of FG syndrome at the age of 3 years. Clinical re-evaluation at the age of 6 years, when he showed signs of general overgrowth, initiated a telomere screen, and a submicroscopic 22q13.3 telomere deletion was detected. The features suggestive of FG syndrome in this boy with a 22q13.3--> qter deletion may indicate testing for submicroscopic 22qter deletions in patients with atypical features of FG syndrome without a definite X-linked family history.

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Year:  2000        PMID: 11149619     DOI: 10.1034/j.1399-0004.2000.580610.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  3 in total

Review 1.  Telomeres: a diagnosis at the end of the chromosomes.

Authors:  B B A De Vries; R Winter; A Schinzel; C van Ravenswaaij-Arts
Journal:  J Med Genet       Date:  2003-06       Impact factor: 6.318

2.  Clinical studies on submicroscopic subtelomeric rearrangements: a checklist.

Authors:  B B de Vries; S M White; S J Knight; R Regan; T Homfray; I D Young; M Super; C McKeown; M Splitt; O W Quarrell; A H Trainer; M F Niermeijer; S Malcolm; J Flint; J A Hurst; R M Winter
Journal:  J Med Genet       Date:  2001-03       Impact factor: 6.318

3.  Molecular characterisation of the 22q13 deletion syndrome supports the role of haploinsufficiency of SHANK3/PROSAP2 in the major neurological symptoms.

Authors:  H L Wilson; A C C Wong; S R Shaw; W-Y Tse; G A Stapleton; M C Phelan; S Hu; J Marshall; H E McDermid
Journal:  J Med Genet       Date:  2003-08       Impact factor: 6.318

  3 in total

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