Home LiteratureArticle Details
PMID: 6876108 Published · ppublish English Journal Article

Fragile X chromosome: clinical and cytogenetic studies on cases from seven families.

Journal of medical genetics ·Vol. 20 ·No. 3 ·1983-06-00 ·Pages 169-78

McDermott A, Walters R, Howell RT, Gardner A

Abstract

Results of detailed clinical and cytogenetic studies on 13 mentally retarded males and two heterozygous females (one normal and one retarded) are reported. Reference is made to technical modifications to enhance the incidence of expression of the fragile X. The addition of excess methionine to the fibroblast cultures (final concentration of 115 mg/l medium TC 199) was found to be particularly valuable, increasing the incidence of expression up to four-fold, and enabling the demonstration of the fragile X in fibroblasts when it could not be demonstrated in blood cultures in at least one case. Studies on replication patterns of the X chromosomes in the two heterozygous females showed that the fragile X chromosome was genetically active in a significantly greater proportion of cells (74%) in the mentally retarded female, whereas the normal X was active in a similar proportion (72%) in the carrier with normal intelligence.

MeSH Terms
Adult Cells, Cultured DNA Replication Female Fragile X Syndrome/genetics Heterozygote Humans Intellectual Disability/genetics Karyotyping Male Middle Aged Pedigree Sex Chromosome Aberrations/genetics
Authors & Affiliations
4 authors, click to expand affiliations / ORCID
McDermott A
Walters R
Howell R T
Gardner A
References (27)
27 references, click to expand
  1. "Rubinstein-Taybi's syndrome".
    J Ment Defic Res. 1965 Dec;9(4):265-70 PMID: 5866882
  2. A marker X chromosome.
    Am J Hum Genet. 1969 May;21(3):231-44 PMID: 5794013
  3. X-linked mental retardation.
    J Med Genet. 1974 Jun;11(2):109-13 PMID: 4841078
  4. X-linked mental retardation associated with macro-orchidism.
    J Med Genet. 1975 Dec;12(4):367-71 PMID: 1240971
  5. Inherited congenital normofunctional testicular hyperplasia and mental deficiency.
    Hum Genet. 1976 Jul 7;33(1):23-33 PMID: 939556
  6. Familial X-linked mental retardation with an X chromosome abnormality.
    J Med Genet. 1977 Feb;14(1):46-50 PMID: 839500
  7. Fragile sites on human chromosomes: demonstration of their dependence on the type of tissue culture medium.
    Science. 1977 Jul 15;197(4300):265-6 PMID: 877551
  8. Expression in fibroblast culture of the satellited-X chromosome associated with familial sex-linked mental retardation.
    Hum Genet. 1980 Feb;53(2):267-9 PMID: 6928413
  9. X-linked mental retardation, macro-orchidism, and the Xq27 fragile site.
    J Pediatr. 1980 May;96(5):837-41 PMID: 7189210
  10. Familial X-linked mental retardation and fragile X chromosomes in two Swedish families.
    Clin Genet. 1981 Feb;19(2):101-10 PMID: 7193540
  11. Fragile X in a normal male: a cautionary tale.
    Lancet. 1981 Apr 4;1(8223):780 PMID: 6110980
  12. Significance of phenotypic and chromosomal abnormalities in X-linked mental retardation (Martin-Bell or Renpenning syndrome).
    Am J Med Genet. 1980;7(4):417-32 PMID: 6938131
  13. X-linked mental retardation with macro-orchidism and marker-X chromosomes.
    Am J Med Genet. 1980;7(4):433-41 PMID: 6938132
  14. Nonspecific X-linked mental retardation II: the frequency in British Columbia.
    Am J Med Genet. 1980;7(4):461-9 PMID: 7211956
  15. X-linked mental retardation: a study of 7 families.
    Am J Med Genet. 1980;7(4):471-89 PMID: 7211957
  16. X-linked mental retardation: Renpenning revisited.
    Am J Med Genet. 1980;7(4):491-5 PMID: 7211958
  17. Fragile sites in human chromosomes II: demonstration of the fragile site Xq27 in carriers of X-linked mental retardation.
    Am J Med Genet. 1980;7(4):497-501 PMID: 6938133
  18. X-linked mental retardation.
    Arch Dis Child. 1981 Jun;56(6):410-1 PMID: 7020610
  19. Transmission of fragile (X) (q27) site from a male.
    Lancet. 1981 Nov 28;2(8257):1231-2 PMID: 6118659
  20. Toxic-shock syndrome not associated with menstruation. A review of 54 cases.
    Lancet. 1982 Jan 2;1(8262):1-4 PMID: 6119410
  21. Association of fragile X syndrome with autism.
    Lancet. 1982 Jan 9;1(8263):100 PMID: 6119460
  22. The 'fragile' X chromosome in the Martin-Bell-Renpenning syndrome and in males with other forms of familial mental retardation.
    J Med Genet. 1981 Oct;18(5):366-73 PMID: 7328617
  23. Fragile X-linked mental retardation: the Martin-Bell syndrome.
    J Ment Defic Res. 1981 Dec;25 Pt 4:253-6 PMID: 7328634
  24. Is the fragile X syndrome amenable to treatment?
    Lancet. 1982 Jan 30;1(8266):273-4 PMID: 6120287
  25. Measurement of testicular volume. Its application to assessment of maturation, and its use in diagnosis of hypogonadism.
    Arch Dis Child. 1962 Oct;37:514-7 PMID: 13975542
  26. Familial sex-linked mental retardation.
    Can Med Assoc J. 1962 Nov 3;87:954-6 PMID: 13981686
  27. X-linked mental retardation.
    Am J Med Genet. 1980;7(4):407-15 PMID: 7011031
Article Info
Journal
Journal of medical genetics
Abbr.
J Med Genet
ISSN
0022-2593
Published
1983-06-00
Pages
169-78
Language
English
Region
England
NLM ID
2985087R
PMCID
PMC1049041
Subset
IM
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: [email protected]