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PMID: 12058345 Published · ppublish English Journal Article

Presence of large deletions in kindreds with autism.

American journal of human genetics ·Vol. 71 ·No. 1 ·2002-07-00 ·Pages 100-15

Yu CE, Dawson G, Munson J, D'Souza I, Osterling J, Estes A, Leutenegger AL, Flodman P, Smith M, Raskind WH, Spence MA, McMahon W, Wijsman EM, Schellenberg GD

Abstract

Autism is caused, in part, by inheritance of multiple interacting susceptibility alleles. To identify these inherited factors, linkage analysis of multiplex families is being performed on a sample of 105 families with two or more affected sibs. Segregation patterns of short tandem repeat polymorphic markers from four chromosomes revealed null alleles at four marker sites in 12 families that were the result of deletions ranging in size from 5 to >260 kb. In one family, a deletion at marker D7S630 was complex, with two segments deleted (37 kb and 18 kb) and two retained (2,836 bp and 38 bp). Three families had deletions at D7S517, with each family having a different deletion (96 kb, 183 kb, and >69 kb). Another three families had deletions at D8S264, again with each family having a different deletion, ranging in size from <5.9 kb to >260 kb. At a fourth marker, D8S272, a 192-kb deletion was found in five families. Unrelated subjects and additional families without autism were screened for deletions at these four sites. Families screened included 40 families from Centre d'Etude du Polymorphisme Humaine and 28 families affected with learning disabilities. Unrelated samples were 299 elderly control subjects, 121 younger control subjects, and 248 subjects with Alzheimer disease. The deletion allele at D8S272 was found in all populations screened. For the other three sites, no additional deletions were identified in any of the groups without autism. Thus, these deletions appear to be specific to autism kindreds and are potential autism-susceptibility alleles. An alternative hypothesis is that autism-susceptibility alleles elsewhere cause the deletions detected here, possibly by inducing errors during meiosis.

MeSH Terms
Adult Aged Alleles Autistic Disorder/genetics Base Sequence Case-Control Studies Child Child, Preschool Chromosome Mapping DNA/genetics Female Genetic Markers Humans Male Pedigree Sequence Deletion Tandem Repeat Sequences
Chemicals
Genetic Markers DNA
Authors & Affiliations
14 authors, click to expand affiliations / ORCID
Yu Chang-En
Geriatrics Research Education and Clinical Center, Puget Sound Veterans Affairs Medical Center, University of Washington, Seattle 98108, USA.
Dawson Geraldine
Munson Jeffrey
D'Souza Ian
Osterling Julie
Estes Annette
Leutenegger Anne-Louise
Flodman Pamela
Smith Moyra
Raskind Wendy H
Spence M Anne
McMahon William
Wijsman Ellen M
Schellenberg Gerard D
References (53)
53 references, click to expand
  1. Case reports of autism with interstitial deletion of chromosome 17 (p11.2 p11.2) and monosomy of chromosome 5 (5pter-->5p15.3).
    Psychiatr Genet. 1994 Summer;4(2):109-11 PMID: 8055249
  2. Genomic disorders: structural features of the genome can lead to DNA rearrangements and human disease traits.
    Trends Genet. 1998 Oct;14(10):417-22 PMID: 9820031
  3. Brief report: duplication of chromosome 15q11-13 in two individuals with autistic disorder.
    J Autism Dev Disord. 1994 Aug;24(4):529-35 PMID: 7961335
  4. A case-control family history study of autism.
    J Child Psychol Psychiatry. 1994 Jul;35(5):877-900 PMID: 7962246
  5. Autism Diagnostic Interview-Revised: a revised version of a diagnostic interview for caregivers of individuals with possible pervasive developmental disorders.
    J Autism Dev Disord. 1994 Oct;24(5):659-85 PMID: 7814313
  6. Autism as a strongly genetic disorder: evidence from a British twin study.
    Psychol Med. 1995 Jan;25(1):63-77 PMID: 7792363
  7. The PML gene is linked to a megabase-scale insertion/deletion restriction fragment length polymorphism.
    Genomics. 1995 Mar 20;26(2):327-33 PMID: 7601459
  8. Genetic studies of autistic disorder and chromosome 7.
    Genomics. 1999 Nov 1;61(3):227-36 PMID: 10552924
  9. An autosomal genomic screen for autism. Collaborative linkage study of autism.
    Am J Med Genet. 1999 Dec 15;88(6):609-15 PMID: 10581478
  10. Identification of a novel gene on chromosome 7q31 that is interrupted by a translocation breakpoint in an autistic individual.
    Am J Hum Genet. 2000 Aug;67(2):510-4 PMID: 10889047
  11. Identification and analysis of error types in high-throughput genotyping.
    Am J Hum Genet. 2000 Sep;67(3):727-36 PMID: 10924406
  12. Genetic studies in autistic disorder and chromosome 15.
    Neurogenetics. 2000 Mar;2(4):219-26 PMID: 10983717
  13. Discovery of allelic variants of HOXA1 and HOXB1: genetic susceptibility to autism spectrum disorders.
    Teratology. 2000 Dec;62(6):393-405 PMID: 11091361
  14. Childhood-onset schizophrenia/autistic disorder and t(1;7) reciprocal translocation: identification of a BAC contig spanning the translocation breakpoint at 7q21.
    Am J Med Genet. 2000 Dec 4;96(6):749-53 PMID: 11121174
  15. Analysis of a 1-megabase deletion in 15q22-q23 in an autistic patient: identification of candidate genes for autism and of homologous DNA segments in 15q22-q23 and 15q11-q13.
    Am J Med Genet. 2000 Dec 4;96(6):765-70 PMID: 11121177
  16. The 28-kb deletion spanning D15S63 is a polymorphic variant in the Ashkenazi Jewish population.
    Am J Hum Genet. 2001 Jan;68(1):261-3 PMID: 11083946
  17. Cloning and characterization of a Golgin-related gene from the large-scale polymorphism linked to the PML gene.
    Genomics. 2000 Dec 15;70(3):364-74 PMID: 11161787
  18. Reelin gene alleles and haplotypes as a factor predisposing to autistic disorder.
    Mol Psychiatry. 2001 Mar;6(2):150-9 PMID: 11317216
  19. Creation of genome-wide protein expression libraries using random activation of gene expression.
    Nat Biotechnol. 2001 May;19(5):440-5 PMID: 11329013
  20. Further characterization of the autism susceptibility locus AUTS1 on chromosome 7q.
    Hum Mol Genet. 2001 Apr 15;10(9):973-82 PMID: 11392322
  21. A study of novel polymorphisms in the upstream region of vasoactive intestinal peptide receptor type 2 gene in autism.
    J Child Neurol. 2001 May;16(5):357-63 PMID: 11392521
  22. Are rare variants responsible for susceptibility to complex diseases?
    Am J Hum Genet. 2001 Jul;69(1):124-37 PMID: 11404818
  23. Evidence for a susceptibility gene for autism on chromosome 2 and for genetic heterogeneity.
    Am J Hum Genet. 2001 Jun;68(6):1514-20 PMID: 11353400
  24. Evidence supporting WNT2 as an autism susceptibility gene.
    Am J Med Genet. 2001 Jul 8;105(5):406-13 PMID: 11449391
  25. A genomewide screen for autism susceptibility loci.
    Am J Hum Genet. 2001 Aug;69(2):327-40 PMID: 11452361
  26. A genomewide screen for autism: strong evidence for linkage to chromosomes 2q, 7q, and 16p.
    Am J Hum Genet. 2001 Sep;69(3):570-81 PMID: 11481586
  27. A polymorphic genomic duplication on human chromosome 15 is a susceptibility factor for panic and phobic disorders.
    Cell. 2001 Aug 10;106(3):367-79 PMID: 11509185
  28. Assessment of the total number of human transcription units.
    Genomics. 2001 Sep;77(1-2):71-8 PMID: 11543635
  29. Linkage disequilibrium at the Angelman syndrome gene UBE3A in autism families.
    Genomics. 2001 Sep;77(1-2):105-13 PMID: 11543639
  30. Molecular genetic delineation of 2q37.3 deletion in autism and osteodystrophy: report of a case and of new markers for deletion screening by PCR.
    Cytogenet Cell Genet. 2001;94(1-2):15-22 PMID: 11701947
  31. The DNA sequence and comparative analysis of human chromosome 20.
    Nature. 2001 Dec 20-27;414(6866):865-71 PMID: 11780052
  32. Identification of a novel gene on chromosome 7q11.2 interrupted by a translocation breakpoint in a pair of autistic twins.
    Genomics. 2002 Aug;80(2):129-34 PMID: 12160723
  33. Genetic influences and infantile autism.
    Nature. 1977 Feb 24;265(5596):726-8 PMID: 558516
  34. Infantile autism: a genetic study of 21 twin pairs.
    J Child Psychol Psychiatry. 1977 Sep;18(4):297-321 PMID: 562353
  35. Concordance for the syndrome of autism in 40 pairs of afflicted twins.
    Am J Psychiatry. 1985 Jan;142(1):74-7 PMID: 4038442
  36. Partial 6p trisomy associated with infantile autism.
    Clin Genet. 1988 May;33(5):356-9 PMID: 3378366
  37. A twin study of autism in Denmark, Finland, Iceland, Norway and Sweden.
    J Child Psychol Psychiatry. 1989 May;30(3):405-16 PMID: 2745591
  38. Autism associated with marker chromosome.
    J Am Acad Child Adolesc Psychiatry. 1991 May;30(3):489-94 PMID: 2055888
  39. Performing the exact test of Hardy-Weinberg proportion for multiple alleles.
    Biometrics. 1992 Jun;48(2):361-72 PMID: 1637966
  40. Mutation of human short tandem repeats.
    Hum Mol Genet. 1993 Aug;2(8):1123-8 PMID: 8401493
  41. Association study with two markers of a human homeogene in infantile autism.
    J Med Genet. 1995 Apr;32(4):269-74 PMID: 7643354
  42. Latent-class analysis of recurrence risks for complex phenotypes with selection and measurement error: a twin and family history study of autism.
    Am J Hum Genet. 1995 Sep;57(3):717-26 PMID: 7668301
  43. A broader phenotype of autism: the clinical spectrum in twins.
    J Child Psychol Psychiatry. 1996 Oct;37(7):785-801 PMID: 8923222
  44. Autism or atypical autism in maternally but not paternally derived proximal 15q duplication.
    Am J Hum Genet. 1997 Apr;60(4):928-34 PMID: 9106540
  45. Evidence of linkage between the serotonin transporter and autistic disorder.
    Mol Psychiatry. 1997 May;2(3):247-50 PMID: 9152989
  46. Intersitial deletion of 20p: new candidate region for Hirschsprung disease and autism?
    Am J Med Genet. 1997 Aug 22;71(3):298-304 PMID: 9268100
  47. Linkage-disequilibrium mapping of autistic disorder, with 15q11-13 markers.
    Am J Hum Genet. 1998 May;62(5):1077-83 PMID: 9545402
  48. Xp deletions associated with autism in three females.
    Hum Genet. 1999 Jan;104(1):43-8 PMID: 10071191
  49. Genome-wide scan for autism susceptibility genes. Paris Autism Research International Sibpair Study.
    Hum Mol Genet. 1999 May;8(5):805-12 PMID: 10196369
  50. A genomic screen of autism: evidence for a multilocus etiology.
    Am J Hum Genet. 1999 Aug;65(2):493-507 PMID: 10417292
  51. Deletion of chromosome 2q37 and autism: a distinct subtype?
    J Autism Dev Disord. 1999 Jun;29(3):259-63 PMID: 10425588
  52. Xeroderma pigmentosum group C splice mutation associated with autism and hypoglycinemia.
    J Invest Dermatol. 1998 Nov;111(5):791-6 PMID: 9804340
  53. Clinical and molecular analysis of five inv dup(15) patients.
    Eur J Hum Genet. 1993;1(1):37-50 PMID: 8069650
Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
2002-07-00
Epub
2002-00-07
Pages
100-15
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC384967
Subset
IM
Grants
NCRR NIH HHS · M01 RR000064 · United States
NICHD NIH HHS · P01 HD035476 · United States
NICHD NIH HHS · P50 HD033812 · United States
NICHD NIH HHS · U19 HD035476 · United States
Databases
OMIM
102578, 104300, 105830, 131310, 137192, 142955, 142968, 147870, 176270, 181500, 182138, 191100, 192430, 209850, 235730, 278720, 309550, 600514, 600833, 601970, 603509
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