Home LiteratureArticle Details
PMID: 21937992 Published · epublish English Journal Article Research Support, Non-U.S. Gov't

Deep sequencing reveals 50 novel genes for recessive cognitive disorders.

Nature ·Vol. 478 ·No. 7367 ·2011-09-21 ·Pages 57-63

Najmabadi H, Hu H, Garshasbi M, Zemojtel T, Abedini SS, Chen W, Hosseini M, Behjati F, Haas S, Jamali P, Zecha A, Mohseni M, Püttmann L, Vahid LN, Jensen C, Moheb LA, Bienek M, Larti F, Mueller I, Weissmann R, Darvish H, Wrogemann K, Hadavi V, Lipkowitz B, Esmaeeli-Nieh S, Wieczorek D, Kariminejad R, Firouzabadi SG, Cohen M, Fattahi Z, Rost I, Mojahedi F, Hertzberg C, Dehghan A, Rajab A, Banavandi MJ, Hoffer J, Falah M, Musante L, Kalscheuer V, Ullmann R, Kuss AW, Tzschach A, Kahrizi K, Ropers HH

Abstract

Common diseases are often complex because they are genetically heterogeneous, with many different genetic defects giving rise to clinically indistinguishable phenotypes. This has been amply documented for early-onset cognitive impairment, or intellectual disability, one of the most complex disorders known and a very important health care problem worldwide. More than 90 different gene defects have been identified for X-chromosome-linked intellectual disability alone, but research into the more frequent autosomal forms of intellectual disability is still in its infancy. To expedite the molecular elucidation of autosomal-recessive intellectual disability, we have now performed homozygosity mapping, exon enrichment and next-generation sequencing in 136 consanguineous families with autosomal-recessive intellectual disability from Iran and elsewhere. This study, the largest published so far, has revealed additional mutations in 23 genes previously implicated in intellectual disability or related neurological disorders, as well as single, probably disease-causing variants in 50 novel candidate genes. Proteins encoded by several of these genes interact directly with products of known intellectual disability genes, and many are involved in fundamental cellular processes such as transcription and translation, cell-cycle control, energy metabolism and fatty-acid synthesis, which seem to be pivotal for normal brain development and function.

MeSH Terms
Brain/metabolism,physiology Cell Cycle Cognition Disorders/genetics Consanguinity DNA Mutational Analysis Exons/genetics Gene Regulatory Networks Genes, Essential/genetics Genes, Recessive/genetics High-Throughput Nucleotide Sequencing Homozygote Humans Intellectual Disability/genetics Metabolic Networks and Pathways Mutation/genetics Organ Specificity Synapses/metabolism
Authors & Affiliations
45 authors, click to expand affiliations / ORCID
Najmabadi Hossein
Genetics Research Center, University of Social Welfare and Rehabilitation Sciences, 19857 Tehran, Iran.
Hu Hao
Garshasbi Masoud
Zemojtel Tomasz
Abedini Seyedeh Sedigheh
Chen Wei
Hosseini Masoumeh
Behjati Farkhondeh
Haas Stefan
Jamali Payman
Zecha Agnes
Mohseni Marzieh
Püttmann Lucia
Vahid Leyla Nouri
Jensen Corinna
Moheb Lia Abbasi
Bienek Melanie
Larti Farzaneh
Mueller Ines
Weissmann Robert
Darvish Hossein
Wrogemann Klaus
Hadavi Valeh
Lipkowitz Bettina
Esmaeeli-Nieh Sahar
Wieczorek Dagmar
Kariminejad Roxana
Firouzabadi Saghar Ghasemi
Cohen Monika
Fattahi Zohreh
Rost Imma
Mojahedi Faezeh
Hertzberg Christoph
Dehghan Atefeh
Rajab Anna
Banavandi Mohammad Javad Soltani
Hoffer Julia
Falah Masoumeh
Musante Luciana
Kalscheuer Vera
Ullmann Reinhard
Kuss Andreas Walter
Tzschach Andreas
Kahrizi Kimia
Ropers H Hilger
References (43)
43 references, click to expand
  1. Mutations in GDI1 are responsible for X-linked non-specific mental retardation.
    Nat Genet. 1998 Jun;19(2):134-9 PMID: 9620768
  2. Array CGH identifies reciprocal 16p13.1 duplications and deletions that predispose to autism and/or mental retardation.
    Hum Mutat. 2007 Jul;28(7):674-82 PMID: 17480035
  3. Oligophrenin-1 encodes a rhoGAP protein involved in X-linked mental retardation.
    Nature. 1998 Apr 30;392(6679):923-6 PMID: 9582072
  4. A novel interaction between adrenergic receptors and the alpha-subunit of eukaryotic initiation factor 2B.
    J Biol Chem. 1997 Aug 1;272(31):19099-102 PMID: 9235896
  5. Homozygosity mapping in consanguineous families reveals extreme heterogeneity of non-syndromic autosomal recessive mental retardation and identifies 8 novel gene loci.
    Hum Genet. 2007 Mar;121(1):43-8 PMID: 17120046
  6. Mutation of the conserved polyadenosine RNA binding protein, ZC3H14/dNab2, impairs neural function in Drosophila and humans.
    Proc Natl Acad Sci U S A. 2011 Jul 26;108(30):12390-5 PMID: 21734151
  7. Adaptor protein complex 4 deficiency causes severe autosomal-recessive intellectual disability, progressive spastic paraplegia, shy character, and short stature.
    Am J Hum Genet. 2011 Jun 10;88(6):788-795 PMID: 21620353
  8. Exome sequencing identifies MLL2 mutations as a cause of Kabuki syndrome.
    Nat Genet. 2010 Sep;42(9):790-3 PMID: 20711175
  9. Exome sequencing identifies the cause of a mendelian disorder.
    Nat Genet. 2010 Jan;42(1):30-5 PMID: 19915526
  10. Folate receptor alpha defect causes cerebral folate transport deficiency: a treatable neurodegenerative disorder associated with disturbed myelin metabolism.
    Am J Hum Genet. 2009 Sep;85(3):354-63 PMID: 19732866
  11. Mutations in the ZNF41 gene are associated with cognitive deficits: identification of a new candidate for X-linked mental retardation.
    Am J Hum Genet. 2003 Dec;73(6):1341-54 PMID: 14628291
  12. A de novo paradigm for mental retardation.
    Nat Genet. 2010 Dec;42(12):1109-12 PMID: 21076407
  13. A genetic screen identifies the Triple T complex required for DNA damage signaling and ATM and ATR stability.
    Genes Dev. 2010 Sep 1;24(17):1939-50 PMID: 20810650
  14. Mutations in the JARID1C gene, which is involved in transcriptional regulation and chromatin remodeling, cause X-linked mental retardation.
    Am J Hum Genet. 2005 Feb;76(2):227-36 PMID: 15586325
  15. Identification of a nonsense mutation in the very low-density lipoprotein receptor gene (VLDLR) in an Iranian family with dysequilibrium syndrome.
    Eur J Hum Genet. 2008 Feb;16(2):270-3 PMID: 18043714
  16. A splice site mutation in the methyltransferase gene FTSJ1 in Xp11.23 is associated with non-syndromic mental retardation in a large Belgian family (MRX9).
    J Med Genet. 2004 Sep;41(9):679-83 PMID: 15342698
  17. Identification of mutations in TRAPPC9, which encodes the NIK- and IKK-beta-binding protein, in nonsyndromic autosomal-recessive mental retardation.
    Am J Hum Genet. 2009 Dec;85(6):909-15 PMID: 20004765
  18. The novel BTB/POZ and zinc finger factor Zbtb45 is essential for proper glial differentiation of neural and oligodendrocyte progenitor cells.
    Cell Cycle. 2010 Dec 15;9(24):4866-75 PMID: 21131782
  19. A PEX6-defective peroxisomal biogenesis disorder with severe phenotype in an infant, versus mild phenotype resembling Usher syndrome in the affected parents.
    Am J Hum Genet. 2002 Apr;70(4):1062-8 PMID: 11873320
  20. Mutations in the alpha 1,2-mannosidase gene, MAN1B1, cause autosomal-recessive intellectual disability.
    Am J Hum Genet. 2011 Jul 15;89(1):176-82 PMID: 21763484
  21. A defect in the TUSC3 gene is associated with autosomal recessive mental retardation.
    Am J Hum Genet. 2008 May;82(5):1158-64 PMID: 18452889
  22. Whole-genome sequencing in a patient with Charcot-Marie-Tooth neuropathy.
    N Engl J Med. 2010 Apr 1;362(13):1181-91 PMID: 20220177
  23. Mutational analysis of CACNA1G in idiopathic generalized epilepsy. Mutation in brief #962. Online.
    Hum Mutat. 2007 May;28(5):524-5 PMID: 17397049
  24. The RNA polymerase III transcription apparatus.
    J Mol Biol. 2001 Jun 29;310(1):1-26 PMID: 11419933
  25. MutationTaster evaluates disease-causing potential of sequence alterations.
    Nat Methods. 2010 Aug;7(8):575-6 PMID: 20676075
  26. A systematic, large-scale resequencing screen of X-chromosome coding exons in mental retardation.
    Nat Genet. 2009 May;41(5):535-43 PMID: 19377476
  27. Mutations in SYNGAP1 in autosomal nonsyndromic mental retardation.
    N Engl J Med. 2009 Feb 5;360(6):599-605 PMID: 19196676
  28. KIF7 mutations cause fetal hydrolethalus and acrocallosal syndromes.
    Nat Genet. 2011 Jun;43(6):601-6 PMID: 21552264
  29. A nullimorphic ERLIN2 mutation defines a complicated hereditary spastic paraplegia locus (SPG18).
    Neurogenetics. 2011 Nov;12(4):333-6 PMID: 21796390
  30. The genetic basis of non-syndromic intellectual disability: a review.
    J Neurodev Disord. 2010 Dec;2(4):182-209 PMID: 21124998
  31. Disruption of PC1/3 expression in mice causes dwarfism and multiple neuroendocrine peptide processing defects.
    Proc Natl Acad Sci U S A. 2002 Aug 6;99(16):10293-8 PMID: 12145326
  32. Genetics of early onset cognitive impairment.
    Annu Rev Genomics Hum Genet. 2010;11:161-87 PMID: 20822471
  33. Mutations in the FTSJ1 gene coding for a novel S-adenosylmethionine-binding protein cause nonsyndromic X-linked mental retardation.
    Am J Hum Genet. 2004 Aug;75(2):305-9 PMID: 15162322
  34. A defect in the ionotropic glutamate receptor 6 gene (GRIK2) is associated with autosomal recessive mental retardation.
    Am J Hum Genet. 2007 Oct;81(4):792-8 PMID: 17847003
  35. Excess of de novo deleterious mutations in genes associated with glutamatergic systems in nonsyndromic intellectual disability.
    Am J Hum Genet. 2011 Mar 11;88(3):306-16 PMID: 21376300
  36. SRD5A3 is required for converting polyprenol to dolichol and is mutated in a congenital glycosylation disorder.
    Cell. 2010 Jul 23;142(2):203-17 PMID: 20637498
  37. Next generation sequencing in a family with autosomal recessive Kahrizi syndrome (OMIM 612713) reveals a homozygous frameshift mutation in SRD5A3.
    Eur J Hum Genet. 2011 Jan;19(1):115-7 PMID: 20700148
  38. Autosomal recessive mental retardation: homozygosity mapping identifies 27 single linkage intervals, at least 14 novel loci and several mutation hotspots.
    Hum Genet. 2011 Feb;129(2):141-8 PMID: 21063731
  39. Human CNK1 acts as a scaffold protein, linking Rho and Ras signal transduction pathways.
    Mol Cell Biol. 2004 Feb;24(4):1736-46 PMID: 14749388
  40. The role of neuronal complexes in human X-linked brain diseases.
    Am J Hum Genet. 2007 Feb;80(2):205-20 PMID: 17236127
  41. A frameshift mutation of ERLIN2 in recessive intellectual disability, motor dysfunction and multiple joint contractures.
    Hum Mol Genet. 2011 May 15;20(10):1886-92 PMID: 21330303
  42. Neonatal hepatic steatosis by disruption of the adenosine kinase gene.
    Proc Natl Acad Sci U S A. 2002 May 14;99(10):6985-90 PMID: 11997462
  43. Role of voltage-gated calcium channels in epilepsy.
    Pflugers Arch. 2010 Jul;460(2):395-403 PMID: 20091047
Article Info
Journal
Nature
Abbr.
Nature
ISSN
1476-4687
Published
2011-09-21
Epub
2011-00-21
Pages
57-63
Language
English
Region
England
NLM ID
0410462
Subset
IM
Corrections
CommentIn
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: product@genelibs.com