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PMID: 12768443 Published · ppublish English Comparative Study Journal Article Research Support, Non-U.S. Gov't

LMNA is mutated in Hutchinson-Gilford progeria (MIM 176670) but not in Wiedemann-Rautenstrauch progeroid syndrome (MIM 264090).

Journal of human genetics ·Vol. 48 ·No. 5 ·2003-00-00 ·Pages 271-274

Cao H, Hegele RA

Abstract

Hutchinson-Gilford progeria syndrome (HGPS; MIM 176670) is an extremely rare disease that is characterized by accelerated aging and early death, frequently from coronary artery disease. Wiedemann-Rautenstrauch syndrome (WRS; MIM 264090) is another extremely rare disease that is characterized by progeroid features from birth with multiple somatic anomalies and paucity of subcutaneous fat. Because mutations in LMNA, encoding nuclear lamin A/C, cause other lipodystrophy syndromes, we sequenced LMNA (MIM 150330) from the genomic DNAs of seven unrelated HGPS probands and two unrelated WRS probands. We found four novel LMNA coding sequence variants among the HGPS probands, namely R471C, R527C, G608S and c.2036C>T. All seven HGPS probands had at least one LMNA variant, which were found in none of the genomes of 100 normal subjects ( P<4 x 10(-11)). In contrast, neither of the WRS proband genomes had any LMNA sequence abnormality. The strong association of rare LMNA coding sequence mutations with HGPS implicates this syndrome as a laminopathy, while WRS is most probably due to mutations in another gene.

MeSH Terms
Adolescent Adult Cell Line Child Child, Preschool Female Fibroblasts/metabolism Genetic Testing Genetic Variation Humans Infant Infant, Newborn Lamin Type A/genetics Male Mutation Progeria/genetics,metabolism Sequence Analysis, DNA
Chemicals
Lamin Type A lamin C
Authors & Affiliations
2 authors, click to expand affiliations / ORCID
Cao Henian
Blackburn Cardiovascular Genetics Laboratory, Robarts Research Institute, 406-100 Perth Drive, London, Ontario, N6A 5K8, Canada.
Hegele Robert A
Blackburn Cardiovascular Genetics Laboratory, Robarts Research Institute, 406-100 Perth Drive, London, Ontario, N6A 5K8, Canada. [email protected].
References (19)
19 references, click to expand
  1. An unidentified neonatal progeroid syndrome: follow-up report.
    Eur J Pediatr. 1979 Jan 18;130(1):65-70 PMID: 569581
  2. Recurrent de novo point mutations in lamin A cause Hutchinson-Gilford progeria syndrome.
    Nature. 2003 May 15;423(6937):293-8 PMID: 12714972
  3. Hutchinson-Gilford progeria: familial occurrence.
    Am J Med Genet. 1990 Aug;36(4):431-3 PMID: 2389799
  4. Severe bone changes in a case of Hutchinson-Gilford syndrome.
    Ann Genet. 2002 Jul-Sep;45(3):151-5 PMID: 12381448
  5. Del(1)(q23) in a patient with Hutchinson-Gilford progeria.
    Am J Med Genet. 2002 Dec 1;113(3):298-301 PMID: 12439901
  6. Life at the edge: the nuclear envelope and human disease.
    Nat Rev Mol Cell Biol. 2002 Aug;3(8):575-85 PMID: 12154369
  7. Wiedemann-Rautenstrauch syndrome.
    J Med Genet. 1990 Apr;27(4):256-7 PMID: 2325106
  8. Wiedemann-Rautenstrauch neonatal progeroid syndrome: report of three new patients.
    J Med Genet. 1997 May;34(5):433-7 PMID: 9152846
  9. The Wiedemann-Rautenstrauch or neonatal progeroid syndrome. Neuropathological study of a case.
    Neuropediatrics. 1984 Feb;15(1):43-8 PMID: 6200796
  10. Two sibs with Wiedemann-Rautenstrauch syndrome: possibilities of prenatal diagnosis by ultrasound.
    J Med Genet. 1992 Jun;29(6):434-6 PMID: 1619643
  11. The Wiedemann-Rautenstrauch neonatal progeroid syndrome: a case report and review of the literature.
    Clin Dysmorphol. 1995 Jul;4(3):239-45 PMID: 7551161
  12. Nuclear lamin A/C R482Q mutation in canadian kindreds with Dunnigan-type familial partial lipodystrophy.
    Hum Mol Genet. 2000 Jan 1;9(1):109-12 PMID: 10587585
  13. Progeria: a cell culture study and clinical report of familial incidence.
    Eur J Pediatr. 1977 Jan 26;124(2):101-11 PMID: 319005
  14. The Wiedemann-Rautenstrauch or neonatal progeroid syndrome. Report of a patient with consanguineous parents.
    Eur J Pediatr. 1981 Jul;136(3):245-8 PMID: 7262096
  15. Neonatal progeroid (Wiedemann-Rautenstrauch) syndrome: report of five new cases and review.
    Am J Med Genet. 2000 Jan 17;90(2):131-40 PMID: 10607952
  16. Progeria, a pathologic study.
    J Pediatr. 1960 Jul;57:70-7 PMID: 13825821
  17. Mutations in the gene encoding lamin A/C cause autosomal dominant Emery-Dreifuss muscular dystrophy.
    Nat Genet. 1999 Mar;21(3):285-8 PMID: 10080180
  18. Mandibuloacral dysplasia is caused by a mutation in LMNA-encoding lamin A/C.
    Am J Hum Genet. 2002 Aug;71(2):426-31 PMID: 12075506
  19. Case of Progeria.
    Proc R Soc Med. 1923;16(Sect Study Dis Child):42 PMID: 19983361
Article Info
Journal
Journal of human genetics
Abbr.
J Hum Genet
ISSN
1434-5161
Published
2003-00-00
Epub
2003-00-03
Pages
271-274
Language
English
Region
England
NLM ID
9808008
Subset
IM
Databases
OMIM
150330, 176670, 248370, 264090
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