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Mutations in the pericentrin (PCNT) gene cause primordial dwarfism.

Authors :
Rauch A
Thiel CT
Schindler D
Wick U
Crow YJ
Ekici AB
van Essen AJ
Goecke TO
Al-Gazali L
Chrzanowska KH
Zweier C
Brunner HG
Becker K
Curry CJ
Dallapiccola B
Devriendt K
Dörfler A
Kinning E
Megarbane A
Meinecke P
Semple RK
Spranger S
Toutain A
Trembath RC
Voss E
Wilson L
Hennekam R
de Zegher F
Dörr HG
Reis A
Source :
Science (New York, N.Y.) [Science] 2008 Feb 08; Vol. 319 (5864), pp. 816-9. Date of Electronic Publication: 2008 Jan 03.
Publication Year :
2008

Abstract

Fundamental processes influencing human growth can be revealed by studying extreme short stature. Using genetic linkage analysis, we find that biallelic loss-of-function mutations in the centrosomal pericentrin (PCNT) gene on chromosome 21q22.3 cause microcephalic osteodysplastic primordial dwarfism type II (MOPD II) in 25 patients. Adults with this rare inherited condition have an average height of 100 centimeters and a brain size comparable to that of a 3-month-old baby, but are of near-normal intelligence. Absence of PCNT results in disorganized mitotic spindles and missegregation of chromosomes. Mutations in related genes are known to cause primary microcephaly (MCPH1, CDK5RAP2, ASPM, and CENPJ).

Details

Language :
English
ISSN :
1095-9203
Volume :
319
Issue :
5864
Database :
MEDLINE
Journal :
Science (New York, N.Y.)
Publication Type :
Academic Journal
Accession number :
18174396
Full Text :
https://doi.org/10.1126/science.1151174